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uncontrolled bleeding remains major challenge , responsible for 40% trauma - related deaths , which could be prevented by timely intervention . in blunt trauma , extent of hemorrhage can not be determined by physical examination , as vital signs may not give a clear picture in all the patients , especially young healthy once . although abnormal vital signs does indicate shock but the absence of abnormality does not exclude hypoperfusion in trauma patients . decision - making is difficult , regarding surgical intervention to treat injuries hidden from physical examination . these patients are intensely vasoconstricted and may suffer end organ ischemia even with normal systolic blood pressure ( sbp ) . although shock index ( si ) can be a useful parameter in acute hemorrhage , but it still needs more study in its support to be better than simple vital sign analysis . blood ammonia has been shown to be elevated in hemorrhagic shock in animals and humans . intestinal bacterial enzymes acting on it produces ammonia which is freely diffusible and moreover liver in not fit for detoxification of ammonia . we investigated the significance of ammonia levels estimation in blunt trauma abdomen patients to predict internal hemorrhage , complications and correlated with need of intervention . one hundred blunt trauma abdomen patients ( > 12 years old ) presented in trauma ward / emergency department of tertiary care hospital were included in the study . group i required blood transfusion 2 units and/or intervention to control bleeding within 24 h following admission . it was analyzed within 20 min of sampling on autoanalyser cobas 6000 ( roche diagnostics india pvt ltd ) . routine investigations such as blood sugar , kidney function test , hemogram , x - ray chest , and ultrasound abdomen were done at admission . group i comprised 62 patients , forty patients needed intervention and blood transfusion both , whereas 22 patients needed only 2 units of blood . average age was 31.84 14.8 and 32.37 13.6 years , respectively , almost same in both the groups . most of the patients had more than one organ system involved and most frequently injured organ was liver ( 40% ) followed by spleen ( 29% ) . mean si and arterial ammonia levels were significantly higher in group i compared to group ii patients [ table 1 ] . about 88.7% patient of group i had si > 0.9 compared to only 13.1% patients in group ii [ table 2 ] . significantly higher number of patients developed complications in group i compared to group ii ( 38 vs. 7 ) and 8% mortality rate was noted in group i only . patients , who developed complications or died , had higher levels of ammonia at admission [ table 3 and figure 1 ] . mean shock index and ammonia levels percentage of patients showing shock index 0.9/0.9 ammonia levels in patients with complications and mortality in group i receiver operating characteristic analysis of hospital stay , shock index , and ammonia levels the cutoff point for ammonia levels is > 58.85 mol / l ( sensitivity 77.42% and specificity 93.37% ) . positive predictive value and negative predictive value at this point are 96.7% and 78.9% , respectively . the cutoff point for ammonia levels is > 58.85 mol / l ( sensitivity 77.42% and specificity 93.37% ) . positive predictive value and negative predictive value at this point are 96.7% and 78.9% , respectively . roadside accidents were a major cause of trauma ( motor vehicle injury ) in our study probably due to increase population and number of vehicles on the road . similar cause has been reported by other studies also . a maximum number of patients ( 78% ) were below the age of 40 years suggesting this age group people are more outgoing and rash drivers making them more prone to accidents . male preponderance ( 93% ) noted in our study is higher than earlier studies , may be reflecting indian culture where male member of the family being breadwinner , needs to travel more than females . the routine evaluation of blunt trauma abdomen patients includes various clinical parameters , physical examination , radiological and laboratory findings . increased ammonia levels as observed in our study could itself be danger signal in these patients as it is sign of blood in abdominal cavity due to blunt trauma . intestinal bacterial enzymes acting on it produces ammonia which is freely diffusible and moreover liver in not fit for its detoxification as hemorrhage decreases hepatic blood flow through portal vein causing dysoxia of cells . increased ammonia levels have been reported in animal hemorrhagic shock and in few human studies as well . cutoff value for ammonia was also calculated to maximize sensitivity and specificity in identifying patients requiring intervention . the purpose of this study was to augment the diagnostic accuracy of routine clinical assessment of such patient . high ammonia levels in bleeding patients ( group i ) correlated well with si as 88.7% of these patients had si > 0.9 , i.e. , having potentially severe trauma requiring immediate treatment . other workers have also reported similar si in bleeding patients . although si can be useful clinical parameter in acute hemorrhage which depicts severity of situation in heterogeneous patients presented in emergency but it still needs more study in its support to be single reliable parameter . five patients died in group i had high ammonia levels ( 87.4 mol / l ) at admission . high ammonia itself is toxic and can cause various complications , so it must be diagnosed and treated as early as possible . moreover , trauma patient loses lots of valuable time in reaching trauma center , but once reached the hospital , they need to be diagnosed correctly and quickly . biochemical marker which is time and cost effective such as ammonia estimation can predict need of intervention in these patients and best treatment can be provided in the golden hour .

background : in blunt trauma , extent of hemorrhage can not be determined by physical examination , and vital signs may also not give clear picture in all the patients , especially young healthy ones . hemorrhagic shock has been reported to increase blood ammonia levels . arterial ammonia was analyzed in blunt trauma abdomen patients and correlated with shock index ( si ) . its predictive value was determined for timely decision of intervention.materials and methods : hundred blunt trauma abdomen patients presented in the emergency ward of tertiary care hospital were included in the study . group i comprised 62 patients requiring either blood transfusion 2 units and/or intervention to control bleeding within 24 h following admission . group ii had 38 patients : not requiring transfusion / intervention during hospital stay . arterial blood sample was taken immediately after admission ; ammonia was analyzed within 20 min of sampling on cobas 6000 ( roche ) . si was calculated . predictive value of ammonia was determined using receiver operating characteristic curve.results:ammonia levels and si were significantly ( p < 0.001 ) higher in group i compared to group ii patients ( 68.55 14.36 umol / l vs. 37.55 7.41 umol / l and 1.28 0.5 vs. 0.74 0.12 , respectively ) . significantly higher number of patients in group i ( 88.7% vs. 13% ) had si > 0.9 . ammonia levels were significantly higher in patients with complications and in those expired.conclusions:ammonia levels were significantly higher in patients requiring blood transfusion / intervention in 24 h of admission . the best cutoff value to maximize sensitivity and specificity was ammonia > 58.85 mol / l . ammonia estimation at admission can be clinically significant indicator of traumatic hemorrhage needing intervention .

darier 's disease is an autosomal dominant disorder of keratinization affecting the skin , mucosa , and nails that typically manifests in the first and second decade of life . we describe an elderly man with nonmetastatic gastric adenocarcinoma presenting with an acquired variant of darier 's disease presumably of paraneoplastic origin . a 62-year - old muslim farmer was in otherwise good health until 2 months back when he suddenly started noticing some dark - colored , mildly itchy skin eruption . he also complained of nausea , vomiting , and loss of appetite and a single episode of passage of black , tarry stool . on examination of his skin , numerous hyperpigmented , warty papules were seen over the face , neck , trunk , and scalp . the lesions were characteristically distributed in seborrheic areas like head neck area , back , and front of chest [ figure 1 ] . there was punctate keratoderma of palms and soles with a few palmar pits as well [ figure 2 ] . v-like notch at the free edge of the nail plate without longitudinal streaks . on the basis of these clinical features , there was neither a past history of similar lesions nor any family history of the same . dirty , warty papules over the back palms showing pits and keratoderma histopathology of the skin lesions revealed suprabasal acantholytic cleft and dyskeratotic cells like corps ronds and grains [ figure 3a and b ] . diminution of lesions following tumor resection ( a ) on back ; ( b ) on chest and palm he was referred to the gastroenterology department where after a series of investigations including endoscopic biopsy , he was diagnosed with gastric adenocarcinoma . on his next follow - up in our outpatient department ( opd ) 2 months later , we discovered that the skin lesions had remarkably diminished [ figure 4a and b ] . by that time it was then that we retrospectively considered darier 's disease to be a paraneoplastic phenomenon . histopathology showing ( a ) prominent hyperkeratosis and suprabasal acantholytic cleft with villi ( h and e , 100 ) ; ( b ) dyskeratotic cells ( h and e , 400 ) paraneoplastic dermatoses describes those benign skin changes in which there is a direct , often parallel course of a dermatosis with an underlying malignancy . there are several proposed hypotheses on the pathomechanism of paraneoplastic dermatoses , which highlight the role of various tumor - derived growth factors like transforming growth factor ( tgf- ) , fibroblast growth factor , and so on . regarding the pathogenesis of paraneoplastic darier 's disease , it can be speculated that the tumor product interferes with keratinocyte calcium homeostasis leading to disturbances in intracellular trafficking of desmosomal proteins . the autosomal dominant variety is due to mutations in the serca2 ( sarco / endoplasmic reticulum calcium atpase type 2 ) , which regulates calcium homoestasis in the endoplasmic reticulum . our case involves an acquired form of darier 's disease whose diagnosis was supported both clinically and histopathologically . although there are several variants of darier 's disease , the paraneoplastic variety is extremely rare . there is only a single case report on its association with metastatic papillary carcinoma of thyroid . there are some sporadic reports on the occurrence of squamous cell carcinoma of skin , nail bed , esophagus , mouth , and vagina in patients with pre - existing darier 's disease but paraneoplastic association is very rare . according to the criteria proposed by helen ollendorff curth , a specific dermatosis occurs with a specific neoplasm , and a high percentage of association between two conditions is noted . this does not hold true in our case . the paraneoplastic dermatoses that have been reportedly associated with gastric adenocarcinoma are acanthosis nigricans , tripe palms , and florid cutaneous papillomatoses . we consider this a paraneoplastic process due to its concurrent onset and almost parallel course with malignant neoplasm , which are also important requisites in the curth criteria . moreover , in this case , onset of darier 's disease was late , occurring in the seventh decade of life in contrast to its usual onset in the second decade of life . also the skin changes resolved with treatment of the underlying cancer . this further served as an eye - opener for the clinical investigators of this case . its paraneoplastic association with gastric adenocarcinoma is very unusual and not known to be mentioned in any dermatology literature so far and hence reported here . darier 's disease is an unusual paraneoplastic manifestation in gastric malignancy , not reported before .

darier 's disease is an autosomal dominant genodermatosis resulting from atp2a2 gene mutation . a 62-year - old male presented at our outpatient ( opd ) with sudden - onset numerous dirty , warty papules over the head , neck , and back since 2 months . histopathology of the skin lesions revealed acantholytic dyskeratosis suggestive of darier 's disease . he was referred to the gastroenterology department for some gastrointestinal ( gi ) symptoms where he was diagnosed with adenocarcinoma of the stomach and was subsequently operated . on his next visit to our department for follow - up , we found a marked diminution of the skin lesions in the absence of any specific treatment . in view of the above finding , we concluded that paraneoplastic dermatosis in the form of darier 's disease occurred in this patient . paraneoplastic darier 's disease with gastric adenocarcinoma is not yet described in dermatology literature and is hence reported here .

recently , serratia , pseudomonas / providencia , indole - positive proteus / acinetobacter / morganella , citrobacter , enterobacter and hafnia group of organisms ( spice ) were described to cause peritoneal dialysis ( pd)-related peritonitis with a particularly high morbidity and mortality . peritonitis caused by citrobacter is relatively uncommon and citrobacter freundii is the most common species involved . we report a case of pd - related peritonitis caused by c. freundii , which was successfully treated with double antibiotic coverage . a 76-year - old male with end stage renal disease due to type ii diabetes mellitus on pd for 6 months presented to the emergency room with abdominal pain and bloody peritoneal dialysate for 1 day . he was on regular maintenance automated pd using a cycler and reported no history of breakdown of aseptic technique or contamination during the catheter care . upon examination , he was hypotensive with a blood pressure of 98/54 mm hg but afebrile and the catheter exit site was benign . a dialysate cell count and culture was obtained and the patient was started on empirical intravenous broad - spectrum antibiotic coverage with vancomycin and aztreonam ( he was allergic to penicillin ) . pd was continued during the hospital stay with the addition of 500 units of unfractionated heparin per liter of dialysate . his dialysate white blood cell count was > 3700 with 92% neutrophils and c. freundii was isolated upon its culture . a computed tomography scan of the abdomen showed no evidence of intestinal perforation , appendicitis or diverticulitis . subsequently , his clinical condition stabilized with subsiding abdominal pain clearing of dialysate in 2 days . his antibiotic coverage was switched to oral ciprofloxacin along with intraperitoneal gentamycin , requiring an additional daytime manual exchange with 6-h dwell time in order to provide sustained antibiotic exposure to the infected peritoneal membrane , which is not possible with the cycler - assisted pd exchanges . the antibiotics were continued for a total duration of 14 days , and he was discharged in a stable condition . peritonitis caused by the members of the family enterobacteriaceae is an important cause of mortality and morbidity in pd patients and accounts for up to 12% of all peritonitis episodes in some series . citrobacter species , a part of this family , were not commonly associated with peritonitis until recently , when they were described among the spice organisms causing severe peritonitis . most commonly , c. freundii has been associated with urinary tract infections , superficial wound infections and bacteremia especially in elderly , immunocompromised and hospitalized patients . it colonizes the gastrointestinal tract of humans and other animals and its translocation to the blood stream in dialysis patients especially in the setting of abnormal bowel habits is implicated in the development of peritonitis . our patient had a particularly severe peritonitis with hemodynamic instability , and prompt use of intravenous broad spectrum antibiotics was life - saving . we encountered a bacterial strain that was resistant to ampicillin ; a commonly described property of c. freundii and ascribed to the ampc gene , which provides high - level resistance to ampicillin and first generation cephalosporins . indeed , the last published guidelines of the international society of pd in 2010 recommend following the sensitivity patterns in case of spice organisms and consider using double antibiotic coverage for 23 weeks . earlier literature reports a high incidence of mortality and morbidity from catheter losses from pd - related peritonitis caused by c. freundii . prompt institution of broad antimicrobial coverage in unstable patients and following the culture sensitivity pattern reduces mortality and catheter losses from peritonitis caused by c. freundii .

serratia , pseudomonas / providencia , indole - positive proteus / acinetobacter / morganella , citrobacter , enterobacter and hafnia group of organisms cause peritoneal dialysis ( pd)-related peritonitis with high morbidity and mortality . peritonitis caused by citrobacter freundii is uncommon , and it may lead to catheter removal despite antimicrobial treatment . we describe a case of pd - related peritonitis caused by c. freundii , which was successfully treated with double antibiotic coverage .

sepsis has been classically considered the archetypal clinical condition with molecular links between inflammation and coagulation . both inflammation and thrombosis can be orchestrated by the interactions between circulating cells , such as lymphocytes , platelets , and vascular cells , which under activation or apoptosis lead to the release of circulating microparticles ( mps ) . in the previous issue of critical care , prez - casal and colleagues hypothesized that circulating mps may retain their anti - inflammatory and cytoprotective properties in septic patients during recombinant human activated protein c ( rhapc ) infusion in vivo and probably participate in its clinical benefit . the same group has previously shown that activated protein c ( apc ) can generate mps in vitro from endothelial cell protein c receptor ( epcr)-expressing cells , which retain anticoagulant and protease - activated receptor-1 ( par-1)-dependent anti - inflammatory properties . apc binding to epcr at the endothelial cell surface and apc on mp - epcr could cleave and activate par-1 , sphingosine 1-phosphate receptor and kinase insert domain receptor . rhapc treatment for severe sepsis can induce the generation and release of mps in vivo , with a clinical correlative trend towards improved outcome . circulating mps from patients during rhapc treatment express apc , epcr and cd13 . these mps interact with endothelial cells and induce changes in gene expression to inhibit apoptosis and reduce endothelial permeability . these effects require par-1 activation by apc in an epcr - bound conformation , confirming the evidence for the assembled epcr - apc complex on in vivo - derived mps . the present work suggests that mps could disseminate apc function and activate endothelial par-1 at distal vascular sites . the mps represent an additional circulatory form of apc receptor in human plasma , which is different from soluble epcr . mp - associated apc is stable in measurable levels , and activities would point to physiological and clinical relevance as bioactive effectors in rhapc - treated patients and contribute to the effectiveness of rhapc in severe sepsis the authors did not analyze the subpopulation of cd13 mps upon rhapc treatment , whether they are from endothelial or leukocyte origins . indeed , they have shown that apc induces mp - associated epcr formation from monocytes and human endothelial cells . furthermore , whether such mps contribute to the ability of rhapc treatment to improve cardiovascular function - including arterial contractility and endothelial dysfunction by decreasing tissue inflammation and oxidative stress as reported in an experimental model of sepsis - remains to be determined . a fragile balance between the harmful and helpful effects of mps especially during severe sepsis should be underlined . circulating mps from septic patients might exert a protective role at the vascular level by compensating hyporeactivity , but they might also contribute to the cause of multiorgan failure in sepsis and induce deleterious protein changes in target tissues . it would be of interest to test the hypothesis that rhapc via increased certain subtypes of mps bearing epcr / apc would contribute to the correction of multiple organ failure , which would lead to increased survival . the clinical relevance of rhapc treatment via increased apc / epcr - mps requires further exploration , especially in larger numbers of patients with septic shock and higher mortality . in conclusion mps could potentially be developed as new therapeutic tools to transfer biological vectors of cellular communication and are able to modulate important cellular regulatory functions at a distal site of its production in response to pharmacological agents such as rhapc . apc : activated protein c ; epcr : endothelial protein c receptor ; mp : microparticle ; par-1 : protease - activated receptor-1 ; rhapc : recombinant human apc .

activated protein c ( apc ) , a physiological coagulation inhibitor , has been shown to reduce mortality in patients with severe sepsis . apc exerts pleiotropic cytoprotection by a mechanism that requires its interaction with endothelial cell protein c receptor and protease - activated receptor-1 on target cells . in the previous issue , prez - casal and colleagues elegantly demonstrate that apc , using its recombinant form ( rhapc ) , can communicate to target cells through release of microparticles ( mps ) , small membrane vesicles released from activated cells , to induce anti - apoptotic and anti - inflammatory properties that might participate in the improvement of patient outcome . of interest is the fact that apc itself promotes the release of mps from target cells including endothelial cells and monocytes . these mps bear the endothelial cell protein c receptor / apc molecules and can transfer the message to target cells including those of origin to induce cytoprotection . the long - range apc signal can thus be mediated by mps in vivo upon pharmacological treatment using rhapc in severe septic patients . a novel pharmacological approach targeting mp production and properties could therefore be used to treat severe sepsis in addition to other well - known actions of apc via direct interaction with the cells of interest .

diastolic mitral regurgitation has been described in patients with acute aortic regurgitation and those with atrioventricular block or atrial fibrillation with slow ventricular responses . evidence of diastolic mitral regurgitation in these patients has been demonstrated previously by left ventriculography and recently by pulse doppler echocardiography . the mechanisms of diastolic mitral regurgitation in patients with acute aortic regurgitation have been considered to be the summation of the following pathophysiology : reversed atrio - ventricular pressure gradient due to aortic regurgitation in the non - compliant ventricle , increased mitral annulus due to ventricular relaxation and lack of papillary muscle tension . that of the patients with atrioventricular block or atrial fibrillation has been considered to be lack of atrial factors for mitral valve closure . we documented diastolic mitral regurgitation by color doppler echocardiography in a patient with acute aortic regurgitation . color doppler flow imaging is highly useful in detecting valvular regurgitation , especially in determining flow direction . therefore , this technique may provide additional information regarding the mechanism by which diastolic mitral regurgitation is produced . reversibility of diastolic mitral regurgitation after correction of hemodynamic loading , that was widely believed but not actually demonstrated , was also observed . a 38-year - old japanese man with no previous cardiovascular disease was admitted to our hospital because of paroxysmal nocturnal dyspnea and low grade fever of four weeks duration . he had the history of the resection of subcutaneous abscess in the neck , several days before the onset of symptom . on admission , an early systolic murmur and a diastolic blowing murmur with thrill were detected at the left parasternal border . increased pulmonary vascular marking was observed in his chest x - ray and the cardio - thoracic ratio was 50% . electrocardiogram revealed a prolonged p - q interval of 0.28 second , left ventricular hypertrophy and st depression in the left precordial leads . after admission , first - degree of atrioventricular block normalized in 0.18 second of p - q interval . echocardiography was performed with a tohshiba 65a after normalization of p - q interval , which revealed the dilated and hyperkinetic left ventricle and the slightly dilated left atrium . 1 ) , and the left ventricular end diastolic dimension was 6.4 cm and the end systolic dimension was 4.4 cm . the right coronary cusp and left coronary cusp of the aortic valve were extremely elongated and the left valsalva sinus was dilated . both aortic cusps were prolapsing and no coaptation was seen . severe aortic regurgitation was observed on color doppler flow imaging . in late diastole , mitral regurgitation which ran just behind the posterior mitral leaflet and extended to the posterior wall of the left atrium was detected ( fig . the diastolic mitral regurgitation began with the atrial systole and disappeared with the ventricular systole ( fig . although several blood cultures were all negative , the diagnosis of acute aortic regurgitation due to infective endocarditis was made from the clinical history and destructive changes of the aortic valve . after intensive antibiotic therapy for three weeks , aortic valve replacement was performed . at surgery , commissural rupture between the right coronary cusp and the left coronary cusp was detected and an abscess was present in the root of the left coronary cusp . diastolic mitral regurgitation in patients with acute aortic reguritation in patients with acute aortic regurgitation is considered to be produced by hemodynamic abnormalities , not by intrinsic abnormalities of mitral leaflets . however , the proof of this consideration is somewhat obscure . in most reported cases with diastolic mitral regurgitation , the concurrent presence of systolic mitral regurgitation , which indicates the presence of intrinsic abnormalities of mitral leaflets or apparatus , was reported . if the mitral valve is truly normal , systolic mitral regurgitation should be absent and diastolic mitral regurgitation should disappear with resolution of the hemodynamic abnormalities . the reversibility of diastolic mitral regurgitation after aortic valve replacement has never been described . in this case , the mitral regurgitation was observed only in the period of late diastole , disappearing with the ventricular systole . furthermore , the diastolic mitral regurgitation in this case could not be detected after the successful aortic valve replacement . our case clearly indicates that acute aortic regurgitation can produce diastolic mitral regurgitation in a normal mitral valve . in this case , the flow of the diastolic mitral regurgitation was directed to the posterior wall of the left atrium through just behind the posterior mitral leaflet . from above mentioned mechanisms of diastolic mitral regurgitation in acute aortic regurgitation , if intrinsic abnormalities of the mitral leaflets are absent , regurgitant flow should be directed to the center of the left atrium . extreme deviation of mitral regurgitant flow is observed in cases of mitral valve prolapse or those with severely restricted mitral valve leaflets , as is seen in rheumatic mitral disease . because this case showed no abnormalities in the mitral leaflets under echocardiography , severely restricted movement of the mitral leaflets is unlikely . we speculated either that the aortic regurgitant jet might depress the anterior mitral leaflet toward the left atrium or that acute left ventricular enlargement due to aortic regurgitation might retract the chordae connecting to the posterior mitral leaflet . these processes would result in the dislodgment of the tips of both leaflets , creating extreme deviation of the mitral regurgitant flow similar to mitral prolapse . there are a few reports on the observation of diastolic mitral regurgitation by color doppler echocardiography , but no comments for the flow direction . this case indicated the possibility of additional mechanisms of diastolic mitral regurgitation , namely , depression of the anterior mitral leaflet or retraction of the chordae connecting to the posterior mitral leaflet .

a 38-year - old man was admitted with coingestive heart failure due to infective endocarditis . echocardiography with color doppler imaging revealed severe aortic regurgitation , mitral valve premature closure and diastolic mitral regurgitation . the flow of the diastolic mitral regurgitation was directed to the posterior wall of the left atrium through just behind the posterior mitral leaflet . the diastolic mitral regurgitation was observed only in the period of late diastole and no mitral regurgitation could be detected in the systolic phase . after successful aortic valve replacement , the diastolic mitral regurgitation disappeared completely .

brucellosis ( malta fever ) is an infectious disease with a wide range of manifestations . there are four types of brucella , b. melitensis , b. abortus , b. canis and b. suis . bacteria are transmitted to humans via the injection of non - pasteurized dairy products , uncooked raw meat or by contact through skin , blood , conjunctiva , gastrointestinal or respiratory tracts.12 although the incidence of brucellosis has declined , it is still remains an important health problem in endemic areas such as the middle east , the mediterranean and asia . iran is considered an endemic country as are peru , saudi arabia , kuwait and turkey.34 according to the report of the diseases prevention and fight department of iran s health center , published in 2009 , the incidence of brucellosis was 25 in 100,000 people . in hamadan , studies show that b. melitensis is the most common and virulent species with a high prevalence in latin america , mediterranean and developing countries . b. abortus is reported mostly in europe and north america . despite the decrease in total prevalence of the disease a 29-year - old male was referred to infectious disease clinic of hamadan sina hospital in summer 2008 with acute onset of fever , headache , malaise , sweating and low back pain . on examination laboratory test findings were : white blood cell ( wbc ) count of 13350 ( neutrophil : 85% ) , erythrocyte sedimentation rate ( esr ) 48m / h , crp , rf , ana , wright s agglutination titer of 1/1280 . blood cultures were negative and electrocardiogram and chest - x - ray ( cxr ) were normal . the patient was diagnosed with brucellosis , and treated with rifampin 600 mg / day and doxycycline 100 mg / twice daily . after a week , the patient was referred for an ophthalmology consult with complaints of ocular pain and redness and visual complaints . on ophthalmic examination , the conjunctional injection was a mixture of ciliary injection , episcleritis and conjunctivitis but more severe in the ciliary area . on funduscopy , there was bilateral optic disc swelling along with retinal hyperemia ( optic disc hyperemia and vascular tortuosity ) and diffuse intraretinal hemorrhage [ figure 1 ] . intraocular pressure ( iop ) was 26 mmhg in the right eye and 24 mmhg in the left eye . the patient was hospitalized with a probable diagnosis of ocular brucellosis and was treated with co - trimoxazole adult two tablets , three times a day ( tid ) , rifampin 600 mg / day doxycycline 100 mg / bid and prednisolone 1 mg / kg for 2 months . computed tomography and magnetic resonance imaging studies of the brain and optic nerve were requested . fever and headache diminished within 48 hours of the treatment however , ophthalmic complaints lingered . bilateral optic disc swelling with retinal hyperemia ( optic disc hyperemia and vascular tortuosity ) and diffuse intraretinal hemorrhage fundoscopic findings 13 months after treatment for brucellosis brucellosis presents with a spectrum of clinical manifestations and diagnosis of this disease is based on clinical signs , positive bacteriological and serological tests . some ocular manifestations including dacryoadenitis , episcleritis , chronic iridoscleritis , nummular keratitis , cataract , glaucoma , multifocal choroiditis , exudative retinal detachment , maculopathy , and optic neuritis.814 rolando et al.14 showed that the most frequent ocular manifestation is uveitis predominantly posterior uveitis . it seems that optic nerve involvement is secondary to meningeal inflammation and flow change of the optic nerve due to axonal degeneration.14 visual improvement of the patient following corticosteroid administration is proof of ischemic or vasculitic involvement.1410 ophthalmic manifestations of brucellosis are not common and acceptable outcomes following treatment with antibiotics and steroids are low.9101517 cavallarro et al.18 reported a patient with papilledema due to brucellosis that was treated with sole anti - brucellosis treatment without steroid administration . abd - elrazak19 reported a case of bilateral optic neuritis caused by brucellosis that resolved following anti - brucellosis and steroid administration . lashay et al.20 from iran reported a case of bilateral optic nerve head swelling following brucellosis , which led to bilateral optic nerve atrophy and visual loss . in our case , antibiotic and steroid administration led to complete visual recovery and in 13 months follow - up after treatment , ophthalmologic examinations were normal . the outcome in our case is likely due to early diagnosis and treatment , lack of a drug resistant strain and better prognosis in males compared to females.142123 in the current case , imaging studies were normal . however , such lesions may be missed on routine imaging studies if magnetic resonance angiography ( mra ) is not performed . considering the rapid response to the therapeutic interventions likewise , other possible causes of conjunctival injection associated with increased venous pressure such as cavernous sinus thrombosis or orbital apex syndromes were ruled out due to this quick and appropriate response to treatment . the prevalence of brucellosis has decreased in many developed countries and ophthalmic complications are rare in these regions , but it is suggested that in endemic areas , routine ophthalmic examination for brucellosis be considered , as it seems that early diagnosis and prompt treatment of the disease could decrease vision - threatening complications.23

a 29-year - old male diagnosed with brucellosis a week earlier was referred to the ophthalmology clinic with visual complaints . on examination , visual acuity was 20/25 , he had conjunctival injection on slit lamp examination . there was also bilateral optic disk swelling plus retinal hyperemia ( optic disc hyperemia and vascular tortuosity ) and intraretinal hemorrhage on funduscopy . the patient was admitted and treated with cotrimoxazole , rifampin , doxycycline and prednisolone for 2 months . ocular manifestations subsided gradually within 6 months after treatment . brucellosis can affect the eye and lead to serious ocular complications . early diagnosis and prompt treatment should be considered in endemic areas .

linear and whorled nevoid hypermelanosis ( lwnh ) is a rare sporadic pigmentary anomaly , characterized by swirls and streaks of macular hyperpigmentation following the lines of blaschko , without preceding inflammation , and is usually seen in the first 2 years of life . include zosteriform hyperpigmentation , zosteriform lentiginous nevus , zebra - like hyperpigmentation , reticulate hyperpigmentation of iijima , and nevoid hyperpigmentation following blaschko lines . a 22-year - old male presented to the outpatient department with chief complaints of asymptomatic , dark - colored skin lesions over the body since 10 years of age . according to the patient , the lesions first appeared over both his arms and progressed within the next 2 years to involve the trunk and lower limbs . the lesions darkened with age and no new lesions appeared over the next 9 years ; however , over the last 1 year he started noticing multiple white - colored , pinpoint lesions appearing over the hyperpigmented lesions without any preceding skin itching or redness . there was no history of warty lesions or blisters prior to the appearance of these lesions . there was no history suggestive of any recurrent lower respiratory infections or involvement of the cardiovascular and the central nervous systems . the patient described presence of similar dark - colored skin lesions involving the trunk , arms , and legs in his mother without a history of any light - colored patches . cutaneous examination revealed presence of multiple , whorled , hyperpigmented macules , arranged bilaterally and symmetrically , along the lines of blaschko on the abdomen , chest , and back [ figure 1 ] . similar lesions were arranged linearly on the extensor and flexor aspects of the arms and legs . multiple pinpoint depigmented macules were seen along the areas of streaky macular pigmentation [ figure 1 ] . the texture of skin was normal over the streaks , the intervening skin , and over the depigmented areas . classical whorled , hyperpigmented macules along the lines of blaschko on the trunk with pinpoint areas of depigmentation hematological and routine biochemical tests revealed no abnormalities . histopathological evaluation of the depigmented lesion showed complete absence of melanocytes [ figure 2a ] . however , the pigmented macules revealed increased pigmentation of the basal cell layer with melanocytes present up to the mid - epidermis [ figure 2b ] . no focal areas of pigmentary incontinence were noticed in the dermis . based on clinicopathological correlation , a diagnosis of lwnh with punctate hypopigmentation was made . however , treatment for depigmented areas with narrow band ultraviolet b radiation and topical corticosteroids was advised . picture collage showing histopathological examination ( a ) complete absence of melanocytes in the depigmented area ( b ) abundance of melanocytes in the hyperpigmented area ( h and e , 40 ) linear and whorled nevoid hypermelanosis is a rare disorder of pigmentation characterized by hyperpigmented macules in a linear or whorled streaky configuration . onset is within a few weeks of age , with no preceding inflammation or palpable lesion . the usual age of the onset of hyperpigmentation is within the first few weeks of life , which continues to progress for a year or two before stabilization . clinically , reticulate hyperpigmented macules coalescing to form streaks and whorled areas are seen over the trunk , extremities , and neck following the lines of blaschko . the pigmented streaks display a v - shaped pattern over the spine , an s - shaped or whorled pattern over the anterior and lateral aspects of the trunk , and a linear arrangement over the extremities and genitalia . there is sparing of the face , palms and soles , eyes , and mucous membranes . central nervous system diseases include microcephaly , arhinencephaly and epilepsy whereas cardiac defects include ventricular septal defect and tetralogy of fallot . developmental retardation , facial and body asymmetry , deafness , and brachydactyly have been mentioned in the literature in association with lwnh . histopathology reveals diffuse moderate hyperpigmentation in the basal layer and prominence or vacuolization of melanocytes . genetic studies suggest somatic mosaicism as a cause for lwnh with mosaic trisomy of 7 , 14 , 18 , 20 ; x - chromosomal mosaicism has been reported . dermatoscopic feature mentioned include net like pattern of pigmentation in both linear and whorled parts by naveen et al . , and ertam et al . described a parallel pattern which consisted of linear or circular arrangement of parallel whorled streaks along lines of blaschko . monogenic skin disorders are the ones commonly described with lines of blaschko , however , polygenetic skin disorders such as psoriasis , lichen planus , segmental vitiligo , granuloma annulare , etc . can also present in similar patterns . literature search reveals an entity called as blaschkolinear vitiligo , which has been described in association with segmental vitiligo , acrofacial vitiligo , and non - segmental vitiligo . kovacevic et al . have reported a new entity called as mixed vitiligo of blaschko lines , where they discussed presence of segmental and non - segmental vitiligo in blaschkolinear pattern . till date , no cases of lwnh with areas of depigmentation have been mentioned in the literature , and to the best of our knowledge , this is most probably the first case report of familial lwnh superimposed by pinpoint spots of depigmentation .

linear and whorled nevoid hypermelanosis ( lwnh ) is a rare disorder of pigmentation characterized by hyperpigmented macules in a linear or whorled streaky configuration . lesions are distributed mainly on the trunk and extremities , sparing the palms , soles , and mucosae . depigmentation occurring in the pre - existing whorls has not been reported till date . herein , we report one such rare case of familial lwnh with depigmentation .

common variable immunodeficiency ( cvid ) is the most common form of severe antibody deficiency affecting both children and adults . the characteristic immune defect in cvid is impaired b - cell differentiation with defective secretion of immunoglobulin ( ig ) . the disorder is associated with a broad spectrum of clinical manifestations , including infections , chronic lung disease , gastrointestinal ( gi ) disease and autoimmune disease . bacterial infections of the sinopulmonary tract , particularly sinusitis and pneumonia , are experienced by most patients with cvid . here , a 29-year - old female patient was admitted with complaints of cough , fever , diarrhoea and swelling all over her body . physical examination revealed massive oedema , fever of 39c , crepitations on the lower lungs , increased bowel sounds and retarded development with weight of 40 kg and height of 145 cm . pathological : the laboratory results that were out of limits were leukocyte 11 400/l , hemoglobin 10 g / dl , hematocrite 31% , platelets 568 000/l , c - reactive protein ( crp ) 399 mg / dl , total protein 3 g / dl , albumin 1 g / dl , proteinuria 9 g / day and many leukocytes and fatty acids on direct examination of the faeces . ig levels were low : igg < 33.3 mg / dl ( n = 7511560 ) , iga < 6.67 mg / dl ( n = 82453 ) , igm 7.08 mg / dl ( n = 46304 ) . serum amyloid a deposition was detected on biopsies ( figure 1 ) obtained during gastroduodenoscopy and colonoscopy . renal biopsy performed to evaluate nephrotic syndrome was also consistent with aa amyloidosis ( figure 2 ) . with the history of recurrent infections and low ig levels , she was diagnosed as having cvid leading to secondary amyloidosis . after her hospitalization , her signs and symptoms cleared with antibiotic and antiproteinuric treatment and with antibiotherapy , intravenous ig , antiproteinuric treatment including losartan and cilazapril ; oedema and pleural effusion regressed with mild pretibial oedema remaining , crp level declined to 18 mg / dl , proteinuria declined to 7 g / day and albumin level rised to 2.2 g / dl . after resolution of gi symptoms , she was started on colchicines therapy ; she is under follow - up with intravenous ig treatment without any infection during the last 10 months . age of onset is typically after puberty and before 30 years of age , with some evidence of a bimodal distribution demonstrating peaks between 1 and 5 years and between 18 and 25 years . cvid is a primary immune deficiency disorder characterized by markedly reduced serum levels of igg and low iga or igm , with impaired antibody responses , despite the presence of b cells . however , cvid is associated with a high incidence of inflammatory , autoimmune and malignant conditions , features of more fundamental immune dysregulation . sinopulmonary infections , including pneumonia , bronchitis and sinusitis , as well as otitis and conjunctivitis , are observed in the majority of patients with cvid . these infections may be acute , chronic or recurrent . over three - quarters of patients have at least one episode of pneumonia prior to diagnosis . chronic lung disease is a common problem in patients with cvid and can lead to recurrent hospitalizations , significant morbidity and early death . in a large clinical study of 248 patients , 27% had either bronchiectasis or restrictive or obstructive lung disease . another study of 224 patients found that 34% had chronic lung disease at the time of diagnosis , which increased to 46% during a mean follow - up of 11 years . the risk factors for the development of chronic lung disease in patients with cvid have not been fully defined . one report of 18 cvid patients found that those with reduced total memory b cells ( cd27 + b cells ) and very low numbers of switched memory b cells ( cd27+igmigd ) were more likely to have chronic lung disease . gi disease is identified in ~20% of cvid patients and may be the presenting disorder in some . specific disorders include inflammatory bowel disease , sprue - like illness with flat villi , nodular lymphoid hyperplasia , pernicious anaemia , chronic giardiasis , protein - losing enteropathy and nonspecific malabsorption . one biopsy study of gi pathology in 20 cvid patients over a 26-year period found that over one - half of the patients lacked plasma cells throughout the intestinal tract , and 47% showed lymphoid aggregates . we detected deposition of serum amyloid a besides nodular lymphoid hyperplasia in biopsies taken from the stomach , duodenum and colon . routine laboratory studies are often normal in cvid , in the absence of an associated disorder in addition , modest lymphopenia and a reduced cd4 + level may develop over time . our case had ig levels at undetectable levels and serious hypoalbuminaemia due to proteinuria and malabsorption . the management of cvid involves sufficient gamma globulin replacement therapy and monitoring for and treatment of associated inflammatory disorders and malignancies . ig replacement therapy reduces the frequency of most types of infections as in our case , as well as slows the progression of chronic lung disease and offers some protection against autoimmune disorders . the usual initial dosing for intravenous ig is 300400 mg / kg , given every 34 weeks , with the goal of maintaining a trough igg level in the middle of the normal range . isolated nephrotic syndrome cases responsive to steroid therapy have been reported in the literature associated with cvid . but there is no case with nephrotic syndrome due to amyloidosis documented with renal biopsy . another patient with nephrotic - range proteinuria ( 9 g / day ) was reported to have amyloid deposition in gastric and duodenal biopsy , but renal biopsy was not performed . even if intravenous ig treatment may prevent infections and consequently amyloid deposition , insufficient treatment may lead to amyloidosis . otherwise , with the increasing life expectancy of the patients and resultant increased number of infections , renal amyloidosis may be expected to increase in frequency . renal amyloidosis in our patient is also thought to be due to delayed diagnosis and gamma globulin treatment . patients with late diagnosis and insufficient treatment of infections are prone to develop amyloidosis and nephrotic syndrome which worsens the prognosis of the disease which has already high morbidity and mortality rates . cvid must be kept in mind in patients with recurrent sinopulmonary infections in order to prevent co - morbidities .

common variable immunodeficiency ( cvid ) is the most common form of severe antibody deficiency . the disorder is associated with a broad spectrum of clinical manifestations , including infections and chronic lung , gastrointestinal and autoimmune diseases . a 29-year - old female patient has had frequent sinopulmonary infections and gastroenteritis for the last 20 years and had been given broad - spectrum antibiotics for treatment . immunoglobulin ( ig ) levels were at undetectable levels . renal biopsy was consistent with aa amyloidosis . she is now under follow - up with periodic intravenous ig treatment without any infection during the last 10 months . cvid must be kept in mind in patients with recurrent sinopulmonary infections .

the national cooperative dialysis study , the first randomised controlled study of dialysis dose , defined an adequacy threshold for end - stage chronic kidney disease patients receiving chronic haemodialysis based on the dialyser clearance of urea , a small solute marker of nitrogen turnover , which was defined in terms of a dimensionless parameter known as the normalised urea clearance , or kt / v ( k , dialyzer urea clearance ; t , dialysis session duration ; and v , urea volume distribution ) . below a sessional threshold kt / v of 0.9 for standard thrice - weekly schedules , complication - free survival was compromised within months . subsequent observational studies suggested that higher doses resulted in improved longer - term outcomes , and by consensus the minimum target kt / v was raised to 1.2 . a subsequent prospective randomised controlled study , the hemodialysis ( hemo ) study , reported that higher doses did not appear to further improve outcome . however , subgroup analysis suggested that women may benefit from higher kt / v doses , fuelling suggestions that using standard kt / v targets to prescribe dialysis may lead to under - dosing in women and small men . these studies suggest that , for standard thrice - weekly therapy , medium - term survival ( measured in months ) is dependent on achieving a minimum level of small solute removal , as defined by the national cooperative dialysis study . just as the amount of dialysis delivered to patients with end - stage chronic kidney disease is important in determining survival , it was reported that the dose of intermittent haemodialysis or continuous renal replacement therapy ( crrt ) was also important in determining survival in patients with acute kidney injury ( aki ) [ 8 - 10 ] , although this was not a universal finding . as patients with aki continue to have high mortality , and evidence - based clinical management is somewhat limited , two prospective multicentre trials were designed to investigate the effect of dose of renal replacement therapy on outcome in patients with aki . the veterans affairs / national institutes of health ( va / nih ) study essentially randomised patients to initially receive either an intensive or less intensive dose of intermittent haemodialysis , or an intensive or less intensive dose of crrt , depending upon severity of illness at the time of randomisation . ( during the course of the study patients were switched between treatment modalities according to haemodynamic stability . ) during haemofiltration , it is assumed that urea is effectively cleared ( to the extent that the concentration in the effluent ultrafiltrate is equal to that of plasma water ) so that urea clearance can simply be assessed by the total ultrafiltration volume achieved . in this study , more intensive renal replacement therapy did not show any survival advantage for either the intermittent haemodialysis or crrt groups . however , the minimum haemodialysis target kt / v of 1.2 was somewhat higher than that typically prescribed for patients with aki by the recruiting centres . in addition , there was no survival advantage for the haemofiltration cohort compared to those treated by dialysis . haemofiltration clears solutes primarily by convection , thus removing a larger spectrum of solutes than haemodialysis , which predominantly clears small water soluble solutes by diffusion . the second study , the renal ( randomised evaluation of normal versus augmented level of renal replacement therapy in icu ) study , assessed the effect of an augmented dose of crrt ( an ultrafiltration rate of 40 ml / kg / h versus 25 ml / kg / h ) . once again , this study failed to show any significant effect of dose of convective renal replacement therapy on patient outcomes , although the delivered dosages were less than that prescribed and both small patients ( < 60 kg ) and very heavy patients ( > 120 kg ) were excluded . although urea can dissociate to cyanate in plasma water and then form carbamylated products in a reversible fashion , with some analogy to glycosylation , it would appear that toxicity from the accumulation of small nitrogenous solutes is not the major determinant of short - term outcome ( days to weeks ) in patients with aki . aki frequently occurs in the setting of multiple organ failure , and mortality remains high , with patient outcome typically dependent upon the severity of the underlying condition and associated co - morbidities . the replacement of organ function may play a critical short - term role in maintaining life in patients already destined by other factors to have the potential to recover . however , urea clearance is only one component of renal replacement therapies . for example , failure to correct persistent volume overload is associated with not only increased post - surgical morbidity , but also increased risk of aki and mortality . thus , for patients with aki , the adequate removal of even smaller moieties than urea is the principal determinant of the adequacy of renal replacement . these moieties are the neglected uraemic toxins , including potassium , sodium , hydrogen ions and water ( figure 1 ) . the consequences of the accumulation of these moieties , hyperkalaemia , pulmonary oedema , and acidosis , may be lethal in minutes . toxins play a role over time , and as such may require different clinical management strategies . hemo , hemodialysis ; ncds , national cooperative dialysis study ; renal , randomised evaluation of normal versus augmented level of renal replacement therapy in icu ; va / nih , veterans affairs / national institutes of health . although the delivery of higher doses of haemofiltration or more frequent haemodialysis did not improve overall outcome , higher volume crrt exchange cycles and more frequent haemodialysis treatments will help correct acidosis , and may be appropriate during the initial resuscitation phase of aki . correction of volume overload may help explain the positive findings and improved clinical outcomes reported from some of the earlier trials of increased dose of renal support , compared to the more recent va / nih and renal studies , which had similar fluid balance targets .

traditionally , the dose of haemodialysis or haemofiltration delivered to patients with kidney failure is assessed by urea clearance . for patients with chronic kidney disease below a critical urea clearance threshold , patient wellbeing is compromised . it was suggested , therefore , that the dose of dialysis or haemofiltration delivered could also affect outcomes for patients with acute kidney injury . two major prospective multicentre clinical trials have recently reported that a higher intensity of renal support , by either intermittent haemodialysis or continuous renal replacement therapy , did not improve patient survival or recovery from dialysis . it must be recognised , however , that urea clearance is only one component of renal supportive therapy , and other aspects , including volume control , electrolyte homeostasis and acid - base balance , may be equally important targets for patients with acute kidney injury .

a 12-year - old female patient visited the department of oral medicine and radiology , yenepoya dental college , with a complaint of missing anterior teeth since three years ago . she had a history of exfoliation of decidious teeth due to mobility three years ago and there was no eruption of the permanent teeth . there was a history of presence of two mandibular natal teeth which were exfoliated two weeks after birth . her parents had no consanguineous marriage , however her elder sibling died of similar medical problem . her medical history revealed that she had visited genetic clinic and they have diagnosed her with ellis - van creveld syndrome . she also had dyspnoea because of congenital lobar emphysema for which she had undergone left upper lobectomy when she was one and half year old . intraoral examination showed v - shaped notching of the upper lip at the mid line ( fig . 2a ) , absence of mucobuccal fold in maxillary and mandibular anterior region ( figs . 2b and c ) , conical shaped teeth , missing mandibular permanent anterior and retained deciduous mandibular canines , and right lateral incisor teeth ( fig . 2c ) . chest radiograph showed homogenous opacity in the left upper zone associated with elevation of hilum and left dome of diaphragm suggestive of left upper lobectomy ( fig . antero - posterior view of the legs showed genu valgum , hypoplasia of lateral end of tibia associated with medial tibial diaphysial outgrowth called exostosis , and mild shortening of fibula ( fig . hand - wrist radiograph showed carpal fusion , postaxial polydactyly , and shortening of metacarpal and phalangeal bone with cone shaped epiphysis . there was fusion of capitate and hamate on the right hand and hamate and triquadral on the left hand ( fig . a team work of pedodontist , oral and maxillofacial surgeon , and prosthodontist was required to correct the craniofacial and dental defects in this patient . since the patient was in growing phase , the prosthodontist decided to give acrylic partial dentures for the missing teeth . oral and maxillofacial surgeon performed frenectomy for the high labial frenum as it was hindering the placement of the partial denture . ellis - van creveld syndrome ( evc ) otherwise known as chondroectodermal dysplasia has autosomal recessive inheritance . it is a syndrome found in amish population of pennsylvania in usa , affecting male and female equally.2 familial history may include parental consanguinity or affected siblings or family members . our patient was the second child of non - consanguineous and normally developed parents , however her elder sibling had died of similar medical problem . these patients die due to either cardiac problem or respiratory distress.2 our case did not have any cardiac problem , however there was congenital lobar emphysema because of her dyspnoea . she had undergone left upper lobectomy for the same reason when she was one and half year old child . the syndrome affects mainly the bones such as the lower limb with genu valgum , which requires surgical correction . the upper limbs show the characteristic bilateral postaxial polydactyly.3,4 our case had all these limb anomalies . other ectodermal features in this syndrome are the absence or hypoplastic features of finger and toe nails , natal teeth , conical shaped or missing teeth , and absence of labial vestibule because of fusion of upper lip to the gingival margin , leading to the notching of upper lip . this could be due to the continuation of the normal serrated condition of the gingiva from the third to seventh month in the uterine life of the fetus . it is considered as the characteristic and diagnostic feature of this syndrome.5 - 7 all of these features were also present in this case . there are about 40 independent evc / evc2 mutations.8 ruiz - perez and goodship mentioned that the abnormalities in evc syndrome resulted from the tissue specific disruption of the response to hh ligands.9 weyer 's syndrome , asphyxiating thoracic dystrophy , and orofacial digital syndrome should be considered in the differential diagnosis of this syndrome . evc syndrome and weyer 's acrodental dysastosis are allelic conditions caused by loss of function mutation in evc and evc2 . these are separated by 2 - 6 kb of genomeric sequence on chromosome 4p16.10 all the features are similar in both conditions except for the fact that there is delay in fusion of mandibular symphysis in acrodental dysastosis.6 patients with evc syndrome and asphyxiating thoracic dystrophy will have identical features in hand , pelvis , and long bones . presence of cardiac anomalies , nail hypoplasia , fusion of upper lip to gingiva and neonatal teeth in evc syndrome , and renal failure with hypertension in asphyxiating thoracic dystrophy will help in distinguishing these two disorders from each other.6,11 presence of multiple gingivolabial frenula is similar to evc in orofacial digital syndrome . hypoplastic nasal cartilage , moderate mental retardation , fissured tongue , and ankyloglossia helps to differentiate this from evc syndrome.11 in conclusion , the effective management of this syndrome will require a team which includes pedodontist , oral and maxillofacial surgeon , prosthodontist , clinical geneticist , cardiologist , pulmonologist , orthopaedician , urologist , psychologist , pediatrician , and pediatric neurologist . since the oral manifestation is one of the characteristic diagnostic features , and it affects the esthetic problem , speech , jaw growth of the child , the dentists have an important role to play in proper management of such cases .

ellis - van creveld syndrome is a rare congenital genetic disorder having autosomal recessive inheritance . it is a syndrome affecting the amish population of pennsylvania in usa with prevalence rate of 1/5,000 live at birth . in non - amish population , the birth prevalence is 7/1,000,000 . the syndrome is characterized by bilateral postaxial polydactyly of the hands , chondrodysplasia of long bones resulting in acromesomelic dwarfism , ectodermal dysplasia affecting nails as well as teeth and congenital heart malformation . there were very rare reports of this syndrome in dentistry . the present case focuses on the striking and constant oral findings of these patients , which are the main diagnostic features of this syndrome . since the oral manifestations affect the esthetic , speech , and jaw growth of the child , the dentists have an important role to play in proper management of such case .

in clinical practice , paradoxical clinical results that require alternative strategies may be encountered . here we present a case of a patient with results of cardiac catheterization that are incompatible with st segment elevation and elevated cardiac enzymes . although technologically advanced diagnostic examinations are becoming increasingly available , noninvasive bedside ultrasound examination remains to be indispensable . although cardiac myxomas are relatively uncommon compared with coronary and valve lesions in the elderly , they are easily diagnosed by basic echocardiography ; this diagnostic test may be the key to solving puzzling presentations and should a part of most cardiac workups . stroke is a complication in 10% to 30% of cardiac myxoma patients as found in a case series spanning 20 years in belgium and a 11-year series conducted at the mayo clinic ; no cases of cardiac myxoma in either study had concurrent myocardial infarction . forty cases of myxoma - related myocardial infarction were recorded in a systematic review of published data spanning 32 years . this case report shares our experience with a rare case of cardiac myxoma with concurrent stroke and st - elevation myocardial infarction with normal coronary angiography . signed consent from the patient was obtained , and approval was granted by the institutional review board of taipei tzuchi hospital for this case report . a 67-year - old unconscious woman presented to the emergency department of our hospital within 30 minutes of collapsing at an outdoor market . she was afebrile with blood pressure of 100/80 mm hg , heart rate of 90 beats per minute , and respiratory rate of 26 breaths per minute on mask oxygen . on physical examination , the patient displayed grimace in response to painful stimuli , pupils were equal and reactive to light , heart beats were regular without obvious murmurs , breath sounds were coarse without wheezing , abdomen was soft and nontender , urine was clear with adequate output into a foley bag , and legs were warm and nonedematous ; examination was otherwise unremarkable . electrocardiogram showed significant st elevation in the inferior and lateral leads ( figure 1 ) . chest x - ray showed lung congestion , minimal effusion in the pericardial and pleural spaces , and a narrow mediastinum . computed tomography of the brain showed no hemorrhage or detectable ischemia . although awaiting cardiac catheterization , she regained consciousness and was able to follow commands and move all extremities . however , severe orthopnea with respiratory distress developed , and she was endotracheally intubated . we then performed echocardiography , which revealed an oscillating round mass measuring 3 3 cm inside the left atrium attached by a pedicle to the interatrial septum that was obstructing inflow through the mitral valve during diastole ( figure 2 ) . although in the intensive care unit , the patient 's lung edema deteriorated rapidly , resulting in cardiogenic shock that required increased inotropic support and high - setting ventilator support . echocardiograph of the oscillating left atrial myxoma ; surgical specimen of the completely resected atrial myxoma . the myxoma was resected en bloc with its pedicle and adjacent interatrial septum ( figure 2 ) . ataxia was noted during rehabilitation , and magnetic resonance imaging of the brain revealed left cerebellar infarction . cardiac atrial myxoma is uncommon , with an estimated incidence of < 0.1% by autopsy series . often initially asymptomatic , symptoms arise because of obstructive , embolic , or constitutional events . a 20-year surgical cohort of cardiac myxoma cases in belgium estimated 65.6% of presenting symptoms to be obstructive and 15.6% to be embolic . as in our case , emergent resection may be necessary if cardiogenic shock develops because of mitral valve obstruction . echocardiography can readily reveal obstruction by a cardiac myxoma , usually of mitral valve inflow , allowing timely determination of severity and necessity for emergent surgical resection . myxoma - related stroke is relatively more common than myxoma - related myocardial infarction for myxoma - related embolic events . in the aforementioned 20-year myxoma surgical series , brain ischemia was involved in 15.6% of cases , brain and peripheral artery embolic ischemia in 6.2% , and angina in 3.1% , whereas no cases involved myocardial infarction . myxoma - related coronary artery embolism is rare and much less common than brain embolism . estimated incidence of myocardial infarction in cardiac myxoma ranges from close to 0% to as high as 10% if coronary emboli is included . a review of cardiac myxoma cases spanning 11 years conducted by the mayo clinic found that 12% of cases had image - evident stroke . therefore , coexisting stroke and myocardial infarction , as in our case , are exceedingly unusual . myocardial infarction with normal immediate angiography is rare for cardiac myxoma . in a systematic review of published data from 1970 to 2002 , patent coronary arteries were found only on latent angiography ( performed more than 2 weeks after the initial event ) , but not on immediate angiography . negative latent angiography may be explained by recanalization performed days or weeks after the initial coronary events . possible explanations may include rapid and spontaneous recanalization , a washout effect of embolic myxoma fragments or induced thrombi , or compromised hemodynamics because of mitral valve obstruction . echocardiography , a noninvasive and portable examination , is often critical for properly identifying the etiology of cardiac conditions such as acute heart failure . basic echocardiography , or focused cardiac ultrasound , is easily learned and often sufficient for detecting intracardiac masses or intrathoracic effusion . our case demonstrates the importance of basic echocardiography for optimizing the management of cardiac cases , and supports the recommendation that basic echocardiography be readily available for most cardiac teams or acute care centers . our atypical case of normal immediate coronary angiography in myxoma - related myocardial infarction with mitral valve obstruction and multisystem embolism ( heart and brain ) deserves attention because of the rarity of this combination of presentations . the case stresses the value of basic echocardiography in the efficient and effective management of the patient and the importance of including it in most cardiac workups . in addition , this experience is a reminder for clinicians to include cardiac myxoma in the differential diagnosis list and to manage it timely if found . although cardiac myxoma may have unexpected presentations , it can be easily diagnosed by basic echocardiography and should not be missed . once diagnosed , cardiac myxoma requires timely surgical en bloc resection to avoid deterioration resulting from worsening embolism or obstruction .

abstractwe present a case of cardiac myxoma with atypical presentations of concurrent stroke and angiography - negative myocardial infarction . the case emphasizes the importance of basic echocardiography and timely surgery in the management of cardiac myxoma.an elderly woman presented to the emergency department in an unconscious state . electrocardiogram and elevated cardiac enzymes suggested acute myocardial infarction ; however , immediate coronary angiography proved patency . basic echocardiography revealed an oscillating left atrial myxoma obstructing inflow through the mitral valve . after regaining consciousness while in the intensive care unit , the patient developed respiratory distress and shock , and emergent en bloc resection was performed . ataxia was noted in her postoperative course and multiple small cerebellar infarcts were found on magnetic resonance imaging . after a 1-month period of rehabilitation , the patient recovered well and continues to be followed as an outpatient.cardiac myxoma requires timely management and may be missed if not included in the differential diagnoses . basic echocardiography , also called focused cardiac ultrasound , may aid in the diagnosing of perplexing cardiac cases .

most reported cases have documented the development of significant complications , since it is difficult to diagnose this rare entity preoperatively . the literature is reviewed and the radiologic findings and management of this rare entity are discussed . a 90-year - old woman was referred to our hospital for acute appendicitis complicated with right incarcerated groin hernia . she initially presented to a local hospital with right lower quadrant abdominal pain for one day . the abdomen and pelvic ct with intravenous and oral contrast showed swelling of the appendix with perifocal fat stranding and cecal wall thickening ( figure 1a ) . there was initial improvement , however , a painful and irreducible lump in the right groin developed three days thereafter . on referral , a previously performed ct revealed the finding of a fluid - contained mass with stranding of surrounding fat in the right groin lateral and inferior to the pubic tubercle . neither the appendix nor cecum was in close proximity to the mass ( figure 1b ) . the appendix was inflamed and severely adherent to the cecum and terminal ileum but there was neither perforation nor abscess . the femoral hernia neck was constricted , and there was no herniation of the abdominal viscera ( figure 2a ) . a separate groin incision was made , and phlegmonous inflammation of the distended femoral hernia sac was found ( figure 2b ) . dissection was carried out to free the entire circumference of the hernia sac and the neck . the hernia sac was excised , and the neck was closed with suture - ligation . mcvay hernioplasty was performed . on opening , the sole content of the hernia sac was pus . the reported incidence of appendicitis within a groin hernia is 0.13% of all cases of acute appendicitis.1 it most frequently occurs in the right inguinal and right femoral hernias . rene jacques croissant de garengeot , in 1731 , first reported a case of acute appendicitis within a femoral hernia.2 since then , there have been only scattered case reports of appendicitis within femoral hernias . the clinical presentation usually suggests an incarcerated or strangulated femoral hernia , and acute appendicitis is incidentally found at operation . with the increased use of ct for evaluating patient with atypical presentation of incarcerated hernia and lower abdomen peritonitis , there have been occasional reports in which the diagnosis of hernial appendicitis is made preoperatively . the typical ct features include a tubular structure within the hernia sac with surrounding fat stranding and low position of the cecum or close proximity to the hernia sac.35 this prospective ct diagnosis may be helpful in establishing appropriate treatment strategy . our case illustrates a very rare variety of femoral hernia appendicitis . the appendix was not contained in the hernia sac . instead zuckerkandl first described this phenomenon in 1891 , albeit within an inguinal hernia sac.6 as with any peritoneal recess , the hernia sac may contain pus secondary to any intraperitoneal infectious process . the pus causes distention and inflammation of the sac , which was mistaken for strangulated hernia . the appendix may be either in close proximity to or distant from the hernia sac . there is usually no evidence of intestinal obstruction , which will raise suspicion of strangulated omentum or richter s hernia . once the diagnosis of strangulated femoral hernia is made , most surgeons prefer direct surgical intervention to avoid considerable risk of postoperative complications . thus , the diagnosis of appendiceal pus - containing femoral hernia is made only during the operation . despite wide availability of ct , the ct features of pus - containing femoral hernia is almost never reported in the literature . in our patient , the ct showed an inflammatory fluid - filled mass lateral and inferior to the pubic tubercle , a typical location of acute appendicitis , and no evidence of intestinal obstruction . the differential diagnosis is acute appendicitis complicated with a rare complication of pus - filled femoral hernia or a richter s hernia . as illustrated in our case , the awareness of the ct findings facilitated the decision on a treatment strategy . although drainage of purulent content or appendectomy via hernia sac has been reported in many cases in which diagnosis is not expected , it carries a high rate of infectious complications . separate laparotomy and groin incision would be very helpful in complete dissection and direct excision of the hernia sac . there are some controversies surrounding the method of femoral hernia repair in cases of hernial appendicitis or pus - containing femoral hernia . however , if no spillage of infectious content occurs , mesh repair is a reasonable alternative . in conclusion , selective use of ct for evaluating an atypical case of incarcerated femoral hernia may be helpful in early diagnosis and decision on the treatment strategy . the ct features of typical acute appendicitis and incarceration of inflammatory fluid - filled hernia sac without evidence of intestinal obstruction should raise suspicion of this rare entity .

acute appendicitis involving the hernia sac is infrequent but well - documented in medical literature . in most instances , it occurs within the right inguinal ( amyand s hernia ) or right femoral hernia ( de garengeot hernia ) . the diagnosis is always mistaken for incarcerated groin hernia . during surgery , the appendix itself , either perforated or strangulated , is most commonly encountered within the hernia sac . in very rare occasions , only appendiceal pus is found in the hernia sac . in this paper , we report the case of a 90-year - old woman with acute appendicitis and a tender mass in the right groin . typical findings of acute appendicitis by computed tomography ( ct ) and incarcerated femoral hernia with groin cellulitis misled us into preoperative diagnosis of strangulated femoral hernia . acute phlegmonous inflammation of the incarcerated femoral hernia sac containing pus only and acute suppurative appendicitis were found intraoperatively . this case presents a rare complication of acute appendicitis and the first report of ct - documented appendiceal pus - contained femoral hernia . knowledge of this rare condition is helpful in establishing preoperative diagnosis and patient management decisions .

e. coli cultures were grown in lb liquid media or on lb agar plates with antibiotics when necessary ( 50 g / ml kanamycin , 50 g / ml apramycin , 25 to 50 g / ml chloramphenicol , 50 g / ml carbenicillin ) at 37 c with shaking . streptomyces liquid precultures were grown in tryptic soy broth with the appropriate antibiotics ( 50 g / ml kanamycin , 50 g / ml apramycin , 25 g / ml nalidixic acid ) , inoculated into ssm media ( 1% soytone , 1% soluble starch , 2% maltose , 0.5% trace element solution , ph 5.7 ) and grown at 30 c with shaking . streptomyces were also grown on mannitol soya flour medium plates ( 2.0% agar , 2.0% mannitol , 2.0% soya flour ) with appropriate antibiotics ( 50 g / ml kanamycin , 50 g / ml apramycin , 25 g / ml nalidixic acid ) . salinispora were grown in a1 media ( 1.0% starch , 0.4% yeast extract , 0.2% peptone , and 2.8% sea salt ) at 30 c with shaking . the method of tar capturing used in this study has been previously reported . the isolation of high - quality genomic dna from salinispora was performed according to standard procedures . the capture vector , pcap01 , containing yeast arsh4/cen6 and trp1 marker , e. coli puc ori , streptomyces c31 integrase gene(int ) , its attachment site ( attp ) and origin of dna transfer ( orit ) , aph(3)ii gene ( kan / neo resistance ) , and two 1 kb regions flanking the gene cluster on either side was used to capture and propagate the enterocin gene cluster . the vector pcap01 with the captured enc gene cluster from s. pacifica cnt-150 was named pbb01 . the plasmid pbb01 and its derivatives were conjugated into s. lividans tk23 and s. coelicolor m1146 using a standard triparental mating method with e. coli et 12567/pbb01 and e. coli et 12567/pub307 . after 6 days of growth in ssm media , the cultures were extracted with four volumes of etoac and concentrated in vacuo . the extracts were then dissolved in 100 l of mecn and analyzed by hplc using a luna 5u c18 column ( 150 mm 4.60 mm , 5 m beads ) with a linear gradient of 2100% mecn in water with 0.1% tfa over 30 min with a flow rate of 0.7 ml / min . hplc - hr - esi - msms analysis of all extracts was carried out on an agilent 1290 q - tof ( 2002000 m / z , 20 kev ) . the data were subjected to the molecular networking workflow and analyzed as described previously . the inactivation of the acyl - coa synthetase encoding gene , enc16 , in the heterologous host strain s. coelicolor m1146-pbb01 was done using -red recombination with an apramycin - resistant marker ( aac(3)iv ) as described previously .

heterologous expression of secondary metabolic pathways is a promising approach for the discovery and characterization of bioactive natural products . herein we report the first heterologous expression of a natural product from the model marine actinomycete genus salinispora . using the recently developed method of yeast - mediated transformation - associated recombination for natural product gene clusters , we captured a type ii polyketide synthase pathway from salinispora pacifica with high homology to the enterocin pathway from streptomyces maritimus and successfully produced enterocin in two different streptomyces host strains . this result paves the way for the systematic interrogation of salinispora s promising secondary metabolome .

the study was conducted in 100 households of 9 affected villages of engil district on the edge of herat city . households were included if they were located on 3 randomly selected transect lines and if members of the households owned either cattle or sheep . because there are no reliable estimates on seroprevalence of cchf in afghanistan , we estimated prevalence of immunoglobulin ( ig ) g to be 7% ( 2,3 ) . a sample size of 160 persons was required to detect this prevalence at the 95% confidence level . allowing for cluster sampling at the household level , 320 persons was the target sample size ( 90 households , assuming an average of 3.5 persons > 15 years of age per household ) . from each household , all members > 15 years were surveyed , if they gave consent . sheep and cattle were selected as livestock types , and 1 or 2 animals per household were randomly selected . inclusion criteria for humans were residing in a livestock - owning household , giving informed consent , being > 15 years of age , willing to answer the risk factor questionnaire , and willing to give 5 ml of blood . for collection of blood samples and vector specimens from sheep and cattle , permission was given by the head of household . a standardized , structured , and pretested questionnaire that covered individual data for each participant , including personal details , exposure variables , and self - reported disease history , was used . animal data were collected from each household and included each animal s origin , tick exposure , age , and sex . blood samples from human participants were collected by trained health workers according to standard procedures . blood samples were centrifuged at room temperature at the local laboratory on the day of collection . serum was separated , frozen , and transported to kabul for storage and onward shipment . a sandwich / indirect elisa detected specific igg at a 1:100 dilution for all human samples by using the vector - best diagnostic kit , ( vector - best , novosibirsk , russia ) . we have previously compared this kit with an in - house elisa ( us army medical research institute for infectious diseases , fort detrick , md , usa ) , and the results were comparable in all samples tested ( l. mustafa et al . , unpub . we used an in - house elisa using the ibar 10200 strain of cchf as antigen ( us army medical research institute for infectious diseases ) and anti - species igg horseradish peroxidase conjugated ( kpl inc . , gaithersburg , md , usa ) to test for igg in all animal serum specimens . data were analyzed by using stata version 8 ( statacorp , college station , tx , usa ) . the primary outcome was seropositivity among household members , with secondary outcomes being seropositivity in animals and the presence of virus in ticks . exposure factors for seroprevalence were identified on an a priori basis and appropriate measures of statistical significance were applied to detect differences at the 95% confidence level . the study was approved by the ethics boards of namru-3 and the afghan public health institute , afghanistan . in total igg seroprevalence was 37/330 ( 11.2% , 95% confidence interval [ ci ] 8.015.1 ) . of all the potential explanatory variables , only 2 factors were associated with an elevated risk of igg positivity : daily contact with cattle ( 33/264 [ 12.5% ] vs. 1/52 [ 1.8% ] ; = 5.1 , p = 0.02 ) and exposure to raw animal skins ( 24/144 [ 16.7% ] vs. 12/176 [ 6.8% ] ; = 7.7 , p = 0.006 ) . self reported clinical illness ( fever ) occurred in 55% of participants over a 5-month reporting period . among the participants , 20.8% reported that they had had an illness involving bleeding from teeth , gums , and or other parts of the body , but this event was not associated with igg positivity or with age group . these results suggest that the risk for cchf exposure is uniformly high among the population . the oldest age group shows an approximate lifetime risk for exposure and seroconversion of 17% ( 95% ci 10.2%25.8% ) . ninety - two cattle and 40 sheep were included , and serologic analysis of their blood samples was conducted . seroprevalence was 79.1% ( 95% ci 69.0%87.1% ) among cattle and 75.0% ( 95% ci 57.0%88.5% ) among sheep . prevalence was uniformly high regardless of age , sex , or origin of the animals , suggesting that the disease is highly endemic in the livestock population . among our sample , ticks ( n = 259 ) from domestic animals were predominantly adult hyalomma marginatum ( 94.6% ) . engorged females were found more on cattle than on sheep ( 43% and 27% , respectively ) . seroprevalence in this population of animal owners is higher than in other reported studies from the region ( 3,4 ) , and the risk for exposure appears approximately uniform . the route of transmission to humans is either through the bite of ticks or through contact with infected animals or animal products . this second route of transmission is probably more important in iran and afghanistan than tick - borne transmission . control of cchf requires control of the disease vector , and surveillance is necessary to ensure optimum timing of interventions such as livestock dipping or sponging because tick abundance is highly seasonal . further research on rates of antibody acquisition among humans and animals , virus transmission dynamics , and effectiveness of disease control measures is required . cchf is a regional public health concern of larger than previously acknowledged significance and requires control mechanisms from both the health and agriculture sectors .

in response to an outbreak of crimean - congo hemorrhagic fever in western afghanistan , we measured immunoglobulin g seroprevalence among household members and their animals . seroprevalence was 11.2% and 75.0% in humans ( n = 330 ) and livestock ( n = 132 ) , respectively . persons with frequent exposure to cattle had an elevated risk of being immunoglobulin g positive .

the most common ocular adverse event following the use of cyclosporine a ( csa ) 0.05% ophthalmic emulsion is ocular burning ( 17% ) . other adverse effects that have been reported include conjunctival hyperemia ( 1 - 5% ) , discharge , epiphora , eye pain , foreign body sensation , pruritus , stinging and blurred vision . here , we report a specific side effect of csa , namely eye drop - induced eyelash elongation in a patient with refractory giant papillary conjunctivitis . a 32-year - old female with giant papillary conjunctivitis on the left eye , who had undergone papillectomy 3 years previously and was refractory to topical steroid therapy , was treated with csa 0.05% ophthalmic emulsion ( restasis ) 4 times a day , preservative - frees artificial tears and gentamicin ophthalmic solution in the left eye . after 5 months of topical csa treatment , elongated eyelashes of her left eye were observed without other adverse effects . although hypertrichosis and trichomegaly have been documented in the literature as side effects of systemic csa , topical csa 0.05% eye drop - induced elongated eyelashes have not been reported , and we believe ophthalmologists should be mindful and inform patients about this specific side effect . a-32-year - old asian female patient , who had received conjunctival papillectomy on the left eye in 2002 , visited our ophthalmic outpatient department in 2007 complaining of a swollen eyelid and discharge from her left eye . the ocular examination revealed notable injected conjunctiva , especially over the superior part , and multiple papillae with injected and engorged vessels on the left eye ( fig . , she was started on topical steroid medications including fluorometholone 0.1% and prednisolone acetate 1% ophthalmic solutions for 2 months . however , steroid treatment was discontinued due to poor response and an elevated intraocular pressure . therefore , her treatment was switched to topical cyclosporine a ( csa ) 0.05% ophthalmic emulsion ( restasis ) 4 times a day , preservative - free artificial tears and gentamicin ophthalmic solution . after 5 months of topical csa treatment , she came back to our clinic complaining of elongated and darkened eyelashes on her left eye without other adverse effects ( fig . 2 ) . according to the patient , no systemic medications such as calcium channel blockers , erythropoietin or minoxidil were used during the treatment period . csa is a hydrophobic , cyclic polypeptide produced as a metabolite by the fungus tolypocladium inflatum . csa functions as an immunomodulating agent that binds to cyclophilin , a cytoplasmic protein , thus interrupting the signaling for interleukin ( il]-2 production , in addition to inhibiting the proliferation of cd4 t lymphocytes . it also has direct inhibitory effects on both eosinophil and mast cell activation , which has established its role in the treatment of allergic inflammation [ 2 , 3 ] . in the early 1980s , meanwhile , csa eye drops were also prescribed for patients with inflammatory ocular surface disorders , particularly dry eye syndrome and severe allergic keratoconjunctivitis . the major side effect of systemic csa is nephrotoxicity that is reversible with dosage reduction . other documented adverse reactions to systemic csa include mild hepatotoxicity , hypertension , dose - dependent hypertrichosis and trichomegaly , tremor , infection , gum hyperplasia , gastric irritation symptoms and neuropathies . on the other hand , adverse events following the use of csa 0.05% ophthalmic emulsion include ocular burning ( 17% ] , conjunctival hyperemia ( 1 - 5% ] , discharge , epiphora , eye pain , foreign body sensation , pruritus , stinging and blurred vision . however , to our knowledge , no csa ophthalmic emulsion - induced hypertrichosis has been reported in the literature to date . suggested that csa induces telogen follicles to enter an anagen growth phase , implying a role of csa in regulating the hair follicle immune system and its cellular components through the release of inhibitory / stimulatory cytokines [ 8 , 9 ] . the experiments also indicated that the rate of anagen induction is dependent on the dose , time course , and method of administration . in summary , although rarely encountered , csa 0.05% ophthalmic emulsion may induce the growth of eyelashes , and we believe ophthalmologist should be mindful and inform patients treated with topical csa about this side effect .

purposethe most common ocular adverse event following the use of cyclosporine a ( csa ) 0.05% ophthalmic emulsion is ocular burning ( 17% ) . other adverse effects that have been reported include conjunctival hyperemia ( 1 - 5% ) , discharge , epiphora , eye pain , foreign body sensation , pruritus , stinging and blurred vision . here , we report a specific side effect of csa , namely eye drop - induced eyelash elongation in a patient with refractory giant papillary conjunctivitis.designobservational case report.methodscase report and review of the literature.resultsa 32-year - old female with giant papillary conjunctivitis on the left eye , who had undergone papillectomy 3 years previously and was refractory to topical steroid therapy , was treated with csa 0.05% ophthalmic emulsion ( restasis ) 4 times a day , preservative - frees artificial tears and gentamicin ophthalmic solution in the left eye . after 5 months of topical csa treatment , elongated eyelashes of her left eye were observed without other adverse effects.conclusionalthough hypertrichosis and trichomegaly have been documented in the literature as side effects of systemic csa , topical csa 0.05% eye drop - induced elongated eyelashes have not been reported , and we believe ophthalmologists should be mindful and inform patients about this specific side effect .

in recent years , corneal specialists have shown a fast growing interest in collagen crosslinking using the photosensitizer riboflavin and ultraviolet a light as a new therapeutic alternative for the treatment of progressive keratoconus . the aim of this treatment is to modify the biomechanical properties of the cornea , thus increasing its rigidity and preventing the progression of keratoconus.1 promising results have been published recently , and data from the dresden clinical study have shown stabilization of the disease , even after 5 years.2,3 it is necessary to follow strict treatment settings and patient eligibility criteria to avoid serious ocular side effects . the patient s cornea must be irradiated with a small peak - like sector of the ultraviolet a spectrum ( 370 nm ) with 3 mw / cm irradiance for 30 minutes , which corresponds to a dose of 5.4 j / cm.3 the ultraviolet a irradiance must be checked before each treatment using an ultraviolet a meter to ensure that the optimal irradiance dose is delivered . the photosensitizing riboflavin 0.1% solution must be applied 5 minutes before irradiation and every 5 minutes during irradiation . it is also essential to perform preoperative pachymetry on each patient to exclude those with extended areas of less than 400 m of stromal thickness . we describe how to perform collagen crosslinking in the protocol setting using a slightly modified ultraviolet a dermatological lamp and preparing the riboflavin 0.1% solution inhouse . we used a slightly modified 370 nm ultraviolet a lamp ( uv 109a , waldmann , villingen - schwenningen , germany ) designed for dermatological treatment ( figure 1a ) , covered with a black plastic adhesive tape leaving a small window through which ultraviolet light can reach the cornea ( figure 1b ) . the desired irradiance of 3 mw / cm is tested with a ultraviolet a meter ( waldmann ) before each treatment , thus establishing the optimal distance from the ultraviolet a lamp to the cornea . the lamp was held by a clamp mounted on an intravenous pole ( figure 2 ) . patients corneas were irradiated with 3 mw / cm of ultraviolet a ( 370 nm ) for 30 minutes . the riboflavin 0.1% solution was prepared just before treatment by mixing 10 mg of riboflavin-5-phosphate ( guinama , valencia , spain ) with 10 ml of dextran 20% solution . the solution was introduced into an empty 15 ml balanced salt solution bottle ( alcon laboratories , fort worth , tx , usa ) wrapped in aluminum foil to avoid exposure of the riboflavin to ambient light ( figure 3 ) through a 0.22 m sterilization filter ( millipore corporation , bedford , ma , usa ) . the riboflavin 0.1% solution was applied 5 minutes before irradiation and every 5 minutes during irradiation . thus far , we have successfully treated 12 patients with progressive keratoconus using this ultraviolet a lamp and riboflavin 0.1% solution ( figure 4 ) prepared inhouse . the riboflavin solution was also easily prepared in the clinical pharmacy of the hospital by trained personnel . one important issue to be mentioned is the fact that the limbus should be protected against irradiation , and a round 8 mm hole in the tape that covers the lamp could be made for this purpose . the price of a commercially available ultraviolet a lamp designed for collagen crosslinking is approximately 15,000 , and the riboflavin solution prepared inhouse costs approximately 60 per patient . the price of the slightly modified ultraviolet a dermatological lamp we used is approximately $ 400 , the riboflavin 0.1% solution we prepared inhouse costs $ 2.20 per patient , and the ultraviolet light meter price costs around $ 200 . this provides ophthalmologists worldwide with an affordable alternative device for collagen crosslinking , especially for those in developing countries or places where resources are limited .

in this paper , we describe an original , affordable , and available device for performing collagen crosslinking . this is carried out in a safe and reproducible way by means of a simple modification to an affordable ultraviolet a lamp and by preparing riboflavin 0.1% solution inhouse .

attention - deficit / hyperactivity disorder ( adhd ) is a behavioral disorder usually diagnosed during childhood . it is characterized by developmentally inappropriate symptoms of inattention , hyperactivity and impulsivity and significant impairment in multiple domains of functioning . diagnostic and statistical manual of mental disorders ( dsm - iv ) diagnostic criteria define three adhd subtypes based on the presence of inattention and hyperactivity / impulsivity symptoms : predominantly inattentive type , predominantly hyperactive / impulsive type , and combined type . a recent us national epidemiological study of 8- to 15-year - old school children found an 8.7% prevalence of adhd , with the predominantly inattentive type most prevalent ( 4.4% ) among the adhd subtypes ( combined type , 2.2% ; predominantly hyperactive - impulsive type , 2.0% ) . the behavioral manifestations of adhd inattentive type differ from those associated with combined and hyperactive / impulsive type . children with combined and hyperactive / impulsive type exhibit problems with behavioral inhibition , including interrupting , blurting out , getting out of their seat at inappropriate times , and playing loudly . in contrast , the primary difficulties of children with inattentive type are non - disruptive in nature and are related to planning and organizing actions . children with inattentive type often forget to complete or lose assignments , procrastinate , complete work carelessly , have difficulty planning for the completion of long - term projects and studying for tests , and have problems keeping materials organized [ 7 - 9 ] . three treatments qualify as well - established interventions for children with adhd : psychopharmacological therapy , behavioral parent training , and behavioral classroom management . psychopharmacological therapy ( e.g. stimulant medication ) is the most utilized intervention modality for treating patients with adhd and produces marked improvements in sustained attention , impulse control and noisy and disruptive behaviors . while psychopharmacology ameliorates some of the core symptoms of adhd inattentive type , there is minimal evidence to suggest that medication promotes improvements in the ability to effectively plan and organize materials and/or actions . as such , stimulant medication is only minimally effective in improving important areas of functioning such as long - term academic achievement . similarly , evidence - based behavioral treatments , such as parent training and classroom management , primarily target impulsive and disruptive behaviors and do not focus on the problematic behaviors associated with inattentive type . in the past 2 years , several advances have been made in the treatment of behaviors exhibited by children with adhd inattentive type . interventions have been evaluated that target planning , materials organization , time - management , and homework management skills . these interventions typically focus on behavior in the school setting as demands for these skills are greatest at school and can lead to significant academic impairment . children are taught systems for organizing school materials and managing / planning homework responsibilities . checklists with operationalized definitions of behavior are used to monitor skills implementation . similar to existing evidence - based interventions for adhd , these interventions incorporate behavioral therapeutic techniques , such as rehearsal , prompting , shaping and contingency management , to teach and promote skills use and their generalization . for example , point systems or token economies are often utilized to monitor and reward adherence . interventions have been evaluated that target multiple aspects of organization and planning , including : classroom preparation , organization of bookbag , binder , and locker , and planning and tracking homework assignments and long - term projects . these interventions are associated with short - term gains on process measures of materials organization , homework management , planning and procrastination . some of these improvements appear to be sustainable for children with adhd as assessed at 8-week and 3-month follow - ups . at this point , it is unclear if gains on process measures ( e.g. materials organization or homework management checklists ) are ultimately associated with improvement in functional outcomes such as school performance . only a subset of studies completed to date included both process measures and measures of functional impairment . documented improvements in organizational skills , parent ratings of homework problems , and grade point average . however , the gains in grade point average were relatively small . pfiffner et al . documented improvements in organizational skills and a clinician completed clinical global impression , and these gains were maintained at a 3- to 5-month follow - up . abikoff and gallagher showed improvements in both organizational skills and parent - rated homework problems , but it is unknown if improvements in homework problems resulted in overall academic improvements . hence , alternative explanations for observed patient improvements are possible ( e.g. hawthorne effects ) . in addition , generalization across settings was not formally assessed in most of the studies . it would be important to determine if skills generalize across classrooms or from school to home . the behavioral difficulties of patients with adhd inattentive type may be markedly different from those of children with combined or hyperactive / impulsive type and the targets of treatment will need to be tailored accordingly . while behavioral interventions targeting organization , planning and time - management can not be classified as a central challenge will be figuring out how to disseminate these interventions into clinical practice . the interventions evaluated to date have varied in where the intervention is delivered ( e.g. school , home , or clinic ) , and dissemination models will need to account for these factors . providing intervention directly in the setting where the skills are most relevant / problematic ( e.g. school ) should allow clinicians to achieve higher levels of skills generalization . to promote dissemination , intervention protocols should be developed that do not require a high degree of clinical specialization to implement and that can be implemented directly in the setting of interest .

the behavioral difficulties of children with attention - deficit / hyperactivity disorder ( adhd ) inattentive type differ from those of children with adhd combined or hyperactive / impulsive type . existing evidence - based interventions primarily target the disruptive and impulsive behaviors exhibited by children with adhd combined and hyperactive / impulsive type . a number of recent advances have been made in the non - pharmacological treatment of behavioral difficulties associated with adhd inattentive type . additional research using randomized controlled research designs and long - term follow - up evaluation is necessary before these interventions may be considered established evidence - based interventions for patients with adhd inattentive type .

ureteral avulsion is conventionally defined as injuries of the ureter caused by blunt ( non - penetrating ) trauma with acute deceleration or acceleration movement mechanism due to motor vehicle accidents . with the invention of endourology tools , ureteral avulsion can also occur as a result of stretching in the weakest point of the ureter . ureteral avulsion and perforation during ureteroscopy are the most common cause of iatrogenic ureteral injuries . the rate of ureteral injury dropped to 7% in 1990 due to increased surgical experience and development of more advanced uertroscopy tools . the incidence of ureteral perforation has been reported in 1.5% of such procedures of which 0.2% have required surgery . in spite of the prevalent use of ureteroscopy in the treatment of ureteral stones and replacement of open surgical procedures with ureteroscopic procedures , the rate major and severe complications such as avulsion and perforation commands attention [ 6 , 7 ] . ureteral avulsion from the pelvis can be managed by ureteral reimplantation , transureteroureterostomy and ureteroneocystostomy [ 810 ] . renal autotransplantation can be an appropriate whenever a considerable length of the ureter is lost or a previous surgery has failed . meanwhile , sometimes after such procedure renal impairment is encountered . in the present case , we report a successful autotransplantatioin in a patient who underwent urgent operation following avulsion of the ureter at a distance of 4 cm from the pelvis . surgeons should be aware of possible complications attributed to transureteral lithotripsy ( tul ) and their knowledge and expertize in managing such complications . the patient was a 51-year - old lady with left flank colicky pain associated with nauseas and vomiting for a month . study of kub ( kidney , ureter , and bladder x - ray ) and non - contrast abdominopelvic spiral ct scan images revealed a 1-cm proximal left ureteral stone ( fig . 1 ) . accordingly the patient was selected to undergo tul and ureteral stenting . figure 1:abdominopelvic spiral ct scan of the patient before surgery . abdominopelvic spiral ct scan of the patient before surgery . due to a narrow ureteral lumen proceeding with the procedure seemed impossible and we decided to terminate the surgery . while taking out the ureteroscope we noticed avulsion of the ureter at a point approximately 4 cm from ureteropelvic junction . the avulsed ureter was handing on the tip of the ureteroscope and the avulsed fragment came out along with the ureteroscope . after thorough evaluation of various possible methods to approach the problem , the patient was brought to a left flank position from her original lithotomy position . with a left intercostal incision nephrectomy was done . subsequently , the position of the patient was changed to supine and a gibson incision was made over the right lower quadrant of the abdomen . following dissection of the iliac vessels , renal vessels were anastomosed to the iliac vessels ( renal artery to the internal iliac artery and renal vein to the external iliac vein ) . using the lich given the fact that only a short segment of the avulsed ureter ( 4 cm ) was left , we considered implantation of the ureter to the bladder . a drainage tube was inserted to drain any possible bleeding from the operation site and then the surgical wound was closed . the patient was discharged in a good general condition after 3 days and the drainage tube removed . an ultrasound imaging study of the genitourinary system 8 months into the patients follow up showed normal size , echo and cortical thickness in the operated kidney after renal autotransplantation . in follow up , 2 months after renal autotransplantation , the arterial and venous flow of the transplanted kidney were normal on color doppler ultrasound imaging ( figs . 2 and 3 ) . the mean parenchymal arterial vascular resistance in the transplanted kidney was 0.63 which was in the normal range . the height , anteroposterior diameter and cortical thickness of the transplanted kidney were 117.41 mm and 13 mm , respectively . the values of urea and creatinine 3 months after transplantation were 26.4 and 1.09 , respectively . size , echo and cortical thickness of the transplanted kidney were reported normal in the follow up ultrasound 8 months after autotransplantation ( fig . figure 3:color doppler ultrasound of the transplanted kidney 2 months after surgery in the zoomed area of the image . figure 4:gray - scale ultrasonography of the patient 's transplanted kidney 8 months after surgery . color doppler ultrasound of the transplanted kidney 2 months after surgery in the zoomed area of the image . ureteroscopy is widely used for lithotripsy procedure and resection of malignant tumors , but this method may lead to urethral mucosal trauma , hematuria , ureteral stenosis , urinary tract infection and ureteral perforation . among all complications of ureteroscopic lithotripsy procedure , complete avulsion of the proximal ureter is one of the most challenging that happens in 0.060.45% of the cases . it is evident that appropriate decision making and a timely surgical intervention can prevent the need for nephrectomy and possible complications in the future . in general , management of ureteral avulsion depends on the location of injury , the length of the traumatized ureter , time of diagnosis , patient 's age and general health . boari flap and psoas hitch techniques are used in proximal and distal injury of the ureter . finally , in the absence of the previous conditions , autotransplantation can be done as a vital method . it is evident that such surgical procedures require substantial expertize among all healthcare providers , a patient in a suitable physical status and proper age , and an accurate and timely decision making . autotransplantation was done in our patient after quick evaluation and review of different options to repair a traumatized ureter and clinical evidences supported the accomplishment of a successful outcome .

abstractthe patient was a 51-year - old lady with left flank colicky pain accompanied with nausea and vomiting for a month . transurethral lithotripsy and ureteral stent placement was considered for the patient . the patient had a narrow ureteral lumen and while bringing the ureteroscope out , ureteral avulsion occurred at a distance approximately 4 cm from ureteropelvic junction . after bringing the uretroscope out , the avulsed ureter was observed hanging at the tip of the ureteroscope . anastomosis of the ureter to the bladder was accomplished with lich gregoir technique . a drainage tube was inserted at the site of incision and the surgical wound closed . the patient was discharged with acceptable general condition after 3 days and the drainage tube removed . the ureteral stent was removed 4 weeks later by cystoscopy . an ultrasound imaging study of the genitourinary system 8 months into the patients follow up showed normal size , echo and cortical thickness in the operated kidney after renal autotransplantation .

nevertheless , completely laparoscopic colorectal procedures have been adopted in clinical practice by a limited number of surgeons . this is possibly due to increased operating time and costs , to the fact that their relative benefit is as yet unclear , and that these procedures are technically demanding . the latter reason prompted this experimental study , which was carried out to evaluate whether a previously described intracorporeal approach to sigmoid resection could be facilitated and applied to the rectum in a porcine model . ten domestic pigs with a median weight of 40.7 kg ( range 38.5 - 43 kg ) were anesthetized with intubation and ventilation with halothane . prior to surgery , the animals were fasted for 48 hours and kept on fluids ad libitum and received a 2-liter enema . pneumoperitoneum was induced using carbon dioxide insufflated to a pressure of 10 mm hg by placement of a trocar in the infraumbilical skin using a cut - down technique . two 12 mm ports were then inserted in the left hypocondrium and in the midline cephalad to the umbilicus . due to porcine anatomy , a 33 mm cannula ( endopath , ethicon endosurgery , cincinnati , oh ) was inserted in the right iliac fossa rather than suprapubically . all subjects were turned into a steep head - down position in addition to a right lateral tilt until no small bowel was seen in the field . a surgical technique previously described for laparoscopic sigmoid resection with intracorporeal colorectal anastomosis and specimen removal via a suprapubic incision once the proximal site of bowel division was identified , a laparoscopic pursestring clamp ( ethicon endosurgery , cincinnati , oh ) ( figure 1 ) was applied to the colon , and two straight needles ( placed at opposite ends of a monofilament suture and held in position against the clamp by an endoloop ) were passed through the clamp and retrieved through a port , as previously described . the colon was divided with scissors against the pursestring clamp after having been ligated with an intracorporeally knotted suture . the lower third of the rectum was transected with an articulating endoscopic stapler ( ets flex 35 , ethicon endosurgery , cincinnati , oh ) . a plastic bag allowed temporary intra - abdominal storage of the specimen . the rectal stump was irrigated by 1uminal wash - out in order to verify that the linear staple row was tight . the anvil of a circular stapler ( ecs 25 ethicon endosurgery , cincinnati , oh ) held by a modified allis - clamp was inserted through the 33 mm port into the colon and the pursestring was tied intracorporeally around the anvil notch . the same circular gun with a spike fixed to its shaft was introduced per anum ( figure 2 ) , and a doublestapled colorectal anastomosis was performed according to a previously - described technique . the integrity of the anastomosis was checked by irrigation with methylene blue after the application of a noncrushing intestinal clamp just proximal to the circular staple line . the specimen , in a plastic bag , was delivered through the 33 mm port . circular stapler with spike fixed to its shaft is advanced behind the staple line of the rectal stump . complete proximal and distal doughnuts were obtained in all cases and anastomoses were all methylene blue tight . the mean anastomotic level from the anal verge was 5.2 cm ( range 4 - 6 cm ) . all porcine subjects had an uneventful 5-week postoperative course at the end of which both laparoscopy and rigid proctoscopy were carried out . at celioscopy there were no adhesions or other abnormalities at the anastomotic site , while endoscopy revealed normal suture lines . histology of colorectal anastomoses revealed healing mucosa with neither foreign body reactions nor thickening of bowel wall at anastomotic site . transection of the rectum at a lower level can be achieved inserting an open - surgery articulating stapler through a suprapubic incision . however , this approach requires a third stapler ( in addition to endoscopic linear and circular staplers ) , re - establishing pneumoperitoneum twice , and manipulation of an open - surgery stapling device with laparoscopic graspers . the results of the present experimental study show that the use of a laparoscopic articulating stapling device helps to overcome these downsides and allows secure division of the low rectum . intracorporeally hand - sewn pursestring sutures are usually fashioned with curved or ski - shaped needles on the proximal colon , but this is a time - consuming procedure . the introduction of the t - needle technique ( endo - stitch , ussc , norwalk , ct ) failed to facilitate this procedure . in fact , since the t - needle should not be detached from the instrument 's jaws ( unless for reloading ) , the anvil shaft must , at a certain point , be loosened from the allis - clamp to allow 360 suturing and tying around the pursestring notch of the anvil . , that the use of a laparoscopic pursestring suture clamp seems to facilitate the construction of intracorporeal pursestring sutures and results in a safe colorectal anastomosis . according to previously described techniques , once the circular stapling gun 's central spike ( auto suture , pceeatm , ussc , norwalk , connecticut ) is advanced behind the staple line of the rectal stump , the spike must be disconnected and removed through a port ( figure 3 ) . the use of a gun adapted for laparoscopy ( with a spike fixed to its shaft ) ( figure 2 ) eliminates the need for retrieval of the spike . the present study shows that a previously described intracorporeal approach to sigmoid resection can be facilitated and applied safely to the rectum in a porcine model . however , further experimental and clinical comparative studies are needed with regard to safety , time - frame and costs of the construction of intracorporeal pursestring sutures .

background and objectives : an experimental study was undertaken to evaluate whether a previously described technique for laparoscopic sigmoid resection with intracorporeal resection - anastomosis and specimen removal via a suprapubic incision could be facilitated and applied to the rectum.methods:ten domestic pigs ( median weight 41 kg ) underwent low anterior resection of the rectum , which was transected with an articulating endoscopic stapler . pursestring sutures were fashioned intracorporeally with a laparoscopic pursestring clamp . the anvil of a circular stapler was inserted through a 33 mm port into the colon and pursestring tied intracorporeally . a circular gun with a spike fixed to its shaft was introduced per anum and a double - stapled anastomosis performed.results:complete doughnuts were obtained in all cases and anastomoses were all methylene blue tight . all porcine subjects had an uneventful 5-week postoperative course . the median anastomotic level from the anal verge was 5.2 cm . histology of colorectal anastomoses revealed healing mucosa.conclusions:the use of articulating endoscopic stapler , laparoscopic pursestring clamp , and circular stapler with a spike fixed to its shaft seems to facilitate a previously described intracorporeal approach to sigmoid resection which was safely applied to the rectum in a porcine model .

several studies reported histophatological observations during influenza a / h1n1 2009 pneumonia : the major finding was diffuse alveolar damage ( dad ) [ 14 ] . type ii pneumocytes were considered the main target of influenza a / h1n1 infection . a limited number of observations described in vivo samples , such as bronchoalveolar lavage ( bal ) [ 68 ] . therefore , it may play a role in further understanding the pathogenesis of new viral strains . in this case series we present the results of bal performed in three patients with severe influenza a / h1n1 2009 pneumonia complicated by acute respiratory distress syndrome ( ards ) . between august and december 2009 we performed a bronchoscopy - guided bal of three patients admitted to the san gerardo hospital , monza , italy , for influenza a / h1n1 virus pneumonia with subsequent development of ards . all three patients required intensive care unit ( icu ) admission and received mechanical ventilation ( mv ) and extracorporeal respiratory support ( ecmo ) . influenza a / h1n1 diagnosis was confirmed by a / h1n1 virus rna detection on nasal swab samples using influenza a / h1n1 2009 rrt - pcr ( real - time reverse - transcriptase - polymerase - chain - reaction ) assay ( genexpert ) . bal differential cell count on cytocentrifugate was performed in the light microscopy ( lm ) by counting about 300 cells in random fields at 400x magnification . demographics , comorbidities , severity on admission , microbiological isolations , and outcome for each patient are described in table 1 . antiviral therapy with oseltamivir ( 150 mg twice daily ) was started at hospital admission simultaneously with rrt - pcr assay and was continued until rrt - pcr assay on nasal swab turned negative . large , atypical cells with plasmocytoid appearance were observed in all three specimens with similar percentages ( 8 , 9 , and 6% , resp . ) . at lm , they appeared as large , plasmoblastic / plasmocytoid - like cells with eccentric nuclei and paranuclear vacuoles , high ratio nucleus / cytoplasm , and intensely basophilic cytoplasm . immunocytochemical analysis on cytocentrifugate showed that these cells were negative for cd20 ( b lymphocyte marker ) and cd138 ( plasma cell marker ) . ultrastructural examinations at transmission em allowed us to observe multilamellar osmiophilic bodies in the cytoplasm of plasmoblastic / plasmocytoid - like cells . em showed in these cells small round cytoplasmic inclusions with irregular surface and a diameter around 60 nm ( figure 2 ) . they also showed cytoplasmic vesicles with a diameter around 100 nm and an irregular electron - dense core ( figure 3 ) . the main characteristic of the bal of our patients with influenza a / h1n1 pneumonia associated with ards was the presence of large cells with a plasmoblastic / plasmocytoid - like appearance , identified at em as atypical type ii pneumocytes . the first hypothesis regards the reparative action that type ii pneumocytes may have in dad . in our cases , stanley described morphological atypias , such as increased nuclear - cytoplasmic ratio , in type ii pneumocytes found in bals during ards . these atypical pneumocytes are usually aggregated in clusters and are supposed to have a reparative role . all our patients developed ards and we observed pneumocytes aggregated in clusters in the bals performed earlier ( cases numbers 2 and 3 ) . according to this first hypothesis , the atypical cells we observed could be described as reactive immature type ii pneumocytes . type ii pneumocytes have been described as the main target of influenza a / h1n1 2009 infection . nakajima et al . in 2012 described four autopsy cases of influenza a / h1n1 with a histopathological pattern of acute dad who presented with influenza virus antigen - positive type ii pneumocytes , perhaps indicating a direct role of the virus - infected cells in the acute alveolar damage . no specific cytophatic effect or viral inclusion has been described so far at lm in lung tissue specimens during influenza pneumonia . several observations have been performed via em on autopsy specimens . during the recent influenza pandemic , mauad et al . found type ii pneumocytes with vesicles , approximately 100 nm in diameter , with an electron - dense center . bal and colleagues described cytoplasmic inclusions in pneumocytes , which ranged in diameter from 74 to 82 nm and showed surface spikes characteristic of influenza virus . em observations of our specimens revealed some atypical pneumocytes showing both small round cytoplasmic inclusions with an irregular surface and small vesicles ( similar to those described by bal and mauad ) . according to this second hypothesis , atypical type ii pneumocytes could be a specific morphological marker of influenza virus infection . from the data collected so far , we can not favor one explanation ; in fact , they could coexist in cases of severe influenza a / h1n1 pneumonia associated with ards . limitations of the present study include the following : first , the paucity of cases analyzed ; second , we did not perform immunohistochemical staining for h1n1 antigens on bal samples . the description of bal in a control group of patients with ards not associated with h1n1 pneumonia is beyond the scope of this case series . however , other authors described bal cytology in non - h1n1 ards , and although they reported morphological atypias in type ii pneumocytes , they did not describe these peculiar plasmoblastic / plasmocytoid - like cells [ 10 , 12 , 13 ] . the collection of the airway specimens of patients number 2 and number 3 , in which the nosocomial pathogens were isolated , took place after bal was performed . therefore , these pathogens most likely did not affect the cellularity in bal . in conclusion , plasmoblastic / plasmocytoid - like type ii pneumocytes characterize the bal of our patients with influenza a / h1n1 2009 pneumonia associated with ards . they could represent a pathognomonic marker of influenza virus pneumonia as well as reparative cellular activation after dad . more observations of bal cytology in patients with influenza pneumonia are needed to understand their characteristics and role .

we present the results of bronchoalveolar lavage ( bal ) performed in three patients with severe influenza a / h1n1 pneumonia complicated by acute respiratory distress syndrome ( ards ) . light microscopy analysis of bal cytocentrifugates showed the presence of characteristic large , mononuclear , plasmoblastic / plasmocytoid - like cells never described before . via transmission electron microscopy , these cells were classified as atypical type ii pneumocytes and some of them showed cytoplasmic vesicles and inclusions . we concluded that plasmoblastic / plasmocytoid - like type ii pneumocytes might represent a morphologic marker of a / h1n1 influenza virus infection as well as reparative cellular activation after diffuse alveolar damage .

the year 2006 may be remembered in history as the year of the amphioxus . european agencies failed to fund ( or we european researchers failed to convince the agencies of the importance of the amphioxus genome ) . fortunately , at the other side of the atlantic , linda holland ( scripps institution , university of california ) and jeremy gibson - brown ( washington university ) did not fail . they collaborated on a white paper to the joint genome institute of the department of energy 1 that was supported by the relatively few labs concentrated on amphioxus research . [ ricard albalat ( spain ) , marianne bronner - fraser ( usa ) , hector escriv ( france ) , jordi garcia - fernndez ( spain ) , kaoru kubokawa ( japan ) , vincent laudet ( france ) , thruston lacalli ( canada ) , leo pezzement ( usa ) , georgia panopoulou ( germany ) , mario pestarino ( italy ) , pierre pontarotti ( france ) , t.v . ventakatesh ( usa ) , hiroshi wada ( japan ) , ulrich welsch ( germany ) , kinya yasui ( japan ) , and shi - cui zhang ( china ) ] . a number of outstanding scientists outside the amphioxus community also gave support to the proposal ( the list is available in ref 1 ) . that the amphioxus genome was crucial to the understanding of the origin of the human genome maybe was not clear to funding agencies but it was for the human genome consortium , as in the landmark human genome paper in 2000 2 : our results so far are insufficient to settle whether two rounds of wgd occurred around 500 myr ago [ ... ] . another approach to determining whether a widespread duplication occurred at a particular time in vertebrate evolution would be to sequence the genomes of organisms whose lineages diverged from vertebrates at appropriate times , such as amphioxus ) . led by dan rokhsar ( jgi ) , and also thanks to the est collections made by the team of nori satoh ( kyoto university ) in collaboration with jr - kai yu and linda holland ( university of california ) we are about to know it : are we , human , octaploid ? and which were the gene novelties ( if any ) that characterised chordate evolution ? coming soon , very soon ... and second , the big drama with amphioxus : how they do it . to be a model , a genetic , developmental and functional one , amphioxus has to allow researchers to use its eggs and sperm ! and they do it not very often , the american species branchiostoma floridae once every two weeks in the summer , and not much more often the eastern species b. belchei . still , pioneer experiments of gene knockdown , reporter constructs , and cell lineage have been performed 3 - 5 . as with the genome , there is a big new , again not thanks to the european funding agencies , but to the effort of european research groups . an amphioxus consortium headed by hector escriv ( banyuls sur mer ) and composed by members of the labs in barcelona , lyon , paris and genova have been , during three years , trying to understand the reproductive behaviour of the european amphioxus , b. lanceolatum ( fig . now , nearly every day , we are able to obtain eggs and sperm on demand during the breeding season ( april - july ) 6 . this is possible not only in place , at the laboratoire arago , the marine station at banyuls sur mer . but also in our laboratory in barcelona . as far as we know , this is the only dry lab ( not a marine station ) in where amphioxus eggs and sperm are obtained in a controlled manner , a small but big step towards the dream : developing amphioxus as an experimental model system ( fig . most of the amphioxus researchers that have fought for the genome and the embryos are giving their particular data or views in their particular area of research . this second part focuses particularly in the nervous system , one of the major steps forwards to the complexity of vertebrates . homologies , innovations , brain , neural crest , colinearity , ... , interesting , promising and provocative hypothesis arise from the study of our lovely little lancelet . genome by the corner , embryos by the hand , things are getting better , every day . amphioxus in the lab : banyuls ( left ) , barcelona ( center ) and egg injection system ( right ) . developmental series of the amphioxus branchiostoma floridae . photograph courtesy of hector escriv .

the genome of the amphioxus is on the horizon . with linda holland and jeremy gibson - brown at the forefront , with all the amphioxus community behind , and with the joint genome institute , the amphioxus genome will see the light this year , 2006 . hope that it will reflect the prototypical preduplicative genome of vertebrates . it may answer definitively what the human genome did not : are we vertebrates octaploid ? will it shed light on the novelties that helped non - chordates to be chordates ? and more , will amphioxus , with a simpler genome , be developed to a senior experimental model system , allowing the testing of molecular functions in a non - duplicated genome background and allowing genetic modification to recapitulate evolution ? thanks to an outstanding collaboration between labs , the laboratory culture of amphioxus is underway after years of hard work in the field . 2007 looks promising for amphioxus research .

carbohydrates play critical roles during pollen growth and development , then in the final phase prior to dispersal carbohydrates change to prepare pollen for dispersal . following dispersal and during pollen storage , the various metabolites comprising the carbohydrate pool change in relative proportion , a behavior that is understood as an adaptation for sustaining pollen viability in time . sucrose in particular may be of particular importance during pollen storage , as variations among species for pollen desiccation tolerance have been linked to sucrose content . sucrose may replace water to preserve native protein structures and spacing between phospholipids in the plasma - membrane during dehydration . the role of starch and sugars in pollen development , dispersal , and maintenance of viability has not been determined for any member of the cycadales , and is therefore a focus of needed research . several authors have conducted robust surveys and noted that starchy pollen occurred disproportionately among anemophilous species . representatives from the cycadales were absent from these surveys and this should be corrected , as cycad pollination studies have only recently illuminated the sophisticated pollination syndromes that characterize the threatened plant group . although arthropods were collected from reproductive structures on various cycad taxa in the past , the scientific community largely ignored their potential role as pollinators due to the established belief that all cycads were anemophilous . the pendulum swung in the 1980s and 1990s when extensive experimental evidence confirmed mutualisms between cycad species and specialist insect pollinators . more recently , ambophily has been confirmed or proposed for several cycas species where wind has been shown to augment insect pollination . furthermore , variations in aggregation tendencies and settling velocity have been reported for pollen from various cycad species . this is an opportune time to add members of the cycadales to studies that have linked the relationship of pollen carbohydrates to insect versus wind pollination strategies . the insect pollinators of cycads gather on male cones , where the adults socialize , mate , and use the post - dispersal male strobilus tissue as larvae food ( fig . the study of idioblasts within cycad sporophyll tissues has illuminated intricacies in how general tissue chemistry may interact with pollinator feeding behaviors . for zamia furfuracea , these idioblasts remained intact in the male sporophyll tissue but appeared to release their contents into female sporophyll tissue prior to ovule receptivity . the authors concluded that toxins remained sequestered within the idioblasts for male tissue only , such that the pollinators could feed on parenchyma tissue around the idioblasts or consume intact idioblasts which protected insect metabolism from the toxins after consumption . many but not all cycad species studied in this context exhibited similar idioblast traits . for example , cycas rumphii and stangeria eriopus strobili did not contain identifiable idioblasts . in contrast , macrozamia lucida and microcycas calocoma exhibited idioblast breakdown prior to pollination stage for both male and female sporophyll tissues . the interactions among carbohydrates , nutrients , and toxins within cycad strobili tissues in relation to these idioblasts may prove to be of crucial importance for developing a full understanding of how pollinators feed on the plant 's reproductive tissues while enacting effectual pollination . conservationists in need of improved knowledge of various pollination syndromes within the cycadales will require more studies within this context . left : microstrobilus tissue is tunneled and consumed by larvae ( arrows ) immediately after pollen dispersal . right : within days the entire microstrobilus is reduced to frass and pupation ( arrow ) heralds in a new generation of pollinators . the role of cycad sugars in mediating left : microstrobilus tissue is tunneled and consumed by larvae ( arrows ) immediately after pollen dispersal . right : within days the entire microstrobilus is reduced to frass and pupation ( arrow ) heralds in a new generation of pollinators . the role of cycad sugars in mediating the majority of cycad pollinators consume strobilus tissue , but direct consumption of cycad pollen may occur for some cycad pollinators . cumulative research on pollen digestion the addition of cycads to studies that determine carbohydrate and nutritional quality of pollen will greatly improve this line of research whether the mutualisms represent ancient associations that pre - dated angiosperms or examples where the contemporary mutualisms were recently derived from an initial antagonistic relationship . the reproductive structures of cycad species exhibit thermogenesis and volatile emissions at the time of pollination , traits that mediate pollinator behavior and maintain pollinator specificity . research is accumulating in areas such as modeling the plant behavior and parsing the influence of specific volatiles on insect behavior . enacting synchronized thermogenesis and volatile biosynthesis is an exceedingly expensive plant behavior , and no studies have determined the tissues of residence , quantities , and stoichiometric relations of the carbohydrate reserves that are mobilized to fund those activities . droplets have been documented at the micropyle location on cycad ovules , and these droplets contain metabolites that may provide a reward for pollinators . studies that marked pollinators then used deposition of the markers to track pollinator behavior have confirmed that the pollen they vector is trapped by the ovule droplets . this remarkable female strobilus behavior represents a system that would be interesting to study in relation to specificity of pollinator attraction , diversity of sugar rewards , and cycad phylogeny . droplets may exhibit taxon - specific carbohydrates that are suited to attracting and nourishing specific specialist pollinators . our recent report of fructose , glucose , and sucrose content in cycad tissues sets the stage for designing continued research on how non - structural carbohydrates are involved in cycad pollination biology . moreover , cycad horticulturists routinely harvest , store , and ship pollen prior to its use for successful pollination . improved understanding of how carbohydrates and other factors influence pollen viability and longevity would improve protocols for artificial pollination . finally , the risks associated with coextinctions are very real during this phase of the anthropocene , and species with complex life history traits , such as cycads , appear to be at greater risk for direct involvement in coextinctions . an increase in knowledge of how cycad carbohydrates influence successful pollination relationships may help reduce the risks of coextinctions in these mutualisms that support contemporary cycad biology .

cycad biology , ecology , and horticulture decisions are not supported by adequate research , and experiments in cycad physiology in particular have been deficient . our recent report on free sugar content in a range of cycad taxa and tissues sets the stage for developing continued carbohydrate research . growth and development of cycad pollen , mediation of the herbivory traits of specialist pollinators , and support of expensive strobilus behavioral traits are areas of cycad pollination biology that would benefit from a greater understanding of the role of carbohydrate relations .

the objective of this study was to produce maps that could be integrated into the sci - supported national intervention strategies and that explicitly represent uncertainties in spatial predictions so that national control managers could judge the quality of the evidence upon which the strategies will be based . the sci - supported programs involve mass distribution of praziquantel ( for urinary and intestinal schistosomiasis ) and albendazole ( for soil - transmitted helminths ) . the parasitic infection with the highest prevalence is urinary schistosomiasis , caused by flukes ( schistosoma hematobium ) , and the programs are planned to control this disease ( 2 ) . parasitologic data were collected in coordinated school - based field surveys in burkina faso , mali , and niger ( figure 1 ) during 20042006 ( preintervention ) by using standardized protocols ( available on request ) . the collated dataset covered a spatially contiguous area , 2,750 km 850 km , and included the infection status of 27,939 school - age children in 418 randomly selected locations . infection status was defined according to egg count determined by microscopic examination of urine samples ; > 1 s. hematobium eggs indicated infection . prevalence of infection with schistosoma hematobium at 418 survey locations in burkina faso , mali , and niger , 20042006 . spatial prediction was based on a logistic regression model ( table ) , constructed by using the software winbugs , version 1.4.2 ( mrc biostatistics unit , cambridge and imperial college , london , uk ) . the model had infection status as the binary outcome variable , age and sex of the survey participants as individual - level fixed effects , and distance from perennial water body ( derived from electronic maps obtained from the food and agriculture organization ) and land surface temperature ( lst ; with a quadratic term ; see hay et al . for details on how these data were derived ) as survey location level fixed effects . the model also included a geostatistical random effect for residual spatial clustering of infection prevalence ( 11 ) . values for the fixed effects are odds ratios ; note the odds ratios for the climate variables are on a common scale , where the variables were standardized to have a mean = 0 and sd = 1 . the reference group for sex was boys and for age was 68 y. the number of children found to be infected with s. haematobium was modeled by using a binomial distribution described by the proportion infected and the total number sampled in each survey location . the proportion infected was modeled by using logistic regression with an intercept , covariates ( sex , age , distance to perennial water body , land surface temperature , and a quadratic term for land surface temperature ) , and a random effect that described spatial correlation ( i.e. , clustering ) . model outputs were distributions ( termed posterior distributions ) that can be summarized by using the mean , sd , and 95% cri ( representing the range of values that contains the true value with a probability of 95% ) . a prevalence map for the study area was constructed , using the model , by predicting infection prevalence at the centroids of cells of a 0.15 0.15 decimal degree ( 18 km 18 km ) grid . this model was implemented with the spatial.unipred command of winbugs ( details are provided in the technical appendix ) . estimates from bayesian models are distributions ( termed posterior distributions ) that represent the probability of each of a range of plausible values being true for the variable being modeled . to quantify the uncertainties surrounding the model predictions , we plotted the probability of each prediction location having a prevalence > 50% , rather than mean predicted prevalence at each location . the probabilities were calculated from the posterior distributions of predicted prevalence at each location ( i.e. , if 95% of the posterior distribution of predicted prevalence was > 0.5 , the probability of prevalence > 50% at that location was 95% ) . cross - validation was done by randomly allocating survey locations to 3 groups and undertaking 3 separate runs of the model ; 1 of the 3 groups was sequentially omitted , and predicted prevalence at the omitted locations was determined by using the model . predicted prevalence was compared with observed prevalence , dichotomized , according to a 50% observed prevalence threshold . the comparison statistic was the area under the curve ( auc ) of the receiver operating characteristic , and a value of > 0.7 was considered to indicate acceptable predictive ability . an average auc was calculated across the 3 model runs . in the final model ( table ) , statistically significant correlations suggested that infection prevalence was higher in older boys and increased with proximity to perennial bodies of water , but no association was found between prevalence and lst . the range over which spatial correlation was > 5% ( chosen to indicate statistically important spatial correlation ) was 177 km , indicating the approximate radius of clusters . results of the validation analysis showed an average auc of 0.86 , indicating that the model had an acceptable predictive performance . bayesian probability maps were produced for each sex and age group , but for illustrative purposes we present predicted probability of prevalence > 50% in boys ages 1316 years ( the group with the highest infection prevalence ; figure 2 ) . large clusters of prediction locations with a high probability ( i.e. , > 50% ; indicative of low uncertainty ) of prevalence being > 50% intervention threshold were located in a mid - latitudinal band across mali , running from western to central regions , and in the niger river valley region of niger . smaller clusters were located in various parts of southern and eastern mali , northwestern and northeastern burkina faso , and south - central niger . predicted probability of prevalence of infection with schistosoma hematobium being > 50% in burkina faso , mali , and niger in boys ages 1316 years ; results are based on a bayesian geostatistical model . the red areas had a low degree of uncertainty that predicted prevalence was > 50% , and the blue areas had a high degree of uncertainty that predicted prevalence was > 50% . future schistosomiasis control plans should acknowledge uncertainties such as those presented in figure 2 . a possible approach would be to introduce a second threshold for the level of uncertainty that a location is above the intervention prevalence threshold ; if the uncertainty is greater than this second threshold , then the location is excluded until new evidence is obtained that confirms prevalence is above or below the intervention prevalence threshold . this second uncertainty threshold should be determined by the quantity of resources available for disease control and the level of decision risk deemed appropriate . in addition to providing an evidence base for distributing resources in 3 west african countries as part of the sci - supported national control programs , the maps presented here have a potential role in maintaining sustainability of schistosomiasis control after sci support ends ( sci is funded through 2009 ) . they can be used as advocacy tools for channeling funds to high - risk populations in the affected countries and , in the likely event that money for schistosomiasis control in these countries becomes more limited after sci support ends , they can be used to ensure that scarce governmental resources are distributed as efficiently as possible . national coordinators who might face accountability for targeted ( i.e. , unequal ) distribution of resources will benefit from the defendable , scientifically sound methods presented in this article . by focusing on uncertainty in spatial predictions , more flexible tools for disease control can be developed that allow the geographic dimensions of control programs to be scaled and modified according to available resources and acceptable levels of decision risk .

we aimed to map the probability of schistosoma haematobium infection being > 50% , a threshold for annual mass praziquantel distribution . parasitologic surveys were conducted in burkina faso , mali , and niger , 20042006 , and predictions were made by using bayesian geostatistical models . clusters with > 50% probability of having > 50% prevalence were delineated in each country .

a 94-year - old woman was referred to the general internal medicine service for functional decline secondary to longstanding knee pain . her medical history included osteoarthritis and her surgical history was significant for a right total knee arthroplasty in 1996 . on examination , her vital signs were normal ; she was afebrile , but her right knee was red , warm and swollen . her initial blood work showed an erythrocyte sedimentation rate of 54 mm / h ( normal 0 mm / h to 27 mm / h ) , a c - reactive protein level 52.6 mg / l ( normal 0 mg / l to 1 mg / l ) and a white blood cell count of 6.210/l ( normal 4.010/l to 10.510/l ) . a plain radiograph of the knee demonstrated loosening of the tibial component , with a large joint effusion believed to show metallosis a deposition of metallic debris in the periprosthetic soft tissues from abrasion of metallic components ( 1 ) . an aspirate of the knee revealed calcium pyrophosphate crystals , with a total nucleated cell count of 11.310/l and differential of 97% neutrophils . later , an enrichment broth subculture of the aspirate demonstrated growth of a gram - positive bacillus , which was identified as a lactobacillus species based on the following results : unidentifiable profile on rapid ana ii panel ( oxoid canada ) , catalase - negative , pyr - negative , lap - negative and vancomycin resistance . she presented six weeks later with escherichia coli bacteremia arising from an acute urinary tract infection . a reassessment of the right knee showed a marked increase in swelling with persistent pain and tenderness . infectious diseases was subsequently consulted to comment on the significance of the lactobacillus species that had grown on culture from the previous admission . a second aspirate was requested and grew the same organism , which was identified as a lactobacillus species . of note , there were no calcium pyrophosphate crystals apparent in this aspirate and the total nucleated cell count and differential were 3.110/l and 96% neutrophils , respectively . the isolate was sent to the ontario public health laboratory ( toronto , ontario ) for identification using 16s ribosomal rna sequencing ( 2 ) . 16s ribosomal rna sequencing revealed that the organisms from both knee aspirate cultures were identical and were subsequently identified as weissella confusa . susceptibility testing of the organism from both isolates was performed using the agar dilution method and minimum inhibitory concentrations for various antibiotics are summarized in table 1 . weissella was first described as a new genus in 1993 and was named after norbert weiss , a german microbiologist known for his contributions to lactic acid bacteriology . it is identified using 16s ribosomal rna sequencing to differentiate it from other organisms such as lactobacilli and other members of the family leuconostocaceae ( 3,4 ) . usual phenotypic identification methods have been shown to be ineffective and often lead to misidentified organisms ( 4 ) . fourteen species in total have been identified , of which two w confusa and weissella cibaria are clinically important because of their ability to infect human hosts and intrinsic resistance to vancomycin . weissella species are alpha - hemolytic , gram - positive coccobacilli that typically grow in chains and are catalase negative and bile esculin positive . what differentiates w confusa and w cibaria are the acidification of different sugars ( 5 ) . it was named confusa because it was often mistaken for members of the leuconostoc , pediococcus and lactobacillus genera ( 3 ) . w confusa has a widespread environmental distribution and has been found in a variety of foods including sugar cane , carrot juice , milk , fermented meats , garlic mix and banana leaves . congruently , it can be part of the normal microflora of the human intestine and can be found in human stool ( 4 ) . infection and immunocompromised , revealing a total of 19 cases in the english literature : 15 with bacteremia , two with endocarditis , one with osteomyelitis and one with a thumb abscess ( table 2 ) ( 412 ) . to our knowledge , we present the first case of a prosthetic joint infection caused by w confusa . in the largest case series , lee et al ( 10 ) reported 10 patients with bacteremia in a tertiary care centre in taiwan . risk factors for invasive infection in this series included an immunocompromised host , central line catheter insertion and concurrent polymicrobial bacteremia . these findings were similar to case reports by harlan et al ( 8) and salimnia et al ( 12 ) , who identified bacteremia in patients with hepatocellular carcinoma post - liver transplant and acute lymphocytic leukemia undergoing autologous stem cell transplantion , respectively . other case reports have included risk factors for immunocompromised states , which consisted of chronic alcoholism , previous long - term steroid use and severe burn patients ( 7,12 ) . similarly , gastrointestinal manipulation via endoscopy or surgery may be one of the routes of translocation into the bloodstream because six of the reported 19 cases had documented medical procedures within three months of infection ( 811 ) . w confusa is intrinsically resistant to vancomycin and exhibits high minimum inhibitory concentrations ( 412 ) . this is important to recognize because clinicians often use vancomycin empirically as a treatment option in the immunocompromised patient when cultures initially reveal a gram - positive organism in the blood . this can be fatal in cases in which recognition of the organism is not identified in a timely fashion . studies have reported high levels of resistance to trimethoprim / sulfamethoxazole , metronidazole , teicoplanin , ceftazidime and ceftriaxone . in general , penicillin , clindamycin , erythromycin , daptomycin and the fluoroquinolones are effective agents for treating w confusa infections ( 10 ) . what was unique to our case was the lack of predisposing factors in our patient . while the patient did have evidence of a concomitant e coli bacteremia at her second presentation , she had already grown w confusa from the first aspirate of her knee six weeks previously . she received a one - week course of levofloxacin for her e coli bacteremia , which unfortunately , provided minimal clinical benefit to her prosthetic knee infection . she was discharged from hospital and returned to her long - term care facility in stable condition . given her premorbid state and nonambulatory status , it was decided in discussion with the patient and her family to not pursue further medical or surgical therapy for her prosthetic joint infection . w confusa is a rare , but well documented , cause of invasive infection in humans . w confusa infection can be treated with a variety of antimicrobials such as penicillins and fluoroquinolones ; however , it exhibits intrinsic resistance to vancomycin .

this article describes a case involving a 94-year - old woman with an infection of her knee , which had previously undergone total knee arthroplasty . following culture of aspirate from the knee , the causative organism was tentatively identified as a lactobacillus species . the infection was later determined to be caused by weissella confusa . the authors discuss the difficulties in identifying w confusa and present a literature review of infections caused by the species .

road traffic crash ( rtc ) is the main cause of non - intentional injuries and the second cause of deaths after ischemic heart diseases in iran . it accounts for more than 10% of total mortalities and about 23,000 deaths , in average , per year during the last decade . this is approximately equal to the number of deaths caused by the 2003 bam earthquake , one of the worst natural disasters of recent decades . these facts have made the rtc as a man - made disaster and real public health concern in iran . in line with the fourth i.r . iran 's national development plan ( ndp ) , the fifth ndp for 2011 - 2015 has emphasized on development of rural infrastructures , including rural asphalt roads . although this development has led to more traffic in rural roads , no significant improvement is observed in safe driving behavior and construction of roads in rural areas . in addition , compared with urban areas , rural people use older cars with less safety standards . the monitoring by traffic police and coverage of emergency medical services ( ems ) are also lower in rural roads than main roads . this article aims to provide the iranian policy makers with information on the burden of deaths that are caused by rural rtcs and demonstrates its association with expansion of the rural roads . we applied a retrospective analysis of secondary data to demonstrate the association of rural rtcs deaths with expansion of the rural roads in iran . the variables of interest included length of road ( rural and urban ) , number of death ( caused by rural and urban rtcs ) and number of population . the sources that we used for data collection were the iran 's high commission of road safety ( hcrs ) for length of road ( km ) , the iran 's forensic medicine organization ( fmo ) for the number of rtcs death and the iran 's statistical center for population data . international classification of diseases-10 is the basis for coding the causes of death by fmo . for the purpose of analysis , we illustrated the trends of length of rural road from 2005 to 2010 and their corresponding mortality . rates of traffic death in rural and urban roads were also estimated per 100,000 populations . for trend was the test of significance and p < 0.05 was considered as statistically significant . spss statistical software for windows ( version 11.0 , chicago , il ) was used for statistical analysis . the number of rtc deaths in rural roads increased from 1,672 in 2005 to 2,206 in 2010 . this was parallel to construction of the rural asphalt roads that was increased from 56,424 to 86,519 km during the same period [ figure 1 ] . the trend analysis showed a positive association between these two variables ( for trend = 3.93 , df = 1 , p = 0.04 ) . length of rural asphalt roads and number of deaths caused by rural road traffic crashes , iran , 2005 - 2010 the expansion of urban asphalt roads was also on an increasing trend from 71,711 to 77,964 km , but the number of traffic deaths in these roads decreased from 26,083 in 2005 to 21,043 in 2010 . from 2005 to 2010 , adjusted for 100,000 populations , the number of traffic deaths in urban roads decreased from 37.0 to 28.0 , while this number increased from 2.4 to 2.9 in rural roads [ figure 2 ] . our study showed an increasing trend of the rtc deaths in iran 's rural roads and its positive association with expansion of the rural roads . this is while , at the same period , the rate of traffic death in urban roads was on a decreasing trend . therefore , expansion of the road network is one of the most important prerequisites for development of economics and infrastructures in rural areas , the policy makers should take preventive measures in rural roads to improve the safe driving condition . in this line , iran 's ministry of road and urban construction is the lead agency for the hcrs . traffic police , ems and car factories are the main stakeholders of this national coordination body . to reduce the rate of deadly traffic crashes in rural roads , taking the coordinated measures by the hcrs ' members the hcrs may also rely on the successful experience in decreasing the rate of urban rtc deaths . according to available literature , the risk factors of deadly rtcs in rural roads of iran are unsafe roads , excessive speed , risky driving caused by inadequate knowledge and inappropriate risk perception , no use of seat belt and using of old cars with low safety equipments . effectiveness of the enforcement measures for control of rtcs in iran is rated six out of 10 , according to the world health organization . this is expected to be even lower in rural roads as they are less controlled by traffic police compared with freeways , highways and main roads . timely and quality provision of medical care is essential for saving lives in a rtc . in 2010 , the national emergency medical service was only able to cover 15% of minor roads , including rural areas . according to national ems , in addition to the current 1,028 ems stations , rural rtc is a concern not only in developing countries like iran , but also it accounts for the considerable number of death in developed countries . for instances , in germany , britain and the us , about two third of all rtc related fatalities occur on rural roads , which score badly when compared to the high quality motorway network in those countries . all these have said , there are evidences that fortunately show how the preventive measures can be effective in reducing rural rtcs . for example , in germany , reinforcement of speed limits and development of additional passing lanes were found effective . the expansion of rural asphalt roads exposes the people of rural areas with risk of severe rtc if effective preventive measures are not taken . to prevent from this threat , the iranian policy makers need to take the followings into consideration : public awareness , improving the safety of roads and vehicles , law enforcement , increasing coverage of police and ems

background : the 5th iran national development plan , 2011 - 2015 , has emphasized on expansion of rural asphalt roads . this article aims to illustrate the trend of deaths caused by rural road traffic crashes ( rtcs ) and its association with length of the rural roads in iran.methods:we carried out a retrospective analysis on secondary data for the period from 2005 to 2010 . the iranian forensic medicine organization , high commission for road safety and iran 's statistical center were the sources for the number of rtc death , length of the road and population data , respectively.results:number of rtc deaths in rural roads increased from 1,672 in 2005 to 2,206 in 2010 . this was associated with expansion of the rural asphalt roads ( p = 0.04 ) . the construction of urban asphalt roads was also on an increasing trend , but the number of traffic deaths in these roads decreased from 26,083 in 2005 to 21,043 in 2010 . adjusted for 100,000 populations , the number of traffic deaths in urban roads showed a decrease from 37.0 to 28.0 , while this number increased from 2.4 to 2.9 in rural roads during the study period.conclusions:although expansion of rural roads would contribute to economic development in rural areas , it exposes people to risk of severe rtcs if effective preventive actions are not taken . to prevent this threat , the iranian policy makers need to take the followings into consideration : public awareness , improving the safety of roads and vehicles , law enforcement , increasing coverage of police and emergency medical services .

this prospective interventional case series was approved by the review board / ethics committee of the ophthalmic research center of the university . five eyes of 5 patients with diagnosis of refractory csc ( lasting more than 1 year ) were included in this study . diagnosis was made by the history of recurrent blurred vision and metamorphopsia for more than one year , detection of neurosensory detachment in ophthalmoscopy and optical coherence tomography ( oct ) , and observation of active rpe leakage in flourscein angiography . exclusion criteria consisted of any accompanying macular disease , severe media haziness which precludes oct evaluation , and noncompliance . all eyes received a single injection of 0.05 ml ( 1.25 mg ) intravitreal bevacizumab ( avastin ; genentech inc . , south san francisco , ca , usa [ made for f. haffmann - la roche ltd . , basel , switzerland ] ) performed by a 30-guage needle through supratemporal quadrant 4 mm from the limbus under sterile condition . all patients underwent a through ophthalmic examination 1 day , 1 week , and 1 , 2 , and 6 months after the injection . best corrected visual acuity ( bcva ) of the eyes was checked by a masked optometrist . it was changed to the logarithm of minimum angle of resolution ( logmar ) scale for statistical purposes and compared at months 2 and 6 with the baseline values . central macular thickness ( cmt ) measured by oct ( 3d oct-1000 ; topcon corporation , tokyo , japan ) was performed at presentation and repeated 6 months after the intervention . it was measured in a 1-mm circle centered on the fovea by an optician who was masked to the study . none of the patients had history of intraocular surgery , diabetes mellitus , hypertension , cardiovascular disease and smoking . an increase of bcva was noticed during the follow - up in all eyes except case 1 . mean bcva at baseline was 0.60 0.25 that improved to 0.42 0.16 and 0.24 0.21 logmar up to the month 2 and 6 , respectively . this improvement at 2 months did not reach to a meaningful level ( p = 0.064 ) ; however , it was statistically significant at 6 months ( p = 0.025 ) . no recurrence was observed in any of the eyes during the follow - up period . central macular thickness decreased significantly from 370 65 m at baseline to 210 24 m at 6 months after injections ( p = 0.009 ) ( table 2 ) . none of the eyes had intraocular pressure rise ( > 21 mmhg ) or cataract progression during the follow - up period . they include acetazolamide , beta - blockers , vitamins , and non - steroidal anti - inflammatory drugs . there are some controversial recommendations in the literature on the use of laser photocoagulation in this field . some authors reporting that laser photocoagulation shortens the duration of disease and reduces recurrence rate , while others maintain that it does not affect final vision and recurrence rate . furthermore , laser may be associated with permanent scotoma which may enlarge over time with rpe scar expansion , as the possible development of cnv . it may hasten resolution of exudation by reducing choroidal blood flow and hence favoring cessation of leakage . most recently , several case series have reported the use of indocyanine green guided pdt in the treatment of chronic csc . ober et al . reported the successful treatment of focal rpe leaks in csc by pdt in a small pilot series which showed resolution and visual improvement . cardillo piccolino et al . performed indocyanine green guided pdt in 16 eyes with chronic csc and treatment resulted in complete resolution of serous retinal detachment 1 month after treatment in 75% of eyes . at 3 months after pdt , 69% of eyes had visual improvement of 1 or more lines . however , 31% of their cases developed secondary rpe changes at the site of pdt , which were thought to be due to hypoxic damage caused by choriocapillaris occlusion . our study showed a significant visual improvement and cmt reduction following single injection of ivb ( 1.25 mg ) in 5 cases suffering from refractory csc for more than one year . in a similar study on 5 cases with csc , torres - soriano et al . noticed an improvement in bcva , fluorescein angiographic leakage , and reduced or resolved neurosensory detachment . however , they injected 2.5 mg ivb and included cases with history of decreased visual acuity more than 3 months , recurrent episodes of csc or even acute cases with excessive discomfort about visual acuity . in a case . showed that in cases with chronic csc ivb injection improved bcva and reduced cmt . however , they performed multiple injections of 2.5 mg ivb at 6 to 8 week intervals ( range , 1 to 4 weeks ) . however , recurrence did not occur in any case of our study during follow up period . in summery , the present study demonstrated a promising effect of ivb in the treatment of refractory csc ; however , we can not make specific treatment recommendations based on this small , uncontrolled case series .

in a clinical case series , 5 patients with not - resolved central serous choroidoretinopathy ( csc ) lasting more than 1 year received one intravitreal bevacizumab injection ( ivb , 1.25 mg ) injection . all patients underwent a through ophthalmic examination 1 day , 1 week , and 1 , 2 , and 6 months after the injection . best corrected visual acuity ( bcva ) and central macular thickness were compared before and after treatment by optical coherence tomography . mean bcva was improved significantly ( p = 0.020 ) from 0.60 0.25 to 0.50 0.18 and 0.29 0.19 logarithm of minimum angle of resolution at 6 and 18 weeks , respectively . central macular thickness was also decreased significantly ( p = 0.010 ) from 370 65 to 208 23 m at 4 months . no recurrence was occurred during follow - up . ivb injection may have beneficial effect in the treatment of refractory csc .

the patient was a 25-year - old , right - handed woman who presented two months after the sudden onset of lower extremity paresthesias and sensory loss that extended into the abdomen and lower chest and one episode of urinary urgency and subsequent incontinence . her examination was notable for brisk lower extremity deep tendon reflexes but no other abnormalities . mri of the cervical and thoracic spine revealed both cervical and midthoracic plaques consistent with demyelination . a subsequent brain mri revealed five scattered , nonenhancing , white - matter lesions ; including a large ( approximately 5 mm in diameter ) lesion involving the corpus callosum and two lesions perpendicular to the left lateral ventricle . her b12 level , htlv-1 antibodies , vitamin e level , rpr , ana , and lyme titre were all within normal limits she was started on glatiramer acetate following her clinically isolated syndrome of partial transverse myelitis and mri findings confirming dissemination in space . two and a half years after her initial presentation , she developed right hemiparesis that resolved over a few days following treatment with intravenous steroids . she had a subsequent similar episode of left hemiparesis and hemisensory deficit two months after that , confirming a diagnosis of clinically definite multiple sclerosis . one year after her initial presentation , she developed a sudden onset of discomfort in her legs associated with an urge to move that was worse at night and impaired sleep . the symptom resolved while walking and had been present for only 3 - 4 days at the time of presentation . her last menstrual period was two weeks prior to this presentation , and she was faithfully using an oral contraceptive . her iron level was 108 mcg / dl , iron saturation was 28.6% , and ferritin was 94 ng / ml . her symptoms resolved a few days after a course of intravenous methylprednisolone ( 1 g daily for three days ) , and she had no recurrence over the next three years . restless legs syndrome is a movement disorder characterized by distressing urge to move the legs ( akathisia ) associated with discomfort and that is brought on by rest , relieved by movement or walking , and become worse at night or in the evening . her rather abrupt onset suggested the possibility of restless legs syndrome secondary to an underlying cause , such as pregnancy or iron deficiency . however , her diagnostic evaluation excluded these possibilities , and her clinical course and probable response to steroids suggested that her restless legs syndrome was due to an acute exacerbation of her multiple sclerosis . several studies have reported an increased incidence of restless legs syndrome in patients with multiple sclerosis , have suggested both a role for restless legs syndrome in ms - related fatigue , and have suggested that multiple sclerosis is a cause of secondary restless legs syndrome [ 25 ] . however , none of these studies describe self - limited restless legs syndrome occurring as an acute exacerbation of multiple sclerosis as in this case . manconi et al . demonstrated a correlation between rls and cervical cord lesion burden . this patient had a known cervical cord plaque , and this correlation might also explain why no new intracranial lesion was detected during her acute presentation with rls . restless legs syndrome is commonly idiopathic , but it has been associated with a number of conditions other than multiple sclerosis , including pregnancy , renal failure , peripheral neuropathy , and iron deficiency [ 1 , 7 ] . iron deficiency has been recognized in several studies as a cause for secondary restless legs syndrome , particularly at serum ferritin concentrations of less than 50 ng / ml [ 710 ] . the pathophysiology of even idiopathic restless legs syndrome is probably related to low brain iron levels . mri , csf , and neuropathological studies have all implicated central nervous system iron deficiency in the pathophysiology of restless legs syndrome , even in the absence of systemic iron deficiency [ 1116 ] . recently , there has been interest in the role of iron in ms and iron deposition because cerebral venous insufficiency has been suggested as a proposed mechanism for the disease . haacke et al . reported mri evidence of iron accumulation in the basal ganglia and thalamus of ms patients . furthermore , iron appears to be an important cofactor in cns myelination , and van toorn et al . have reported two cases of iron deficiency associated with tumefactive demyelination in children [ 2022 ] . the pathophysiology of ms consists of both an inflammatory demyelinating component and an axonal degeneration component . levine et al . reported normal csf ferritin , transferrin , and iron concentrations in ms patients with stable or relapsing and remitting ms , but elevated levels in the csf of patients with chronic progressive ms . therefore , it is likely that the pathophysiology of rls in ms patients is related to the inflammatory , demyelinating component of the disease which may be associated with low or normal cns iron levels , rather than due to axonal degeneration , which is associated with cns iron accumulation . the observation that rls is associated with spinal cord plaques would also suggest a relationship to the inflammatory and demyelinating component of disease . this patient , presenting with transient rls due to an acute , inflammatory exacerbation of her multiple sclerosis , further supports the notion that the pathophysiology of rls in multiple sclerosis is related to autoimmune inflammatory demyelination .

restless legs syndrome is common in patients with multiple sclerosis but has not been reported as occurring due to an acute , inflammatory , demyelinating attack . restless legs syndrome is known to be related to low brain iron levels . multiple sclerosis has been associated with the abnormal accumulation of iron in the chronic , progressive phase of axonal degeneration . iron deficiency may play a role in demyelination . this suggests that restless legs syndrome may be caused by the inflammatory , demyelinating component of multiple sclerosis rather than axonal degeneration . the author presents a case of self - limited restless legs syndrome occurring as an acute attack of multiple sclerosis , supporting the notion that inflammatory demyelination is the underlying pathophysiology of restless legs syndrome in multiple sclerosis .

sarcoidosis is a multisystemic , granulomatous disease of unknown etiology that may affect any organ system . it is characterized by the accumulation of t lymphocytes and mononuclear phagocytes with the formation of noncaseating epithelioid granulomas in affected organs . the trigger for this t cell and macrophage interaction is still uncertain , although bacterial , viral , and environmental antigens have all been studied . recent literatures renewed the interest in mycobacteria as a causative agent in sarcoidosis tissue [ 2 , 3 ] . here we present a case of presumed ocular sarcoidosis diagnosed by ocular manifestation and treatment course , who developed tuberculous lymphadenopathy 6 years later without recurrence of ocular inflammation . a 35-year - old female was referred to our department with severe visual impairment for months . she was quite healthy until 1 year ago when she began to suffer recurrent episodes of uveitis with mutton fat keratic precipitates ( kps ) in both eyes . on presentation , ocular examination showed mild anterior uveitis with mutton fat kps and dense vitreous hemorrhage in both eyes . systemic workup including general physical examination , complete blood counting , differential classification , blood biochemical tests , rheumatic factor , antinuclear antibody , human immunodeficiency virus , venereal disease research laboratory test , anticardiolipin antibody , and chest radiography were within normal ranges . due to persistent vitreous hemorrhage and disabled vision , pars plana vitrectomy was performed in the left eye and then the right eye 2 months later . sign of peripheral vasculitis with nodular choroid infiltration was noted during operation in both eyes . gallium-67 scans showed panda sign of increased uptake in lacrimal glands and lambda sign of lymph nodes in the mediastinum ( fig . 1 ) . chest computed tomography ( ct ) showed a small ground glass opacity nodule in the subpleural space of the right lower lobe , more in favor of inflammatory process . based on these findings , ocular sarcoidosis was impressed and systemic and topical steroids administered in a tapering dose over 6 months . unfortunately , painless enlargement of lymph node at her right neck was noted 6 years later . lymph node biopsy showed granuloma with caseous necrosis , langhans giant cell , as well as acid - fast bacilli ( fig . 2 ) . three combined antituberculosis medications ( rifampin , isoniazid , and pyrazinamide ) were given for 6 months . the eyes remained silent except cataract progression and glaucoma under two medications during this period ( fig . 1gallium-67 scans showed panda sign of increased uptake in lacrimal glands and lambda sign of lymph nodes in the mediastinumfig . 2pathological finding from lymph node biopsy showed granuloma with caseous necrosis ( hematoxylin and eosin , 40 ) ( a ) , langhans giant cell ( hematoxylin and eosin , 400 ) ( b ) , as well as acid - fast bacilli ( kinyoun 's crystal fuchsin acid - fast stain , 1,000 ) ( c)fig . 3postoperative color fundus picture of both eyes showed sheathing vessels and scattered laser scars at the inferior retina . a right eye . b left eye gallium-67 scans showed panda sign of increased uptake in lacrimal glands and lambda sign of lymph nodes in the mediastinum pathological finding from lymph node biopsy showed granuloma with caseous necrosis ( hematoxylin and eosin , 40 ) ( a ) , langhans giant cell ( hematoxylin and eosin , 400 ) ( b ) , as well as acid - fast bacilli ( kinyoun 's crystal fuchsin acid - fast stain , 1,000 ) ( c ) postoperative color fundus picture of both eyes showed sheathing vessels and scattered laser scars at the inferior retina . a right eye . the gold standard for diagnosis of sarcoidosis is histopathologic examination . considering the fact that ocular sarcoidosis may occur in the absence of apparent systemic involvement , and the fact that biopsy is usually reluctantly accepted by uveitis patients , the international workshop on ocular sarcoidosis ( iwos ) established an international criteria for the diagnosis of ocular sarcoidosis . although there was no pathologic proof of sarcoidosis in our case , her ocular manifestations met at least five of the iwos criteria . though the lacrimal gland uptake of gallium ( panda sign ) is not a specific sign for sarcoidosis , her simultaneous presence of lambda and panda sign on ga-67 scans gave some support to sarcoidosis . as guided , other granulomatous diseases , including tuberculosis , foreign body reaction , bacterial and viral infections , should be excluded before the diagnosis of sarcoidosis . other laboratory and image study ruled out the other possible etiologies . besides , the ocular inflammation subsided after pars plana vitrectomy and systemic corticosteroid . there was no recurrence of inflammation even at the time of tuberculous lymphadenopathy . presumed ocular sarcoidosis was impressed based on the ocular manifestations and treatment course . we could not make any conclusion whether ocular sarcoidosis and tb lymphadenopathy in this patient is just coincident or in real association with each other . despite the increasing understanding of the immune responses behind the formation and maintenance of the granulomatous process in sarcoidosis mycobacteria have been implicated as a cause of sarcoidosis , although the demonstration of mycobacterial dna was inconsistent in different reports [ 6 , 7 ] . recent studies by detecting mycobacterium tuberculosis catalase peroxidase have renewed interest in mycobacteria as a causative agent in sarcoidosis [ 2 , 3 ] . in our case , tb was ruled initially due to negative results of chest x - ray , aqueous tb culture , and pcr . tuberculin skin test was not done as our cdc did not identify the significance of ppd in adult who received routine bacille calmette guerin vaccination . the ocular inflammation revolved after systemic steroid , and remained silent even at the presence of tb lymphadenopathy . our speculation is that the presumed ocular sarcoidosis might be triggered by tb antigen in extraocular infection , which reactivated and presented as tb lymphadenopathy 6 years later . tb could occur coincidently or in association with sarcoidosis , continued follow - up is important for patients with ocular sarcoidosis , even in silent case .

tuberculosis and sarcoidosis share similarity in histopathologic findings and clinically occur in association with each other occasionally . tuberculosis should always be ruled out before the diagnosis of sarcoidosis . but , the diagnosis is often complicated , especially in extrapulmonary cases . here we present a case of bilateral vitreous hemorrhage with uveitis . ocular sarcoidosis was initially diagnosed based on the characteristic ocular findings , negative results on chest radiography , tuberculosis culture , and polymerase chain reaction of aqueous , as well as simultaneous presence of panda and lambda sign on gallium-67 scans . the ocular condition improved after pars plana vitrectomy and systemic steroid therapy . however , tb lymphadenopathy but no recurrent ocular inflammation was found 6 years later . the patient received anti - tb treatment for 6 months thereafter . the eyes remained silent except cataract progression and glaucoma under two medications during this period . in conclusion , tb could occur coincidently or in association with sarcoidosis , continued follow - up is important for patients with ocular sarcoidosis .

cytomegalovirus ( cmv ) colitis is common among immunocompromised patients who are more prone to opportunistic infections . after the infection of cmv is detected by histology or serology , antiviral treatment is usually chosen initially . since cmv disease can not be diagnosed on clinical symptoms alone , diagnosis and treatment are sometimes delayed . here , we report a case of a hemorrhagic cmv colitis that occurred in an immunocompromised patient after a colectomy due to colon cancer and was successfully treated with ganciclovir . an 85-year - old woman underwent esophagogastroduodenoscopy and colonoscopy for investigation of anemia . a laterally spreading tumor of the granular type , 85 mm in diameter , although magnifying image - enhanced colonoscopy suggested a tubulovillous adenoma , surgical resection , not endoscopic mucosal resection , was chosen because of the size of the tumor . however , as she had a medical history including breast cancer , myocardial infarction , general edema due to chronic renal failure ( oral administration of prednisolone 5 mg / day ) , diverticulosis of sigmoid colon , and bilateral ureteral stent placement due to retroperitoneal fibrosis , she did not want to take medical treatment anymore but to monitor her own condition for a while . after 3 months , she finally agreed to have surgery and was admitted to our hospital . the patient underwent ileocecal resection followed by stapled functional end - to - end anastomosis ( fig . the postoperative course was favorable ; dietary intake and oral drug administration were started on postoperative day 4 and both flatus and stool passed . however , she had sudden abdominal pain and heavy bloody discharge on postoperative day 11 . although conservative treatment , including blood transfusion therapy for anemia , was performed initially , there was no improvement after a week . in addition to the endoscopic findings , the patent 's immunocompromised state suggested possible cmv colitis . because cmv antigen was confirmed in peripheral blood samples , intravenous antiviral treatment was started from the postoperative day 23 ( ganciclovir , 150 mg / day ) . after 4 days of the treatment , bloody discharge stopped , and at 8 days , improvement of abdominal pain was observed . the antiviral treatment was continued for 2 weeks until the serological confirmation of negative cmv antigen . after improvement of the colitis - related symptoms , the patient was discharged from our hospital . cmv infection often develops a latent infection with no signs or symptoms after acute infection . although most people are exposed to cmv in their lifetime , typically only the patients with weakened immune systems , such as aids , malignancy , organ transplant or bone marrow transplant , and steroid or immunosuppressive treatments , become ill by reactivating cmv and present with cmv pneumonia , gastroenteritis , retinitis , and encephalitis . cmv disease is often diagnosed by pathologic and serologic confirmation because clinical symptoms are not specific . furthermore , colonoscopic findings of cmv colitis mimic many conditions , including pseudomembranous colitis , ischemic colitis , ulcerative colitis and crohn 's disease . in our case , as we initially considered the bleeding from the diverticulum or anastomosis , conservative treatment was chosen . after one week of conservative therapy , there was no sign of improvement in bloody discharge ; therefore , we decided to perform colonoscopy . we were hesitant to perform colonoscopy due to our patient having had a colectomy . according to the meta - analysis of cmv colitis in immunocompetent hosts , the highest mortality rates were associated with immune modulating conditions that include 16 patients . of those analyzed , 5 patients ( 31.3% ) received colectomy as initial treatment and 8 patients ( 50% ) and 6 patients ( 37.5% ) had coexisting renal failure and diabetes mellitus , respectively . in our case , as the patient was given steroids due to chronic renal failure and the colonoscopy revealed the longitudinal ulcer at the anastomosis , we were able to suspect cmv colitis and confirmed the diagnosis serologically . because endoscopic biopsy was not performed , cmv infection was not evaluated histologically . of note , we performed immunohistochemistry of cmv protein using resected colon cancer specimen , though cmv - positive stain was not detected . this result seems to relate with the fact that cmv is not associated with carcinogenesis and reactivation of latent cmv infection causes cmv colitis [ 5 , 6 ] . the first choice of treatment of cmv infection is antiviral therapy [ 7 , 8 ] . systemic antiviral treatment has provided significant advances and has resulted in dramatically improved outcomes [ 7 , 8 ] . treatment time usually ranges from 1 to 4 weeks , depending on the treatment effect such as improvement of the symptoms and/or confirmation of the negative cmv antigen . in our case , despite the necessity of reducing the dose of ganciclovir due to renal failure , the patient showed good response to systemic therapy and did not require further treatment . fortunately , as she did not show uncontrollable heavy bleeding or symptoms of bowel perforation , surgical resection was not needed . the case we mentioned above can potentially occur in immunocompromised patients . with recent progress in medicine , the number of high - risk patients who undergo surgery is increasing ; therefore , the reactivation of latent cmv infection in immunocompromised patients should be actively considered for the differential diagnosis , leading to timely diagnosis and appropriate treatment .

we report a case of hemorrhagic cytomegalovirus ( cmv ) colitis , occurring in a postoperative patient due to a weakened immune system . an 85-year - old woman with a medical history , including chronic renal failure treated with oral administration of prednisolone , underwent colectomy due to an ascending colon cancer . while the postoperative course was favorable , she exhibited acute severe abdominal pain and massive bloody discharge after 11 days of surgery . her colonoscopic examination showed multiple longitudinal ulcers on the anastomosis . in addition to these endoscopic findings , her past medical history helped suggest cmv colitis . because serological testing revealed positive cmv antigen , she was finally given a diagnosis of cmv colitis and received intravenous ganciclovir for the initial treatment . hemorrhagic cmv colitis after colectomy is an important postoperative complication ; we therefore present our case with diagnosis and treatment experience .

the plant c. chaerophylla was collected from nepal . dried and powdered roots ( 435 g ) were extracted with methanol in a soxhlet extractor . the crude base fraction was chromatographed over sio2 gel column eluting with solvents of increasing polarity . eluants from c6h6-chcl3 ( 15 : 85 ) on crystallisation from methanol furnished colorless granules ( 30 mg ) , rf . . 180~181 , c20 h23 no4 ( m341 ) , [ ]d-300 ( c , 1.5 , etoh ) . mass spectrum exhibited molecular ion peak at m / z 341 and retro - diel 's alder cleavage of ring c gave a base peak at m / z 190 . the hnmr and c nmr spectral data were identical to alkaloid 1-corydalmine ( cava et al . , 1968 ) . it was finally identified as 1-corydalmine by direct comparison with authentic sample ( mixed m.p . , co - tlc and super imposable ir ( fig . stock solution ( 1000 ppm ) of 1-corydalmine was dissolved in 5 mg of compound initially in a few drops of methanol and chloroform ( 1 : 1 ) in a test tube . required concentrations of the chemical ( 100 , 250 , 500 , 750 , 1000 and 1500 l ) were made by the dilution of the solution with distilled water . the chloroform and methanol were evaporated on water bath at 80. a drop ( 30~40 l ) of different concentrations of the chemical was placed separately on grease - free glass slides for studying spore germination . several fungi , e.g. , alternaria brassicae , a. brassicicola , a. solani , curvularia lunata , curvularia maculance , a curvularia sp . , h. penniseti and a heterosporium sp . , were isolated from their respective infected plant parts . small portions of the infected material were incubated in moist petri dishes for 24 h and the fungal growth developed after 24 h on potato dextrose agar ( potato 250 g + dextrose + agar 20 g + distilled water 1,000 ml ) was picked up by an inoculating sterile needle and inoculated on slants which were later purified by single spore isolation technique ( singh et al . , 1990 ) . approximately 200 - 300 spores of each fungus were picked up from fresh sporulating cultures by an inoculating needle aseptically and mixed in a drop of chemical kept on glass slides . the spores of erysiphe pisi causing powdery mildew of pea ( pisum sativum ) were directly removed from the infected leaves and mixed similarly with the chemical . all the slides were kept in petri dishes , which were humidified by fixing moist filter paper on the lower and upper surfaces of the base and lid of the petri dishes . all the petri dishes were incubated at 25 2 for 24 h. after incubation a drop of cotton blue prepared in lactophenol was put on the drop of the chemical containing spores and finally covered with a cover slip . the spore germination was observed under nikon binocular research microscope and finally the percent spore germination was calculated . several fungi , e.g. , alternaria brassicae , a. brassicicola , a. solani , curvularia lunata , curvularia maculance , a curvularia sp . , c. pallscens , fusarium udum , a helminthosporium sp . , h. penniseti and a heterosporium sp . , were isolated from their respective infected plant parts . small portions of the infected material were incubated in moist petri dishes for 24 h and the fungal growth developed after 24 h on potato dextrose agar ( potato 250 g + dextrose + agar 20 g + distilled water 1,000 ml ) was picked up by an inoculating sterile needle and inoculated on slants which were later purified by single spore isolation technique ( singh et al . , 1990 ) . approximately 200 - 300 spores of each fungus were picked up from fresh sporulating cultures by an inoculating needle aseptically and mixed in a drop of chemical kept on glass slides . the spores of erysiphe pisi causing powdery mildew of pea ( pisum sativum ) were directly removed from the infected leaves and mixed similarly with the chemical . all the slides were kept in petri dishes , which were humidified by fixing moist filter paper on the lower and upper surfaces of the base and lid of the petri dishes . all the petri dishes were incubated at 25 2 for 24 h. after incubation a drop of cotton blue prepared in lactophenol was put on the drop of the chemical containing spores and finally covered with a cover slip . the spore germination was observed under nikon binocular research microscope and finally the percent spore germination was calculated . the effect of the chemical was tested at varying concentrations starting from 100 , 250 , 500 , 750 , 1,000 to 1,500 ppm . the maximum effect of the chemical was seen on the spores of h. penniseti as there was no germination at 1,500 ppm and also only 21% at 1,000 ppm as compared to control ( 87.33% ) . similarly , among the four curvularia species , c. lunata was the most sensitive as the spore germination was significantly inhibited even at 250 ppm . significant inhibition of spore germination in a. brassicae was observed at 500 ppm and above and other two species of alternaria showed sensitivity at 750 ppm and above concentrations . while 250 ppm and above doses were significantly inhibitory for the germination of spores of heterosporium species , similar significant effect on erysiphe pisi and f. udum was seen at 1,000 and 1,500 ppm concentrations ( table . a number of chemical compounds isolated from plants are antifungal ( singh et al . , 1990 ; srivastava et al . , 1994 ; sarma et al . , 1999 ; singh et al . , 1999 ; basha et al . , 2002 ; maurya et al . , 2001 , 2002 ; the fungi included in the present study belong to two different groups . but based on the activity of the chemical , it is at present difficult to conclude as to which group of fungi will be most susceptible to this chemical as the number of fungi from different groups is very low . hence , only further detailed study on several members of other groups will decide its limit and specificity of efficacy . nevertheless , as antifungal activity of the present compound has not been reported so far and the efficacy is significantly high at low concentrations , there is enough possibility of using this compound in plant disease control under field conditions .

1-corydalmine , an alkaloid isolated from roots of corydalis chaerophylla inhibited spore germination of some plant pathogenic as well as saprophytic fungi e.g. alternaria brassicae , a. brassicicola , a. solani , curvularia lunata , c. maculans , c. sp . , c. pallscens , erysiphe pisi , fusarium udum , helminthosporium species , h . penniseti and a heterosporium species . 1-corydalmine significantly inhibited spore germination of all the fungi at 100 to 1500 ppm . it was effective against all the fungi at 1500 ppm . c. lunata was highly sensitive to this chemical even at 250 ppm .

dopa - responsive dystonia ( drd ) is characterized by childhood - onset dystonia and a dramatic and sustained response to administration of low - doses of oral levodopa . this disorder typically presents with gait disturbance , caused by lower limb dystonia with diurnal variation , a positive family history , and gradual progression to generalized dystonia . a ten - year - old girl was brought with complaints of difficulty in walking and stiffness of both lower limbs , for the past six months . she also had a history of frequent falls while walking and was unable to stand for prolonged periods . her antenatal , birth , and neonatal periods were uneventful and developmental milestones were appropriate for her age . her higher mental functions were normal , speech was hypophonic ; she had a gait disturbance characterized by leg stiffness , and a tendency to walk in an equinus posture , resulting in difficulty in balancing . her tone was slightly increased in all the four limbs , with cog - wheel rigidity . deep tendon reflexes in all the four limbs were exaggerated with extension of both great toes ( striatal toes ) . , it was found that the symptoms and signs were relatively mild in the morning during rounds , whereas , they gradually worsened as the day progressed , rendering the child almost unable to walk by evening . magnetic resonance imaging ( mri brain and spine were normal . in view of the typical diurnal variation of dystonia , a therapeutic challenge with levodopa / carbidopa was tried and there was a dramatic decrease in dystonia within two days and the child 's gait improved . the child was treated with a combination of levodopa , carbidopa , and trihexyphenidyl . on follow - up genetic studies were planned , but could not be done due to lack of resources . drd is an inherited disorder characterized by dystonia with diurnal variation and favorable response to levodopa / carbidopa . the inheritance is usually autosomal dominant ; however , autosomal recessive inheritance is also seen in some cases . the enzyme deficiency responsible for the manifestations is gtp cyclohydrolase 1 ( gch ) , which is a rate - limiting enzyme in the synthesis of dopamine . this disorder had been referred to as hereditary progressive basal ganglia disease , hereditary progressive dystonia with marked diurnal variation , segawa disease , and drd in the past . at present , segawa disease specifically denotes an autosomal dominantly inherited mutation in the gch 1 gene located on chromosome 14q22.1 to q22.2 . the disease usually manifests in school age children , however , adults with the disease have also been reported . initial manifestations of this disease include postural dystonia of the lower limbs with characteristic equino varus deformity of both feet . the dystonia gets worse as the day progresses , becomes maximal by evening , and decreases after sleep by morning . investigations characteristically reveal low levels of pteridine metabolites in the cerebrospinal fluid , normal neuroimaging , and increased blood phenylalanine levels after phenylalanine loading tests . assessing the therapeutic response to levodopa is a useful and recommended method of diagnosing drd , when the diagnosis is in doubt and when dystonia is not attributable to hypoxic ischemic encephalopathy . in one reported series , administration of low - dose levodopa had resulted in complete to near - complete recovery of symptoms in a cohort of chinese patients , with no significant long - term side effects . in our case , the child presented with gait disorder characterized by dystonic movements of the lower limbs of gradual onset , which disappeared during sleep , and reappeared after getting up from bed and progressively worsened throughout the day with symptoms , for the past six months . with the above - mentioned clinical findings and a dramatic response to the levodopa / carbidopa combination children with recent onset dystonia and gait abnormalities may pose a diagnostic challenge . a careful history and focused neurological examination , looking for diurnal variation in symptoms , holds the key in arriving at the diagnosis . in such children a therapeutic response to levodopa might be a safe and appropriate way of confirming drd .

children with recent onset dystonia and gait abnormalities may pose a diagnostic challenge . a ten - year - old , developmentally normal girl , presented with a six - month history of gait abnormality and dystonia . her complaint worsened as the day progressed . in view of typical diurnal variation of dystonia , a therapeutic challenge with levodopa / carbidopa was given and there was a dramatic response . hence , a diagnosis of dopa - responsive dystonia ( drd ) was made . drd is an inherited disorder characterized by dystonia with diurnal variation and favorable response to levodopa / carbidopa . the inheritance is usually autosomal dominant , however , in some cases , autosomal - recessive inheritance is also seen .

malignant thymic neoplasms can be broadly divided into thymomas and thymic carcinomas , both of which exist almost exclusively in the anterior mediastinum . thymomas have also been described in the neck , posterior mediastinum , lungs , base of the skull and pleural cavity [ 25 ] . very few case reports have described thymomas occurring in the middle mediastinum [ 25 ] . unlike thymomas , thymic carcinomas have only been observed in the anterior mediastinum . to our knowledge , this report will describe the first case of thymic carcinoma in the middle mediastinum . a 55-year - old caucasian female presented with productive cough , dyspnea , chest pain and weight loss . the patient 's medical history was significant for hypertension , hypercholesterolemia , dyslipidemia , chronic renal failure , gastroesophageal reflux disease and chronic anemia . she was a 30 pack - year smoker and her mother was diagnosed with lung cancer . an initial chest x - ray showed a large subcarinal mass . on computed tomography ( ct ) , a 6.2 5 cm tumor was detected in the subcarinal area ( figs 1 and 2 ) . a bronchoscopy with transbronchial biopsy was performed , and the resulting pathological analysis was suspicious for malignant cells ; however , no specific tumor type was identified . a positron emission tomography scan showed a large hypermetabolic subcarinal lesion with a standard uptake value of 14.3 , consistent with malignancy . the differential diagnosis at the time included an infected bronchogenic cyst , esophageal duplication cyst or a malignant subcarinal lymph node . thoracoscopic surgical resection was planned for diagnostic and therapeutic purposes . figure 1:subcarinal tumor on preoperative ct of the chest , coronal cuts . prior to surgical resection , esophagoscopy and bronchoscopy documented the absence of any communication of this mass to the esophagus and airway . one port was made at the mid - axillary line of the eighth intercostal space for the camera , in addition to two anterior and two posterior ports . the dissection of the tumor began at the level of the inferior pulmonary ligament and posterior mediastinum , all the way to the azygous vein . the esophagus was found completely adherent to the tumor , necessitating an esophageal myotomy for en bloc resection . the anterior dissection proceeded at the level of the posterior atrial wall , the inferior and superior pulmonary veins , the main trunk of the pulmonary artery , and the membranous areas of the right and left main stem bronchi . complete gross resection was observed at the conclusion of the operation . at the end of the procedure , air was insufflated into the esophagus to confirm the absence of any leaking from the mucosa . the specimen consisted of an encapsulated red - brown tissue weighing 57 g , and measuring 5.5 4.5 5 cm . some sheets of viable tumor cells were present , and these contained pleomorphic nuclei , vesicular chromatin , and prominent nucleoli . the tumor cells were strongly positive for cam 5.2 , but negative for ck7 , ck5/6 , ck19 , calretinin , ttf1 , cea , afp , hcg , plap , cd5 and cd56 . thus , thymic carcinoma was determined to be the most fitting diagnosis based on the morphology and staining . the patient was referred for adjuvant radiation . at the most recent follow - up visit 9 months after surgery , the patient was well with no evidence of recurrence on ct scan . all thymic carcinomas reported to date have been located in the anterior compartment of the mediastinum . to the best of our knowledge , this is the first case report of a thymic carcinoma occurring in the middle mediastinum . the differential diagnosis of middle mediastinal tumors usually includes bronchogenic cysts , enterogenous cysts , neuroenteric cysts , pericardial cysts and lymphangiomas . very selected cases of thymoma have been reported in the middle mediastinum [ 25 ] . these are attributed to the presence of ectopic thymic tissue in the subcarinal area due to failure of the thymus to migrate into the anterosuperior mediastinum during embryological development [ 25 ] . by way of the same process , it is therefore theoretically possible for these cells to undergo malignant degeneration into thymic carcinoma , although this has not been previously described . when compared with thymomas , thymic carcinomas have considerably worse prognosis . when possible , complete surgical resection is the treatment of choice for thymic carcinomas [ 1 , 6 ] . therefore , when planning surgical resection , it is important to consider malignant neoplasms as part of the differential diagnosis of middle mediastinal tumors . although rare , thymic carcinomas and other malignant tumors of the middle mediastinum should be resected with negative margins , in order to decrease the risk of local recurrence and improve prognosis . this is the first case report of a malignant thymic carcinoma occurring in the middle mediastinum at the subcarinal location . this pathology , although rare , should be added to the differential diagnosis of middle mediastinal tumors . when planning surgical resection of such tumors , complete excision with negative margins should be attempted to minimize chances of local recurrence and improve prognosis .

we report an extremely rare case of a thymic carcinoma occurring in the subcarinal location of the middle mediastinum , masquerading as subcarinal adenopathy . a 6.2 5 cm mass was detected on computed tomography scan and found to be hypermetabolic on positron emission tomography scan . transbronchial biopsy was performed and the mass was found to be suspicious for malignancy . a thoracoscopic resection of the mass was performed and found to be thymic carcinoma . the patient underwent a course of adjuvant radiation . to the best of our knowledge , this is the first reported case of a thymic carcinoma resected from the middle mediastinum .

amyloidoses can be classified into three categories : primary localized cutaneous amyloidosis ( plca ) , secondary localized cutaneous amyloidosis , and systemic amyloidosis with cutaneous involvement . in the plca group , three types can be distinguished : macular , papular ( lichenoid ) and nodular forms . the first two are mainly located on the trunk , and cytokeratins serve as the amyloid precursors . here , amyloid deposition is limited to the papillary dermis [ 1 , 2 , 3 , 4 , 5 ] . however , in the rare cases of nodular plca , amyloid consists of aggregated kappa and lambda light chains , which can be found both in the dermis and the subcutaneous tissue [ 1 , 2 , 3 , 4 , 6 ] . furthermore , nodular plca can originate from systemic amyloidosis or progress to systemic disease [ 3 , 4 ] . here , we present a case of nodular plca on the temple of a 52-year - old woman . a 52-year - old lady presented with a soft , shiny , partially yellow , erythematous tumor ( 3.5 4.5 cm ) with telangiectasia on her right temple ( fig . she recalled having had this tumor for about 4 years and that it had significantly grown lately . the diagnosis was confirmed by electron microscopy , and the typical amyloid fibrils ( 710 nm in diameter ) were found ( fig . a punch biopsy taken during follow - up showed some remaining amyloid so that the patient is now scheduled to undergo a second surgery via curettage . the diagnosis of plca requires histological analysis of a skin specimen , complemented by immunohistochemistry and electron microscopy . histologically , eosinophilia as well as positivity in periodic acid - schiff , congo red , and thioflavin t stainings are characteristics of amyloid deposits [ 1 , 8 ] . immunohistochemistry with antibodies directed against cytokeratin and immunoglobulin light chains ( lambda and kappa ) allows to further distinguish between the different forms of plca . electron microscopy confirms the diagnosis of amyloidosis when the typical amyloid fibrils ( 710 nm in diameter ) are found . image quality improves significantly when the skin sample is fixed in karnofsky 's fixative ( glutaraldehyde ) instead of paraformaldehyde . for patients with nodular plca , it is recommended to asses for progression to systemic amyloidosis on a regular basis . this should include a full history and physical examination along with electrocardiogram , complete blood count , serum creatinine levels , serum liver - associated enzyme levels , serum electrophoresis , and urine examination . furthermore , an abdominal fat biopsy has been suggested as an easy method to detect a potential progression to systemic disease [ 3 , 7 ] . it has to be mentioned though that nodular plca is more frequent in asia and south america when compared to europe or north america .

a 52-year - old woman presented with a large partially yellow and erythematous tumor on her right temple . she reported that it had grown over the last 4 years . regional lymph nodes were impalpable . a punch biopsy showed eosinophilic material in the dermis and subcutis . immunohistochemistry showed positive staining for kappa and lambda light chains . electron microscopy showed the typical amyloid fibrils ( 710 nm in diameter ) . there was no evidence of systemic amyloidosis , paraproteinemia or underlying plasmacytoma . the tumor was completely removed via curettage . at follow - up , the patient presented in good health with no signs of relapse .

palatal rugae are irregular , asymmetric ridges of mucous membrane extending laterally from the incisive papilla and the anterior part of the median palatal raphe . these structures have been used as internal cast reference points for quantification of tooth migration . a few investigators have shown the medial rugae region to be stable or show predictable changes post orthodontic therapy . the rugae patterns are completely formed by the 12 to 14 week of prenatal life and remain stable thereafter . these are unique to each person and show distinctiveness based on ethnic groups and hence are useful in forensic identification . because they are stable landmarks the palatine rugae play a significant role in clinical dentistry as well . the objective of this study was to analyze and characterize the rugae patterns , compare the rugae dimensions in various age groups and to ascertain any relationship between dimensional analysis and palatal depth . cross - sectional casts of 52 females and 48 males were selected from the archival section of the department of orthodontics , m. m. college of dental sciences and research , mullana ( ambala ) and studied employing following parameters : assessment of age ( according to erupted teeth).gender differentiation ( based upon records).division of medial palatal region into : a : distance between incisive papilla length and anterior limit of the anteriormost rugae.b : distance between incisive papilla and posteriormost rugae limits.lateral rugae dimensions . palatal depth ( measured from healthy gingival margin underneath the mesiolingual cusp to the deepest concavity of the palatal arch using a brass wire between two opposing points and measuring the vertical distance at the center).rugae patterns . division of medial palatal region into : a : distance between incisive papilla length and anterior limit of the anteriormost rugae.b : distance between incisive papilla and posteriormost rugae limits.lateral rugae dimensions . a : distance between incisive papilla length and anterior limit of the anteriormost rugae . palatal depth ( measured from healthy gingival margin underneath the mesiolingual cusp to the deepest concavity of the palatal arch using a brass wire between two opposing points and measuring the vertical distance at the center ) . no patient details were disclosed and ethical guidelines as per the declaration of helsinki were followed . correlations among a , b , lateral rugae patterns and palatal depth were calculated by mean s.d . correlations among a , b , lateral rugae patterns and palatal depth were calculated by mean s.d . followed by evaluation of p values . the rugae patterns identified were : common origin ; separate origin ; lateral branching ; secondary rugae and fragmentary patterns . females were found to have slightly higher predilection towards having the common , fragmentary and lateral branching rugae patterns . there was a stronger female predisposition for rugae with separate origin , whereas , the secondary rugae were equally existent in either gender . significant correlation was found between the a ( p=0.03 ) and b values ( p=0.02 ) on comparing the mean s.d . values of age groups between 12 - 13 years and > 14 years , respectively . hence , there is a corresponding anteroposterior increase in palatal dimensions in these age groups [ table 1 ] . this study showed no changes in the lateral rugae dimension and palatal depth with increasing age . the majority of the study cohort ( 57% ) had a palatal depth in the range of 1.6 - 2.0 cm [ table 2 ] . palatal rugae have been used as reference points for many purposes such as evaluating tooth movement pre- and post - orthodontic treatment , population studies and forensic identification . stability of medial palatal region has been a subject due to differences among various investigators . christou and kiliardis evaluated the vertical changes in the medial aspects of the rugae and concluded that these changes over time are due to the alterations in the vertical positioning of maxillary incisors and increase in lower face height . growth periods ( 12 - 13 years and > 14 years ) studied in this paper , showed a downward and forward movement of the maxilla in relation to the cranial base and also , changes in the size and shape of maxilla by structural remodeling . these phenomena can be explained by the deposition of new bone on the oral surface of the palate and at the alveolar crest . therefore , the changes in rugae dimensions can be the result of this differential growth in the palate and alveolar crest.[24 ] rugae patterns showed a strong female predilection for rugae with separate origins [ figure 1 ] whereas slightly higher incidences were noted for rugae with common origin , fragmentary and lateral branching patterns . there was an equal percentage of gender with secondary rugae pattern in the ethnic segment of north indian population studied [ table 1 ] . photograph depicting rugae patterns with secondary origin and fragmentary nature the purpose of this paper was to evaluate a cross - sectional patient database to analyze the anteroposterior stability of the medial rugae region . analysis of 100 study casts showed a significant difference in the medial rugae region ( p= 0.03 , 0.02 ) in a and b values . no significant changes were noted in the lateral rugae dimensions . the palate growth in this period was found to be non - significant . hence , it can be surmised from the analysis of the results that there is a differential growth spurt in the anterior and posterior palate during adolescence as is marked by the significant differences in the measurement values obtained through this study . palatal rugae can be studied as a strong indicator of ethnicity , gender differentiation and study of growth changes in the anterior maxilla . thus , rugae are important tools in clinical investigations involving forensic anthropology and developmental biology .

introduction : rugae patterns are significant markers for analyzing anteroposterior changes in adolescence , and forensic investigations.aim:the purpose of this study was to ascertain the gender - wise predisposition of rugae patterns and to analyze anteroposterior alterations along with any developmental changes in palatal depth.materials and methods : one hundred pre - treatment study cast models were obtained from the archives of the department of orthodontics , m.m . cdsr , mullana . parameters employed were : division of medial palatal region into a : distance between incisive papilla length and anterior limit of the anteriormost rugae ; b : distance between incisive papilla and most posterior rugae limits ; measurement of lateral rugae dimensions and palatal depth . statistical analysis : mean s.d . values were obtained and p values calculated.results:comparison of a and b showed a significant difference in the p values between the age - groups 12 - 13 years and > 14 years . rugae patterns with separate origins showed a predisposition for female gender.conclusion:a significant change in the anteroposterior medial rugae dimensions was seen in adolescent age groups alongside no variation in palate depth . hence , it can be concluded that differential growth potential is present in the premaxilla and can cause shift in medial palatal dimensions without altering the rugae patterns and palatal depth .

median arcuate ligament syndrome ( mals , also called celiac axis compression syndrome or dunbar syndrome ) is known to be caused by compression of celiac artery ( ca ) by a fibrous arch that originates from the diaphragmatic crura on either side of the aortic hiatus and passes superior to the origin of the celiac axis . though it is still debated whether the compression of the ca can cause chronic mesenteric ischemia or not , there have been anecdotal case reports of surgical decompression of ca by dividing the medial arcuate ligament ( mal ) . the common clinical features of mals are chronic postprandial abdominal pain characteristically augmented by full expiration , nausea or vomiting , weight loss and audible epigastric bruit . the diagnosis of mals usually depends on the clinical features and radiologic finding of the focal narrowing at the proximal celiac axis on a lateral view of conventional aortography or computed tomography ( ct ) . while open surgical release of mal has been used for the treatment of this syndrome , laparoscopic release has been reported recently . however , it was usually ineffective owing to the refractory extrinsic compression of the ca by the tight ligament structure . we would like to report a case of surgical treatment of mals describing its clinical features , characteristic findings of diagnostic imaging study , and details of our surgical procedure . a 37-year - old female patient presented with chronic epigastric pain lasting 6 months , food phobia and weight loss of 10 kg during the previous 4 months . the pain was cramping aggravated after meals , and persisted for 20 - 30 minutes after meals . there were no other gastrointestinal symptoms such as diarrhea , constipation , gastrointestinal bleeding and nausea or vomiting . as past history , she underwent coil embolization of the bilateral ovarian veins at another hospital 2 months before visiting us under the impression of pelvic congestion syndrome , which did not improve her abdominal symptoms . physical examination showed mild abdominal tenderness at both lower quadrants without muscle rigidity . laboratory test showed normal range including serum amylase , liver enzyme and complete blood cell count . gastro - duodenoscopy showed chronic atrophic gastritis and 18-fluorodeoxyglucose positron emission tomography - ct showed no spe cific lesion with abnormal hot uptake . 1 ) showed downward angulation of ca and superior mesenteric artery ( sma ) close to their origins by the compression . to decompress ca , we approached through the upper mid line incision and lesser sac . postoperatively , epigastric postprandial pain was resolved and the patient could return to a normal diet . on a follow - up mal was first described by lipshutz as an anatomic structure that caused ca compression in 1917 . thereafter , harjola and dunbar et al . described it as a clinical syndrome causing nausea , vomiting and postprandial pain in 1963 and 1965 . the origin of ca and mal varies in its location from t11 to l1 , and occasionally , their locations are in conflict with each other when mal extends inferiorly or ca originates su periorly . during deep inspiration in the erect position during expiration , the condition is opposite , and compression causes the symptom and is thus called mals . at this point , it is proposed that mals is related with neurogenic pain from the compression and intermittent ischemia of splanchnic nerve plexus . this pain can be caused either by nerve stimulation leading to vasoconstriction or by direct sympathetic fiber irritation . splanchnic nerve plexus is an autonomic nerve plexus supplying upper abdominal organs ( stomach , liver , gallbladder , pancreas ) . this is located in front of diaphragmatic crura around the origin of ca and sma . to make a diagnosis of mals , other common causes of abdominal pain routine laboratory blood tests including amylase , lipase and tumor markers for hidden malignancy , esophago - gastro - duodenoscopy , liver , pancreas and kidney ultrasonography are usually used . a typical feature is focal narrowing of ca with poststenotic dilatation aggravated during deep inspiration . nowadays standard treatment of mals is an open surgical division of mal followed by a dramatic symptom relief . recently , a lapa roscopic approach can be attempted but carries the risk of arterial injury and massive hemorrhage . three series of case reports showed mean rates of open conversion due to bleeding at about 20% , but despite that , laparoscopic treatment showed no other morbidity or mortality and shortened hospital stay . this is probably due to the extraluminal compression by mal , which should be solved outside of the ca . in cases of recurrent symptom after surgical decompression of ca , angioplasty is beneficial .

median arcuate ligament syndrome is a rare cause of abdominal pain which results from compression of the celiac artery ( ca ) or rarely , the superior mesenteric artery by a ligament formed by the right and left crura of the diaphragm . we report a case of open surgical decompression of the ca by division of the median arcuate ligament for a 37-year - old female patient who had suffered from chronic postprandial epigastric pain and severe weight loss . we described clinical features , characteristic angiographic findings and details of the surgical procedure for the patient with this rare vascular problem .

a 38-year - old woman was admitted to our outpatient clinic with accelerating back pain and fatigue following a kick to her back by her husband two days previously . on her physical examination , an ecchymotic area on her back between the scapulae was observed . she had pallor , her blood pressure was 80/60 mmhg in both arms , and was tachycardic on auscultation . st segment elevations were observed in the d1 , avl , and v2 leads , along with accelerated idioventricular rhythm ( fig . 1 ) . transthoracic echocardiography demonstrated akinesia of the anterior septal , apical , basal - mid septal , and basal - mid anterior walls , and her ejection fraction was 20% . we detected a dissection of the left main artery , the left anterior descending artery ( lad ) , and the circumflex artery , originating from the middle portion of the left main coronary artery ( lmca ) ( figs . 2 and 3 , supplemental videos 1 , 2 ) . a saphenous vein was grafted to the distal lad . during the intraoperative evaluation of the epicardial vessels , our team of cardiac surgeons did not plan to place a bypass graft to the circumflex artery because it was thin and non - dominant . since the patient was hypotensive under noradrenaline and dopamine infusions , she was transferred to the cardiovascular surgery intensive care unit on an extracorporeal membrane oxygenator ( ecmo ) and intra - aortic balloon pump ( iabp ) . during follow - up , her blood pressure remained low , at approximately 60/40 mmhg , despite aggressive inotropic and mechanical support . on the second postoperative day , although the patient s lad artery had been revascularized by a saphenous vein graft , her left ventricular ejection fraction remained as low as 10%15% , leading to ventricular failure . asystole and cardiovascular arrest then quickly developed , and despite aggressive cardiopulmonary resuscitation , she died . coronary artery dissection after blunt chest trauma is an extremely rare condition that can be fatal , and some cases are detected in postmortem examinations . left main coronary artery dissection is even rarer . multiple mechanisms exist leading to coronary artery dissection , including intimal tears due to a deceleration injury , compression of the artery between the heart and sternum , coronary spasm , and impairment of the coronary flow by a dissection flap or a superimposed thrombosis . coronary artery dissection is detected most commonly in the lad ( 76% ) , the right coronary artery ( 12% ) , and the circumflex artery ( 6% ) [ 4 , 5 ] . in necropsy series , the most common cause of acquired non - atherosclerotic coronary artery disease is spontaneous coronary artery dissection , and the lad is the artery in which this condition is most commonly detected . the risk factors for spontaneous coronary artery dissection are exercise , arteriosclerosis , cardiovascular disease , oral contraceptive use , marfan syndrome , systemic lupus erythematosis , and connective tissue disorders . however , the time from injury to coronary artery occlusion may vary , ranging from immediately after the trauma to five weeks later . bedside electrocardiography ( ecg ) provides important clues about coronary artery dissection after blunt anterior chest and back trauma . it has been previously reported that patients with baseline ecg changes on admission should be monitored for 24 hours . the ecg may be normal on presentation , but was found to demonstrate st abnormalities in 63% of patients who are admitted for blunt thoracic trauma within 24 hours of observation . our patient presented with st segment elevation the in d1 , avl , and v2 leads , along with accelerated idioventricular rhythm . some emergency therapeutic options exist for patients with spontaneous coronary artery dissection . in some previous case reports , the patients were managed by primary percutaneous coronary angioplasty , especially patients without lmca lesions . however , surgical treatment remains most common treatment and is associated with the best outcomes . an internal mammary artery graft was not utilized in our patient , because she was in cardiogenic shock , and the surgical team tried to save time by using a saphenous vein graft . in order to prevent death , all available measures were used by our surgical team , including intravenous inotropic agents , iabp , and ecmo . nevertheless , using the internal mammary artery for revascularization may have been more effective for restoring left ventricular function . additionally , complete revascularization , including the circumflex artery , spontaneous coronary artery dissection is a rare and potentially fatal complication of blunt chest trauma in younger patients , and early diagnosis and prompt treatment can be life - saving . physicians should be aware of this possibility when evaluating patients in emergency conditions after blunt trauma of any kind . the 12-lead ecg , cardiac bio - markers , and transthoracic echocardiography , along with other imaging modalities , are important in the management of suspected coronary artery dissection in trauma patients .

we present the case of a 38-year - old woman admitted to our outpatient clinic with accelerating back pain and fatigue following a kick to her back by her husband . upon arrival , we detected st segment elevation in the d1 , avl , and v2 leads and accelerated idioventricular rhythm . she had pallor and hypotension consistent with cardiogenic shock . we immediately performed coronary angiography and found a long dissection starting from the mid - left main coronary artery and progressing into the mid - left anterior descending ( lad ) and circumflex arteries . she was then transferred to the operating room for surgery . a saphenous vein was grafted to the distal lad . since the patient was hypotensive under noradrenaline and dopamine infusions , she was transferred to the cardiovascular surgery intensive care unit on an extracorporeal membrane oxygenator and intra - aortic balloon pump . during follow - up , her blood pressure remained low , at approximately 60/40 mmhg , despite aggressive inotropic and mechanical support . on the second postoperative day , asystole and cardiovascular arrest quickly developed , and despite aggressive cardiopulmonary resuscitation , she died .

a fit and healthy 25-year - old male presented with palpitations from a regular broad complex tachycardia ( bct ) of left bundle branch block ( lbbb)-like morphology , 330-ms cycle length , and no discernible p waves . intravenous adenosine terminated the bct , resting electrocardiogram ( ecg ) was not pre - excited , and echocardiogram was normal . during the electrophysiologic study , retrograde conduction was central and decremental . 1a shows that conduction switched from nodal ( narrow qrs complexes with distinct his potential solid blue arrow ) to the atriofascicular pathway ( lbbb - like complexes and interpolation of the his potential dotted blue arrow ) during wenckebach - pacing maneuver . 1b shows that the intellatip mifi ablation catheter was used to map the atriofascicular pathway potential in sinus rhythm . the 8-mm tip ablation catheter has three radially positioned , equally spaced mini - electrodes , 2 mm from the tip . bipolar recordings were made between these mini - electrodes : m12 , m23 , and m31 . this enabled precise signal localization and clearer identification of the pathway potential than the conventional ablation distal bipolar recording ( blue dotted box , fig . the gains on the catheter mini - electrodes were 5000-fold greater , and those in the conventional distal pole of the ablation catheter were 10,000-fold greater . radiofrequency ablation ( rfa ) at this site ( 60 c , 70 w , and 120 s ) resulted in no pathway conduction and non - inducible bct . atriofascicular pathways consist of fibers arising from the right atrial free wall and insert at or adjacent to the distal right bundle . they usually only conduct in an anterograde manner , participating in the anterograde limb of an antidromic atrioventricular reciprocating tachycardia ( avrt ) with lbbb - like morphology and decremental properties . standard therapy involves targeted rfa around the tricuspid annulus as guided by pathway potentials also known as mahaim ( m ) potentials , . these m potentials can be as large as the his deflection or can be small and narrow with a low amplitude . furthermore , unintentional mechanical trauma by catheter manipulation can result in transient abolition of these potentials from a few minutes to a few hours , . these m potentials are also recorded only in close proximity to the atrial insertion site , and thus , accurate localization and ablation at this site result in successful abolition of this pathway , , . the intellatip mifi catheter has been shown to delineate local electograms better in the isthmus than the conventional bipolar electrode during atrial flutter ablation . the signal amplitude in the mini - electrodes has been noted to be higher than that of the conventional bipolar catheter . we used the same principle in this case to make use of the special characteristics of the novel ablation catheter to localize the atrial insertion point accurately . furthermore , the m potentials , as recorded from the distal mini - electrodes , were clearer and of higher amplitude compared with those recorded using the conventional distal pole of the ablation catheter . we did not compare this with a standard ablation catheter in the same patient to keep the costs of the procedure within reasonable limits and to avoid causing unintentional mechanical trauma to the atrial insertion site .

atriofascicular pathways are a rare cause of antidromic atrioventricular reciprocating tachycardia . the intellatip mifi ablation catheter ( boston scientific , ma , usa ) is a novel ablation catheter that allows enhanced signal clarity with highly localized electrograms . this is the first report of this catheter being successfully used to map and ablate the atriofascicular pathway potentials .

in late february 2006 , a 27-year - old pregnant woman , at 6 weeks gestation , was referred to grenoble university hospital , grenoble , france , for investigation of persistent left cervical lymphadenopathy with fever . the lymphadenopathy occurred 3 weeks earlier , along with a sore throat , and persisted despite 10 days treatment with amoxicillin ( 3 g daily ) . at admission , the patient was febrile ( 38c ) and had a tender , swollen , submaxillary cervical lymph node on the left side . magnetic resonance imaging of the left cervical region showed a large mass extending from the parotid region to the submandibular region , with hypo- and hypersignals in t1- and t2-weighted imaging , respectively . lymph node tissue was obtained by needle aspiration , and examination revealed nonspecific lesions of lymphadenitis . laboratory test results showed moderate inflammatory syndrome ( c - reactive protein 67 mg / l ) . serologic results were negative for hiv , hepatitis b and c viruses , rubella virus , treponema pallidum ( syphilis ) , coxiella burnetii ( q fever ) , and borrelia , bartonella , brucella , and legionella spp . and showed only residual igg - type antibodies against cytomegalovirus , epstein - barr virus , herpes simplex virus , parvovirus , mycoplasma pneumoniae , and chlamydophila pneumoniae . because we suspected severe streptococcus pyogenes infection , a second course of amoxicillin was administered for 13 additional days . after initial clinical improvement , the patient relapsed , and the lymphadenopathy evolved to suppuration and necrosis . in march 2006 , a large amount of pus was surgically drained from the site , and inflamed lymph nodes were removed . the patient denied receiving any tick bite but reported that she regularly fed domestic rabbits in cages . her symptoms began a few days after she killed and skinned rabbits , which were then kept frozen but not eaten by the family . routine cultures and mycobacterial cultures remained sterile , but f. tularensis dna was detected in lymph node samples by using specific real - time pcr targeting the gene encoding a 23-kda surface protein ( 5 ) . pcr amplification and sequencing of the 16s rdna23s rdna intergenic spacer region ( 5 ) directly from lymph node tissue confirmed the presence of dna from f. tularensis subsp . serum samples collected in late february and on march 15 , 2006 , were tested by using an immunofluorescence assay and a homemade f. tularensis antigen ( 5 ) . both samples were positive , with igm and igg titers of 320 ( cutoff titers of > 160 ) . because the patient was pregnant and tularemia in france holarctica ( 2 ) , which are naturally susceptible to macrolides , she was treated with azithromycin ( 500 mg / d for 6 weeks ) . the patient reported that the rabbits were reared outdoors , in floor - level , wire - mesh cages ; therefore , we suspected the animals were infected with f. tularensis through contact with wild fauna . attempts to detect f. tularensis in pieces of the frozen rabbits by culture and pcr tests were unsuccessful . clinical symptoms of tularemia are primarily related to the portal of entry of bacteria , the f. tularensis strain virulence , and the immune status of the patient . the incubation period is usually 13 days but may last up to 15 days ( 3,6 ) . the primary clinical forms are glandular and ulceroglandular ( skin inoculation ) , oculoglandular ( conjunctival inoculation ) , oropharyngeal ( oral contamination ) , pneumonic ( inhalation of an infected aerosol ) , and typhoidal ( various modes of infection ) ( 2,7 ) . tularemia may be severe and even fatal ; patients with lymphadenopathy may experience lymph node suppuration in 30% of cases ( 1 ) . our patient s symptoms were fever , pharyngitis , and cervical lymphadenopathy ; treatment with a -lactam drug did not improve her condition , which rapidly evolved to local suppuration , a sign that should prompt physicians to consider oropharyngeal tularemia in disease - endemic regions . because tularemia cases remain extremely rare in the french alps , where the patient lived , this diagnosis was not considered at the time of the first medical consultation . because the rabbit meat was not consumed by the patient , we suspected she became infected at the time she skinned these animals however , the animals had no overt disease at the time they were killed , so low bacterial inoculum may explain why the tests performed on rabbit meat had negative results . diagnosis of tularemia is often made by serologic tests ( 3 ) , although these are negative during the first 2 weeks following the onset of symptoms ( 1 ) . in the case described here , the same high antibody titers were obtained in 2 serum samples taken 2 weeks apart , which indicates that the peak secretion of specific antibodies was achieved . culture of f. tularensis from clinical samples remains poorly sensitive , but pcr - based testing of pharyngeal swab specimens or lymph node suppurations or biopsy specimens enables rapid diagnosis of oropharyngeal tularemia and identification of the f. tularensis subspecies involved ( 1,4,5,7,8 ) . a few tularemia cases occurring in pregnant women have been reported ( 9 ) . severe illness or death caused by infection with f. tularensis could be a risk for a pregnant woman or her fetus ; the role of f. tularensis as an agent of abortion and intrauterine death is well recognized in sheep ( 10 ) but not in pregnant women . a major difficulty in this instance was the choice of the antimicrobial drug regimen , because first - line antibiotics currently recommended for treatment of tularemia , including the aminoglycoside gentamicin , fluoroquinolones , and tetracyclines ( 1,2,7,8 ) , may be toxic for pregnant women or fetuses . no treatment recommendation for tularemia during pregnancy is available ( 9 ) . the pregnancy outcome was favorable , and the patient and the infant were healthy at 12-month follow up . macrolides are usually not recommended for treatment of tularemia patients ( 2,7,8 ) , especially because f. tularensis subsp . holarctica biovar 2 strains , mainly found in eastern europe and asia , are naturally resistant to macrolides ( 1114 ) . the ketolides ( e.g. , telithromycin ) are highly active against f. tularensis in vitro ( 11,15 ) , but their use in pregnant women is currently discouraged . this case emphasizes the usefulness of azithromycin as a first - line treatment for tularemia in pregnant women in areas where infections caused by biovar 2 strains of f. tularensis subsp .

a pregnant woman who had oropharyngeal tularemia underwent treatment with azithromycin and lymph node resection and recovered without obstetrical complication or infection in the child . azithromycin represents a first - line treatment option for tularemia during pregnancy in regions where the infecting strains of francisella tularensis have no natural resistance to macrolides .

cutaneous amyloidosis were classified into primary cutaneous amyloidosis ( pca ) , secondary cutaneous amyloidosis , and systemic cutaneous amyloidosis . pca is a rare , chronic progressive skin disease , defined as deposition of amyloid in previously apparent normal skin without systemic involvement . its prevalence were rarely reported . until now , there are fewer than 40 published cases worldwide . etiological factors associated with pca is still unknown , but its striking familial occurence suggests the role of genetic . we report a case with peculiar mottled pigmentation originally referred for vitiligo , but later proved as acd by histopathological examination . the disorder was thought to be familial as his siblings were affected with the similar condition . a 12-year - old boy presented with asymptomatic , generalized mottled hypo- and hyper - pigmented lesions of 6-year duration . the hypopigmented macules were first noticed on his lower extremities and had been slowly progressing to involve almost the entire body ( figure 1a ) . he did not have any history of systemic or cutaneous disease before the onset of the lesions . history of trauma , rubbing of the skin with any material , and extensive sun exposure was denied . his 11-year - old sister and 6-year - old brother experienced similar yet milder symptoms . physical examination revealed an extensive , discrete , pigmented macules distributed nearly all over the body in symmetrical pattern . he was referred to our hospital for skin biopsy with the initial differential diagnoses of vitiligo and pityriasis alba . histopathological examination ( figure 2 ) showed uneven distribution of melanin in the epidermis and deposits of pale pink amorphous material in the papillary dermis . based on clinical presentation and the histopathology examination , we made the diagnosis of primary cutaneous amyloidosis , presented as amyloid cutis dyschromica . the patient was treated with oral acitretin 25 mg per day . by the third month , some improvement was observed as the pigmented macules were slightly lightened ( figure 1b ) . the patient could tolerate the treatment , as there was no significant increase of transaminases and the lipid profile . the diagnosis of acd in our patient was not readily recognized as it mimicked , to some degree , other relatively common disorders with pigmentation feature . that , combined with its low prevalence , had eluded previous attempts at the correct diagnosis and treatment . the peculiar asymptomatic mottled pigmentation is much likely seen in poikiloderma , but as our case showed , without the corresponding signs such as telangiectasia or atrophy . furthermore , our patient was otherwise healthy , showing no signs that might indicate systemic disorders or photosensitivity , e.g. dermatomyositis , lupus erythematosus , or xeroderma pigmentosum . as defined by morishima , acd characterized by diffuse speckled hyperpigmentation with hypopigmented spots without papulation , atrophy , and telagiectasia , mild or no itching , onset before puberty , and focal amyloid deposition in the papillary dermis . duh begins in infancy or childhood , and unlike acropigmentation of dohi , might encompass the whole skin surface with exception of face . for definite diagnosis , histopathologic examination should be shought . the most common epidermal findings of pca were hyperkeratosis , irregular acanthosis with thinning of rete ridges , and expansion of dermal papillae by amyloid deposition . the finding of amyloid bodies in the papillary dermis was crucial in establishing the diagnosis of acd in our patient and disproved duh , in which such deposition was absent . its visualization under polarized light , showing apple - green birefrigence , confirmed the presence of amyloid . special histochemical stains were helpful for confirming the existence of amyloid . in our case , we tried to obtain histopathological examination of the siblings , but the parents denied the request to perform biopsy on the grounds that their clinical appearances were quite similar that the histopathological findings would likely be the same . multiple factors such as race , genetic , and enviromental may play collective roles , making variable degrees of cutaneous amyloidosis . although most cases of acd were sporadic , many have also reported positive family history of pca , suggesting that the important role of genetic factors in its pathogenesis . our patient s siblings experienced similar yet milder symptoms that the disorder was thought to be familial . , familial relationship was found in 5 of 10 patients , and consanguinity was denied in all . amyloidosis cutis dyschromica is assumed to be a congenital disorder and exposure to sunlight is thought to be the major causal factor . the lesions in our patient were more pronounced on exposed parts of body , that we thought sun exposure might be an important cause . acitretin was given and seems to be effective because it may act by minimizing keratinization defect that causes keratin degeneration to amyloid as proposed by some to be the pathogenesis of this disorder . qiao et al . in his series has convincingly shown 100% positivity to immunohistochemical staining for keratin , ck34 e12 and ck5/6 , that the amyloid is thought to be of epidermal origin . that points at a disturbance of keratinocyte repairs after irradiation with ultraviolet . at the third month his serum transaminase and lipid profile were only slightly increased , that we think the drug was safe to be continued . however , it is interesting to note that some authors consider certain populations of asia , e.g. chinese , japanese and thai , particularly susceptible . therefore , it is possible that several cases of this elusive disorders exist , but are often misdiagnosed . on the other hand , the treatment of acd still remains a challenge .

amyloidosis cutis dyschromica ( acd ) is an extremely rare type of primary cutaneous amyloidosis . to date there are fewer than 40 published cases worldwide ; some were reported affecting several family members . its resemblance to other common pigmentation disorders makes it rarely recognized at first sight . our patient , the 12-year - old firstborn son of non - consanguineous parents presented with generalized mottled pigmentation starting from lower extremities . his siblings suffered from similar condition . the clue for diagnosis is the amyloid deposition in the papillary dermis . the etiology of acd is still unknown , but genetic factors and ultraviolet radiation are implicated . it is proposed that disturbance of keratinocyte repair following ultraviolet radiation results in amyloid deposition . the treatment remains a challenge . oral acitretin treatment , thought to repair keratinization defect , gave a slight improvement in our case . our is the first case of acd reported in indonesia .

a 45-year - old man presented to our outpatient department with painless progressive diminution of vision both eyes for the past 10 days . medical history included xdr - tb on treatment with linezolid ( 600 mg / day ) , ethambutol ( 800 mg / day ) , moxifloxacin ( 400 mg / day ) , cycloserine ( 500 mg / day ) , ethionamide ( 500 mg / day ) , and kanamycin ( 750 mg / day ) for the past 6 months . on examination , his visual acuity was 20/200 ( ou ) , not improving with pin hole . anterior segment examination was unremarkable and pupils were 3 mm , round , regular , and reacting to light in both eyes ( direct and indirect ) . fundus examination revealed hyperemic disc with blurred margins ( ou ) [ figs . 1 and 2 ] . visual field evaluation by humphrey field analyzer showed peripheral constriction and quadrantanopia in the right eye [ fig . 3 ] and low reliable fields in the left eye [ fig . 4 ] . optical coherence tomography ( oct ) revealed increased retinal nerve fiber layer ( rnfl ) thickness in both eyes [ fig . 5 ] . fundus photograph showing disc edema in right eye fundus photograph showing disc edema in left eye visual field showing peripheral constriction and superotemporal quadrantanopia in right eye visual field left eye showing quadrantanopia ( low reliability ) stratus oct showing increase in retinal nerve fiber layer thickness involving superior and inferior quadrants in right eye and superior , inferior , and nasal quadrants in left eye ( arrows ) ethambutol - induced toxic optic neuropathy was initially suspected and tablet ethambutol was discontinued after discussing with the treating physician . after two weeks , the patient 's visual acuity had dropped to 20/400 in both eyes , and the fundus picture remained unchanged . hence , the possibility of toxic optic neuropathy due to linezolid was considered as reported in the literature and linezolid was discontinued ( a total cumulative dose of 126 g had been already consumed by the patient ) . color vision was restored to normal and patient 's vision was restored to 20/20 after one month . fundus examination revealed resolved optic disc edema with setting in of temporal pallor in both eyes [ figs . the patient is under regular follow - up and no toxic effects have been noted at three months of follow - up . fundus photograph showing resolved disc edema with temporal pallor of optic disc in right eye fundus photograph left eye showing temporal pallor of optic disc follow - up visual field after discontinuation of linezolid showing partial recovery in right eye follow - up visual field in left eye after discontinuation of linezolid stratus oct showing reduction in retinal nerve fiber layer edema in both eyes toxic optic neuropathies are characterized by gradual , progressive , painless , bilaterally symmetric visual loss affecting central vision , and causing central or centrocecal scotoma . xdr - tb is defined as resistance to at least rifampicin and isoniazid among the first - line anti - tb drugs ( which is the definition of multidrug - resistant tb ) in addition to resistance to any fluoroquinolone and at least one of the three injectable second - line anti - tb drugs ( capreomycin , amikacin , kanamycin ) . linezolid inhibits protein synthesis by preventing formation of the ribosome complex that initiates protein synthesis . its unique binding site located on 23s ribosomal rna of the 50s subunit results in no cross resistance with other drug classes . hence , linezolid is being increasingly used for the treatment of infections caused by multidrug - resistant gram - positive bacteria . long - term linezolid interferes with bacterial ribosomes and also with mammalian ribosomes , thereby disrupting mitochondrial oxidative phosphorylation and protein synthesis . serious adverse reactions demanding withdrawal of the drug include myelosuppression , peripheral and optic neuropathy , lactic acidosis , and serotonin syndrome . the safety of linezolid treatment has been established for use only up to 28 days . there are several case reports of linezolid - induced optic or peripheral neuropathy in patients treated for a time period beyond 28 days . only two cases of toxic optic neuropathy have been reported following short - term linezolid treatment of 16 days . fundus picture can be varied , showing temporal pallor , disc edema , or essentially normal . the most common indication for long - term linezolid therapy in these patients has been infection with methicillin - resistant staphylococcus aureus . in our case , neuropathy occurred after linezolid had been used for six months at a dose of 600 mg per day for infection with mycobacterium tuberculosis . we attribute toxic optic neuropathy to linezolid in our patient because even two weeks after stopping ethambutol there was deterioration of vision , and it was only after withdrawal of linezolid that visual improvement started . linezolid is recommended by the world health organization ( who ) to treat drug - resistant tuberculosis as a medicine with unclear efficacy . linezolid , approved for gram - positive infections in 2000 has been used off - label for drug - resistant tuberculosis . there is limited data available regarding the efficacy and safety of linezolid in multidrug - resistant tb since it is always administered as part of combination therapy . linezolid may improve the chance of bacteriological cure only in the most complicated xdr - tb cases . its safety profile precludes its use in cases for which there are other alternatives . with increasing emergence of xdr - tb , for which treatment options are limited , physicians are compelled to resort to new drug therapies . although ethambutol is the most common antitubercular drug implicated to cause toxic optic neuropathy , it is pertinent to be aware that if withdrawal of one drug does not show visual recovery or there is further deterioration of vision , the possibility of toxicity due to other drugs should be thought of . with our country bearing the brunt of tuberculosis , ophthalmologists and physicians must be aware that monitoring of visual function is important in patients on long - term linezolid therapy and that early recognition of toxicity and discontinuation of drug results in complete visual recovery .

many systemic antimicrobials have been implicated to cause ocular adverse effects . this is especially relevant in multidrug therapy where more than one drug can cause a similar ocular adverse effect . we describe a case of progressive loss of vision associated with linezolid therapy . a 45-year - old male patient who was on treatment with multiple second - line anti - tuberculous drugs including linezolid and ethambutol for extensively drug - resistant tuberculosis ( xdr - tb ) presented to us with painless progressive loss of vision in both eyes . color vision was defective and fundus examination revealed optic disc edema in both eyes . ethambutol - induced toxic optic neuropathy was suspected and tablet ethambutol was withdrawn . deterioration of vision occurred despite withdrawal of ethambutol . discontinuation of linezolid resulted in marked improvement of vision . our report emphasizes the need for monitoring of visual function in patients on long - term linezolid treatment .

nonsteroidal anti - inflammatory drugs ( nsaids ) are the most commonly used medications during the dental treatment for their creditable efficacy in reducing pain and inflammation.1,2 however , the burden of their unwanted side effects is high particularly with traditional nsaids.1,2 this is more likely due to chronic use and reflecting the fact that nsaids are used extensively in the more vulnerable elderly population.1 the reported adverse effects include gastrointestinal and cardiovascular events , alterations in renal function , effects on blood pressure , hepatic injury , and platelet inhibition which can lead to increased bleeding.1 this case report describes an unusual rare adverse event of the use of ibuprofen for pain control post restorative treatment . a 26-year - old , otherwise healthy male has reported to the restorative clinic at al - farabi college , riyadh for investigation and management of pain affecting the posterior left region of the maxilla . his history of chief complaint showed typical presentation of severe pain due to advanced buccal cervical carious lesion . based on clinical findings , radiographic interpretation , and vitality tests , the carious lesion was removed and replaced by composite material ( filtek p90 , 3 m ) . in addition , cleaning and shaping were performed using crown down technique with protaper rotary instruments ( dentsply ) . the patient reported to has an increased erectile function and libido after 2 h from taking the ibuprofen tablet . he has reported that has done three sexual intercourses with his wife at that day . on the next day , he was curious , and he repeated taking the ibuprofen tablet . surprisingly , he had the same experience of his first use of ibuprofen of having increased libido and erectile function that has lasted for at nearly 6 h. he contacted us to question this unusual experience . his medical history was re - reviewed thoroughly with an endocrinologist , and nothing was mentioned . furthermore , tests for complete blood count , testosterone , thyroid stimulating hormone , cholesterol pre and post ibuprofen intake were assessed , and all results came up as normal . the unwanted side effects are well - reported . to our knowledge , this is the first report to present an unusual side effect of increased libido and erectile function post use of ibuprofen . we searched the literature using medline , accessed via the national library of medicinepubmed interface ( http://www.ncbi.nlm.nih.gov/pubmed ) , for articles relating to the existence of reports of adverse events of erectile dysfunction in relation to the use of ibuprofen or nsaids written in english from 1966 to september 2014 . we used the following mesh terms ( nsaids , ibuprofen , erectile dysfunction , libido , and adverse event ) . two relevant reports were found and presented in table 1 . these included studies reported sporadic erectile or sexual dysfunction associated with the use of different types of nsaids . in an animal model study , uqochukwu et al . ( 2011 ) found that the treatment with nimesulide has an impact on the testosterone and estradiol levels . however , at the doses studied , there were no significant changes in testicular architecture except for mild degenerative changes.5 the most recent study showed that the aspirin was effective in improving lithium - related sexual dysfunction in men with stable bipolar affective disorder.6 with no doubt , we can not speculate that ibuprofen can improve sexual dysfunction , but nsaids could have a role in treating such patients .

nonsteroidal anti - inflammatory drugs are the most commonly used medications for pain control in dentistry . the reported adverse effects include gastrointestinal and cardiovascular events , alterations in renal function , and effects on blood pressure , hepatic injury , and platelet inhibition which can lead to increased bleeding . this case report describes an unusual rare adverse event of the use of ibuprofen for pain control post restorative treatment . a 26-year - old , otherwise healthy saudi male reported an unusual side effect of increased libido and erectile function post use of ibuprofen . the medical and laboratory tests have failed to identify a link between this rare adverse event and either underlying conditions or possibly related etiology . this case represented a puzzling challenge with no clear explanation .

maxillofacial injuries have the potential to cause airway compromise and are associated with pain and swelling causing difficulty in mouth opening , chewing and deglutition . it helps in intraoperative pain relief and also helps in early post operative rehabilitation of maxilla facial trauma patients mandibular nerve block is often performed for diagnostic , therapeutic and anesthetic purposes for surgery involving mandibular region . we report a case where intraoperative and post - operative pain in a case of unilateral fracture mandible was effectively managed through intermittent mandibular nerve block via a catheter . a 30-year - old male with right sided parasymphyseal fracture mandible was scheduled for open reduction and internal fixation . the patient had difficulty in opening the mouth due to pain ( 3 cm ) . informed written consent for the nerve block was obtained and visual analogue scale ( vas ) of 0 - 10 , was explained to patient . in the operation theater , neuromuscular blockade was achieved with vecuronium and anesthesia was maintained with o2 and n2o using controlled ventilation . the right side of the face was prepared for mandibular nerve block with lateral extraoral approach [ figure 1 ] . arrow showing skin site of epidural needle insertion for mandibular nerve block an 18-gauge i.v . cannula was inserted at midpoint of lower border of the zygomatic arch and was advanced perpendicular to face until it contacted the lateral pterygoid plate . the length of the cannula outside the skin was marked and cannula was redirected slightly posterior to reach behind the posterior border and was advanced further by 0.5 cm . catheter was tunnelled subcutaneously and the filter was attached to its other end [ figure 2 ] . for surgical analgesia , a bolus dose of 4 ml of 0.25% bupivicaine was given through the catheter . fentanyl 1mg/ kg i.v . was given only when there was more than 20% increase in heart rate or blood pressure above base line . the surgery lasted for 2 h. at the end of surgery , neuromuscular blockade was reversed and the trachea extubated . following extubation , the patient was conscious and pain free and then shifted to ward . post operatively , he received 4 ml of 0.25% bupivacaine through the epidural catheter every 12 h for two days . vas score was measured immediately after surgery and thereafter at 1 , 2 , 4 , 6 , 12 , 24 , and 48 h respectively . patient was observed for numbness at the surgical site , need for rescue analgesia , complications ( nausea , vomiting ) . numbness in area of lower jaw line was present throughout the period but subsided after discontinuation of local anesthetic through epidural catheter . he was discharged on fourth post operative day with advice for follow up in o.p.d . mandibular nerve block can be used to manage intra as well as post operative pain in cases of fracture mandible . we performed lateral extra oral approach because of restricted mouth opening and the need to retain the catheter for post operative analgesia . 18 g cannula instead of epidural needle was used to minimize the bleeding from pterygoid plexus of veins . the cannula was advanced further after contacting lateral pterygiod plate so that the catheter comes in vicinity of mandibular nerve and there is no displacement during jaw movements . the catheter was further tunnelled subcutaneously to prevent dislodgement and filter was used to prevent infection . there was excellent post operative analgesia achieved with this technique as shown in decreased pain scores , both static as well as dynamic .

mandibular nerve block is often performed for diagnostic , therapeutic and anesthetic purposes for surgery involving mandibular region . advantages of a nerve block include excellent pain relief and avoidance of the side effects associated with the use of opiods or non - steroidal anti - inflammatory drug ( nsaids ) . a patient with maxillo facial trauma was scheduled for open reduction and internal fixation of right parasymphyseal mandibular fracture . the mandibular nerve was approached using the lateral extraoral approach with an 18-gauge i.v . cannula under general anesthesia . he received 4 ml boluses of 0.25% plain bupivacaine for intraoperative analgesia and 12 hourly for 48 h post operatively . vas scores remained less than 4 through out observation period . the only side effect was numbness of ipsilateral lower jaw line , which subsided after local anesthetic administration was discontinued . patient was discharged after four days .

anatomists have observed that the length of the esophagus , i.e. the distance from the incisors to the esophagogastric junction , varies in different individuals from 32 to 50 cm . by endoscopy , the usual length of on adult essphagus is measured as 40 cm from the incisor to the point where mucosal change occurs , but the esophageal length varies from one person to another according to physical status . this variability is often experienced when trying to insert a ph meter , manometer or even nasogastric tube , or when placing the prosthesis for benign or malignant stenosis . we measured various lengths of esophagi in korean adults using a flexible fibercope and measured the external body intervals . then , we analyzed these data and extrapolated the various esophageal lengths with measurable external body intervals to obtain the correlations . the study was conducted for the measurement of esophageal length in 196 persons , 97 males and 99 females , with normal esophagogastric endoscopic findings . the distances from the upper incisors to 5 anatomical levels of esophagus ( cricopharyngeus narrowing , aortic narrowing , left main bronchus narrowing , hiatus and gastroesophageal junction ) were measured by an endoscope with 1 cm markings ( olympus co. q20 foreward view ) ( fig . 1 ) . each length was the mean value of double measurements during full inspiration and expiration respectively . the external body interval lengths studied were standing height , sitting height , the distance from the 7th vertebra to the coccyx and the distance from the upper incisors to the external occipital protuberance . the standing height and sitting height the distance from the 7th cervical vertebra to the coccyx was measured with a tape measure . a specially designed measure was used for the distance from the incisors to the external occipital protuberance ( fig . data analysis and correlation of esophageal lengths to external body intervals were done using spss / pc ( statistical package for social science / pc ) . observations have shown great variation in the length of the esophagus in adults but good correlation between esophageal lengths and external parameters . the measured lengths of external body intervals and various anatomical esophageal levels are depicted in table 1 . the mean standing - height and sitting - height of 196 persons studied were 160.808.64 cm and 86.605.91 cm , respectively . the interval from the upper incisors to each anatomical landmark of the esophagus ( cricopharyngeal narrowing , aortic arch compression , left main bronchus compression , hiatus , and esophagogastric junction ) were 15.70 cm , respectively . the true esophageal length , which is the distance from the cricopharyngeal narrowing to the esophagogastric junction , was 24.932.76 cm . the correlation of each esophageal anatomical level with external body intervals using the multiple stepwise reggression method is shown in table 2 . each of these 4 external variables was statistically significant , but better correlations were demonstrated by the h1 and h3 . although we employed the measurable distance ( h4 ) instead of the unmeasurable distance ( l1 ) , the equations using h4 and h2 did not express the esophageal lengths more accurately than the equation using h1 and h3 only . for example , the distance from the incisors to the esophagogastric junction ( l5 ) could be neatly expressed in simple equations as the following : l5a=0.178 h1 + 0.176 h3 + 2.219 ( r2=0.58 , p<0.01)l5b=0.242 h1 + 2.076 ( r2=0.55 , p<0.01 ) likewise , all other esophageal lengths could be approximated with the equations using h1 and h3 or with the equation using h1 only . the development of improved capability in medical and surgical managment of esophageal disease processes has been significant . to understand and appropriately use current technologic advancements , the anatomic properties of the esophagus available information about esophageal length , however , has been based on cadaveric measurements , because it is not feasible to measure esophageal length in vivo . in adults , the distances from the incisor teeth to the esophagogastric junction have been described to be in the range of 3250 cm by various studies , and the described distance from the cricopharyngeus narrowing to the esophagogastric junction usually varies from 20 to 30 cm . on the other hand , jackson described esophageal lengths according to various age groups . but such wide ranges of esophageal values and different physical status even in the same age group hamper the application of these data to individual patients in esophageal procedures . the best approximation of esophageal length would be useful for various clinical procedures , such as placement of a ph meter , manometer and nasogastric tubes , insertion of an endoprosthesis and colonic interpostition for both benign and malignant stenotic segments . we conducted this study to determine the mean esophageal length of various anatomical levels and to derive a method approximating true esophageal lengths . the mean esophageal lengths from the upper incisors to the esophagogastric junction were 42.612.42 cm in men and 39.472.24 cm in women . these values are not smaller than western data despite the fact that koreans are shorter . this is possible because previous data were based on cadaveric examination and koreans have a different racial stature . we correlated several external body intervals with esophageal lengths by the multiple stepwise regression method : standing height , sitting height , the distance from the 7th cervical vertebra to the coccyx and the distance from the upper incisors to the occiput . all external parameters significantly correlated with the true esophageal lengths . based on our results , the distance from the upper incisors to the esophagogastric junction ( l5 ) can be best approximated by employing following equations : l5=0.178 h1 + 0.176 h3 + 2.219 ( r2=0.58 , p<0.001)l5=0.242 h1 + 2.078 ( r2=0.55 , p<0.01 ) but accuracy determined by the other equations using more variables other than height did not increase remarkably despite their complexities . likewise , other anatomical levels of the esophagus can be predicted accurately as shown in the previous results . in conclusion , we consider that an accurate appraisal of the true length of an adult esophagus can be made by measuring the height and the other external measurable parameters .

previous data for esophageal lengths are based on age related groups or cadaveric examination , both of which are improper for application to various individualized procedures . because of the variability in height for any given age and longer length in vivo , lindividualized approximation is needed . thus , we measured various anatomical levels of the esophagus using a flexible endoscope and correlated these data with measurable external body intervals.the mean esophageal length was 40.922.79 , which is longer than the length derived from cadaveric examinations . also , esophageal lengths which correlated better with height than any other body intervals , could be calculated from the following equation . esophageal length from the upper incisors to the esophagogastric junction ( l ) can be expressed : l = 0.242 height ( ht ) + 2.078 cm ( p = 0.000 , r2 = 0525).the data from our study indicate that esophageal length can be reliably predicted using external body parameters before employing various procedures .

synchronous primary lung cancers are uncommon and the occurrence of synchronous non small cell lung carcinomas ( nsclc ) with different histological morphologies within the same lobe is rare . the aim of this report is to discuss surgical and oncological management of this entity together with a review of the current literature . a 61-year - old female smoker ( 42 pack years ) was found to have left upper zone shadowing on chest x ray following a history of weight loss . her performance status was 1 with a forced expiratory volume in one second ( fev1 ) of 1.94 litres . positron emission tomography in conjunction with computed tomography ( pet - ct ) showed two nodules in the left upper lobe . there was a 2 cm spiculated lesion in the anterior segment with a standardised uptake value ( suv ) max of 5.6 units ( fig 1a ) and a 2.2 cm cavitating lesion in the apico - posterior segment with a suv max of 2.7 units ( fig 1b ) . coronal view pet ct scan of a high uptake nodule in the left upper lobe coronal view of pet ct scan of moderate uptake synchronous nodule in the left upper lobe a left thoracotomy with upper lobectomy was performed and the intra - operative findings were consistent with the radiological appearances seen in the pet - ct scan . there was a 2 cm infiltrating squamous cell carcinoma in the anterior segment with vascular invasion ( pathological ( p ) t2a according to tnm 7th edition ) and a separate 2.4 cm adenocarcinoma with associated broncho - alveolar spread in the apical segment ( pt1b ) . an adjacent peribronchial lymph node was positive for tumour ( pn1 ) , however due to the poorly differentiated morphology of the tumour cells it was unclear as to which tumour it originated . cisplatin and vinorelbine adjuvant chemotherapy was administered with no overt complications ; however recurrence of the disease was discovered radiologically one year post - operatively in the form of a left hilar soft tissue mass with bony metastases . the patient subsequently received radiotherapy without complications but a staging ct chest / abdomen / pelvis performed three months later showed progressive disease with left lung collapse in addition to multiple , palpable subcutaneous nodules . these features were in keeping with the patient s symptoms of worsening breathlessness and wheeze . further investigations including bronchoscopy showed a tumour in left main bronchus which was treated with laser ablation and placement of a tracheo - bronchial stent . histological assessment of this nodule showed a poorly differentiated adenocarcinoma , which was confirmed with immunohistochemical techniques . specialist genetic analysis of the subcutaneous lesion was performed , which showed the presence of an egfr mutation sensitive to anti - egfr tyrosine kinase inhibitors . this enabled the oncologists to use monoclonal antibody therapies ( anti egfr tki ) and the patient was treated for one month with iressa ( gefitinib ) . she responded well with significant decrease in the size of the metastatic subcutaneous nodules . due to her progressive disease the patient deceased with an overall survival ( from the time of the lung resection ) of 20 months synchronous primary lung cancers ( splc ) were first described in 1924 by beyreuther h ( 1 ) . the true incidence of these remains uncertain but evidence has shown figures between 1% to 8% ( 2 ) . the criteria for the diagnosis of splc , which was proposed by martini and melamed in 1975 , are that synchronous tumours are physically distinct and that the histology is different surgical resection of splc was recommended by many of the reviewed authors ( 3 - 5 ) . rostad et al studied the outcome and characteristics of synchronous primary lung cancers . in 15,308 lung cancer resection cases , 94 patients were found to have synchronous non small cell lung cancers , nsclc , 9 patients had synchronous lung cancers with different histological morphologies and only 2 ( 0.01% ) patients had synchronous lung cancers with different histological morphologies within the same lobe . the relative survival rate in patients with different histological morphologies ( n=9 ) was 12.7% . patients with similar morphologies had a better outcome with a relative survival rate of 29.2% , although the difference was not statistically significant ( p=0.24 ) . the authors concluded that surgical resection should be offered to patients with synchronous lung cancers who are operable with respectable tumour . ( 3 ) lymph node metastases were found to be a statistically significant prognostic factor . in a study of 92 patients who had surgical resections for multiple synchronous primary lung cancers , the results showed the 5-year survival were 52.5% and 15.5% for patients without and with lymph node metastasis respectively ( p = 0.001 ) . ( 3 ) skin metastases from lung cancers are not uncommon , and their presence is a poor prognosis factor ( 6 ) . recently anti - egfr tki molecules have been introduced for the treatment of advanced malignancies including lung cancers , however long term treatment resistance remains a therapeutic challenge . our patient had a disease free survival of 12 months post surgery despite a positive lymph node , but she later developed recurrence with distant subcutaneous metastases and an endobronchial mass , which was treated palliatively . she received anti egfr1-tk inhibitors based on genetic analysis from the metastatic adenocarcinoma subcutaneous lesions and her initial response was good . patients with resectable synchronous primary non small cell lung carcinomas within the same lobe should be offered surgical resection after careful pre - operative staging . skin metastases should be biopsied and egfr testing should be requested in order to determine the originating the tumour and to assess the patients suitability for anti egfr - tki treatment .

we report the case of a sixty one year old female diagnosed with two synchronous primary lung cancers located within the same lobe . surgical resection was performed , followed by adjuvant chemotherapy . the patient developed distant bone and skin metastases one year post - surgical resection . in this report we discuss the multimodality therapy used to treat this patient .

in a book titled doing psychiatry wrong the author ( a psychiatrist ) describes several cases treated with medications , mostly inappropriately ( e.g. , patient with borderline personality treated as bipolar disorder , multi drug abuser treated as schizophrenia ) , without much gain . another author ( a sociologist ) argues that in spite of the evidence that psychiatric conditions improve best with a combination of medicines and therapy , psychotherapy is rarely offered . that author 's research has shown that such patients who were treated only with medicines do less well , are readmitted more quickly , diagnosed more inaccurately and medicated more randomly . this case was inaccurately diagnosed , randomly medicated and did less well . properly conducted psychotherapy has improved the patient . are we in to a second wave of anti - psychiatry movement against biological imperialism ? a 23-year - old male , b.e student from upper middle socio - economic status , accompanied by his parents , presented with the complaints of anger outbursts , inability to mingle with people and inability to study . for the past 2 years , he attended neither the classes nor the home tuitions . most of the activities , which he tried to do ended in failure and angry frustrations . most of the time was spent in sleep , which was aided by sedative psychotropic medicines . he could not tolerate inactivity as it led to boredom and this was intolerable . remaining small time in the day he displayed a typical pattern of behavior as noted by pedesky and beck they may discontinue a task or fail to initiate a task they had planned to do . they may turn on the television , pick up some things to read , reach for food or a cigarette , get up and walk around and henceforth . mental status of the patient was that of an inhibited plump person with expressionless serious face , answering questions minimally , but relevantly without any psychotic symptoms . most of his anger outbursts erupted and lasted only for a few seconds and a few are followed by grumbling and shouting for about 10 min . anger resulted in yelling with angry gestures , banging , crumpling , throwing , tearing and breaking of objects ( one per attack ) such as shirt , pen , pencils , spectacles , remote control and rarely , mobile phones or computers . he avoided close relatives , strangers and crowds as it induced severe fear and inhibition . he was scared to talk to house maid , lift operator in the apartment , traffic police and ladies . he looked away from them and walked away at the prospect of an approaching lady . in fact his first visit to a psychiatrist was due to fear of lead contamination from wall paints . his other fears were , being cursed by god , old and sick people if they are disturbed by mistake , fear of going deaf or blind by strong sounds or bright light . he had magical beliefs that bad words if heard in the early morning , they will spoil the whole day and if any bad word is heard while praying , god will curse . he had rigid moral values in the matters of sexuality , religion and right or wrongs . he got infuriated with matters such as muslim religion , female gender , britons , beef eaters and rule breakers . he was pre - occupied with his sagging chest and wore tight banyans to hide it . he wanted to understand the subject perfectly and this inability led to a sense of failure and frustration and anger . patient was born to a highly educated and well - employed couple hailing from upper socio - economic status . his first degree paternal uncle has a chronic psychotic illness , but he is too highly educated and regularly employed . however , he was noted to be angry and used to kick walls if he got angry . in 10 and 11 standard he was bullied frequently by few students and ridiculed for his sagging chest by touching it . patient remained un - assertive and the abuse and emotional trauma inflicted was significant . during adolescence his past psychological assessments revealed above average intelligence and presence of several personalities disorder traits and low self - esteem . he was treated by more than a dozen of psychiatrist , exhausting all psychotropic medicines , including clozapine at 200 mg / day . when he visited the author he was on lithium 400 mg , fluoxetine 60 mg , amisulpride 100 mg , quetiapine 50 mg , clonazepam 3 mg , pregabalin 300 mg . cognitive therapy espoused by pedesky and beck was administered over 1 years , weekly 1 - 2 sessions of 1 - 2 h duration . schema modifications , behavioral experiments , cognitive restructuring , brief repeated exposures ( real and imaginary ) , exposure and/or response prevention for obsessional symptoms were used during therapy . psychiatrist trained to diagnose patients using atheoretical diagnostic systems and to treat by medicines is likely to over diagnose a psychotic illness in this case and treat accordingly . a flat affect , minimal talk , inhibited appearance , apparently unexplained anger outbursts and psychotic illness in a family member can lead to a misdiagnosis . a closer look reveals his warm affect , a keen interest to socialize and reasons for his anger . in fact historically this patient might have diagnosed as pseudo - neurotic schizophrenia , because of pervasive fears , phobias and strange sexual fantasies . in the past , severe avoidant cases were clubbed with schizoid personality . knowledge of cognitive theory and therapy helped the author to arrive at a right understanding ( diagnosis ) of the patient and consequently the proper treatment . patient warrants an additional diagnosis of intermittent explosive disorder because aggression is not a part of avoidant personality . he also had anankastic personality disorder , obsessive - compulsive disorder , body dysmorphic disorder and paraphilia and even a dhat syndrome . one of the foundations of cognitive theory and therapy is that the appraisal ( personal meaning given ) of the events results in the emotions . similarly , his social fear can be explained by extreme sense of shame , sensitivity to insult and appraisal of people as dangerous and paranoid ( patient 's own word ) about him . the typical pattern of behavior noted earlier , can be explained by the cognitive and emotional avoidances . patient was intolerant of dysphoria and to reduce it , he repeatedly attempted to do one or the other activity . this case proves that psychotherapy can be a mainstay of treatment even in severe personality disorders .

over dependence on pharmacotherapy in psychiatry , known as biological imperialism , is a world - wide phenomenon . some authors have opined that the inadequate and ineffective utilization of psychotherapeutic interventions and only dependence on pharmacotherapy amounts to institutional malpractice . here is an example of such a case . a young male mainly received multiple psychotropic medicines , including clozapine ( and also a failed psychotherapy ) over a period of 4 years , without any benefit . his global assessment of function score remained at 30 . however , with proper diagnosis and effectively conducted psychotherapy a significant improvement in global assessment of functioning score of 70 was achieved , over a period of 1 years .

gastric volvulus , an entity seen in both pediatric and adult patients , occurs when the stomach twists upon itself . this event may be transient , producing few if any symptoms , or may lead to obstruction or even ischemia and necrosis . pare described the first case of gastric volvulus in 1579 in a patient with diaphragmatic injury from a sword wound . acute gastric volvulus in pediatric and adult patients has been reported but chronic organo - axial gastric volvulus with diaphragmatic eventration has not been reported . this report describes a rare case of gastric volvulus with a review of the literature . he had a feeling of fullness and discomfort in the upper abdomen for 2 months . twenty years ago , he was told that his chest radiography , performed due to routine survey for military service , was abnormal . but he had received no specific measures since then and he had been relatively healthy . a thorough review of symptoms was performed but did not disclose any respiratory or cardiovascular symptoms . on clinical examination , he appeared not acutely ill looking and his body temperature was 36c , blood pressure 120/70 mmhg , respiratory rate 24/min . auscultation of the chest showed diminished breathing sound at the left lung base and heart sound was normal . no abnormalities were observed in cbc , liver chemistry , serum amylase and stool examination . the chest radiograph showed the left hemidiaphragm to be located at an unusually high intercostal space with large air - fluid level ( figure 1 ) . upper gastrointestinal series demonstrated the typical appearance of an organo - axial volvulus of the stomach ( figure 2 ) . the barium - filled stomach was twisted on an axis from the pylorus to the esophagus . the patient refused surgical or endoscopic correction of the volvulus and has been followed in the outpatient clinic for more than 6 months without symptom aggravation . the stomach is relatively fixed at the esophageal hiatus and the pylorus by the four gastric ligaments . the gastrophrenic ligament and the retroperitoneal attachment of the second part of the duodenum provide the superior and inferior fixation . the gastrohepatic ligament tethers the lesser curve , the gastrocolic ligament connects the stomach to the transverse colon , and the gastrosplenic ligament tethers the greater curve . the clinical symptoms depend upon the extent or degree of rotation , obstruction and associated defect . borchard s triad of pain , violent retching and inability to pass a nasogastric tube6 should lead to a strong clinical suspicion of acute gastric volvulus . an acute volvulus is an emergency situation , with either obstruction or strangulation of the stomach and requires expeditious surgery . in comparison , this explains why the diagnosis is often delayed in the elderly or after complication has occurred . . gastro - esophageal reflux may give rise to epigastric pain , which is intermittent during the periods of engorgement or gastric emptying . gastric ulceration is caused by localized ischemia and acidity within the herniated stomach or mucosal congestion due to venous obstruction of the herniated stomach . angina - like pain and electrocardiographic abnormalities may make the differential diagnosis difficult in the elderly . the diagnosis is confirmed by the presence of a large , unusual gas - filled viscus in the chest or abdomen on plain radiographs . if necessary , a barium swallow study can define the anatomic changes more exactly . on barium examination , the characteristic findings are 1 ) esophagogastric junction lying lower than normal , 2 ) reversal of the greater and lesser curvatures , 3 ) pylorus pointing downward , 4 ) greater curvature crossing the esophagus , 5 ) two air - fluid levels and lowering of the gastric fundus . secondary gastric volvulus is more frequent than idiopathic volvulus , therefore the diagnosis of gastric volvulus can be made after a thorough search for possible causative factors . these conditions have been reported as follows ; para - esophageal hernia , traumatic rupture of the diaphragm , eventration of the diaphragm and phrenic nerve injury . in infants and children , 15 ( 33% ) among 46 patients with gastric volvulus had diaphragmatic eventration . organo - axial volvulus is commonly associated with diaphragmatic hernia and usually manifests as an acute event . diaphragmatic eventration is suggested when a part or all of the hemidiaphragm is located at an unusually high level in the thorax . it does not refer to a defect or hole in the diaphragm with discrete edges , but rather to a diffuse or localized bulging of the diaphragm itself . acquired lesions are usually related to phrenic nerve injury , which may be diverse in origin . radiological investigation combined with fluoroscopy of the diaphragm our patient showed no diaphragmatic movement during respiration , so he had complete left hemidiaphragmatic eventration . acute gastric volvulus and symptomatic chronic gastric volvulus require operative treatment . if the volvulus is secondary , definitive treatment must include correction of the associated abnormalities . this is done by advancing the endoscope just past the point of torsion , turning and locking the tip of the instrument , and rotating it 180 degrees . with rapid diagnosis and modern treatment , the rate of mortality from acute gastric volvulus is now about 15% to 20% . if the stomach is strangulated , the mortality rate of emergency surgery is 4060% . in recognized cases of chronic gastric volvulus

gastric volvulus occurs when the stomach rotates about its longitudinal axis ( organo - axial volvulus ) , or about an axis joining the lesser and greater curvatures ( mesentero - axial volvulus ) . primary gastric volvulus , making up one third of cases , occurs when the stabilizing ligaments are too lax as a result of congenital or acquired causes . secondary gastric volvulus , making up the remainder of cases , occurs in association with a paraesophageal hernia or other congenital or acquired diaphragmatic defects . while gastric volvulus may occur acutely , especially in children , it may not be clinically apparent and discovered incidentally . the authors present a case of chronic organo - axial volvulus of the stomach secondary to left hemidiaphragmatic eventration with a review of the relevant literature .

dilatation of various lengths and severity of the common bile duct ( cbd ) , entitled choledochal cyst , has been detected in utero and usually presents with icterus in infancy , clinically mimicking biliary atresia and neonatal hepatitis1 ) . younger children and occasionally infants tend to present with painless jaundice , and older children present with recurrent abdominal pain , which was actually due to acute pancreatitis12 ) . postnatally , ultrasonography ( us ) is the initial diagnostic modality of choice , allowing for precise measurements of intra- or extrahepatic duct dilatation and identification of stones and sludge . magnetic resonance cholangiopancreatography ( mrcp ) and endoscopic retrograde cholangiopancreatography ( ercp ) has superseded the use of computed tomography ( ct ) for preoperative anatomical delineation of the pancreaticobiliary tract . here we present the biggest choledochal cyst reported in infancy in the literature to our knowledge . a term female baby was born by normal delivery route after consanguineous marriage , at house , in syria as the 6th child of her parents . this family was living the civil war in syria at that time . during 4 months period abdominal distension had increased . because of restlessness and growing abdominal distension but as a result of investigations they were sent home because of normal laboratory results . after 15 days they admitted to hospital because of jaundice and abdominal cyst she had referred to our hospital ( in turkey ) with the preliminary diagnosis of abdomianl cyst and hepatitis developed due to compression of cyst . on her admission , us revealed giant abdominal cyst with thin wall and liquid - debris level extending from right upper quadrant to pelvic region . it was suspected that bile duct 's dilatation was due to abdominal giant cyst 's pressure . she was operated with the differential diagnosis of duplication , omental or mesenteric cyst . at operation , a giant type 1a choledochal cyst , 160 mm in diameter , was surprisingly detected ( fig . serum levels of bilirubin decreased sharply and the patient was discharged without any problems on the tenth postoperative day . a choledochal cyst is a dilation that encloses the intrahepatic or both extra- and intrahepatic portions of the biliary ducts2 ) . type ia is a cystic dilation of the cbd ; type ib is a focal segmental dilation of the distal cbd ; type ic is a fusiform dilation of both the common hepatic duct and cbd . in type ii , the cyst forms a diverticulum from the extrahepatic bile duct . type iii , also known as choledochocele , is a dilation of the distal cbd lying mainly within the duodenal wall . type iv is essentially type i anatomy with either intrahepatic bile duct cyst ( iva ) or choledochocele ( ivb ) . some authors refer to caroli 's disease with multiple cystic dilations of the intrahepatic biliary tree as type v23 ) . in our case there was a cystic dilation of the cbd as a type ia choledochal cyst with minimal dilatation of intrahepatic bile ducts . this malformation primarily affects girls ( 4:1 ) and about 80% become symptomatic during childhood . choledochal cysts remain relatively uncommon in western europe and the united states , although they are appreciably more common in asia . obstructive jaundice is the main presentation symptom in children , but abdominal pain is the commonest symptom in adults . the classical triad of pain , jaundice and a palpable mass is uncommon , occurring in no more than 6% in one uk series4 ) . the complications of congenital cystic dilatation of the bile duct are biliary stone formation , progressive biliary cirrhosis with portal hypertension , and carcinoma . fusiform lesions are never large enough to be palpable while multiple intrahepatic type 4 lesions cause predisposition to stone formation and sepsis . a wide variety of imaging techniques are available which noninvasively reconstruct biliary anatomy and give an excellent idea of biliary function . although us is the first described imaging method to determine the cbd cyst , ct , mrcp , and ercp are superior to assess the extention of the cyst and associated pathologies such as cholangitis , pancreatitis and pancreaticobiliary junction anomaly . in our case , we used us and ct to identify the abdominal mass ' origin . because of the diameter of the cyst , cbd cyst was not thought in the differential diagnosis . cbd malformations should be kept in mind as a differential diagnosis of the cystic mass regardless of size , and patient 's age . surgery is the main choice of treatment but some centres in south american and asia have reported ercp and sphincterotomy alone as definitive treatment for mild fusiform dilatation although their long - term prognosis is not known3 ) . at operation , it was difficult to make the differential diagnosis of our giant cyst . it was important to make the dissection of the cyst carefully in order to avoid the iatrogenic injury . following the diagnosis of choledochal cyst was confirmed , hepaticojejunostomy was easier depending on the wide common hepatic duct of our huge cyst . tang et al.5 ) reported a study that involves 62 children ( average age of 2.3 years ) who had cysts with the average diameter of 42 mm ( range , 12158 mm ) . there is no information about the child'a age with the 158-mm cyst . as our knowledge we report the biggest choledocal cyst case in infancy in the literature . in conclusion , cbd malformations should be kept in mind as a differential diagnosis at the cystic mass regardless of cyst 's size , and patient 's age , especially in children presented with abdominal pain , jaundice , and palpable mass .

choledochal cyst is a dilation that encloses the intrahepatic or both extra- and intrahepatic portions of the biliary ducts . postnatally , ultrasonography is the initial diagnostic modality of choice , allowing for precise measurements of intra- or extrahepatic duct dilatation and identification of stones and sludge . symptoms depend on the age at presentation . common bile duct malformations should be considered as a differential diagnosis of a cystic mass regardless of the cyst 's size or patient 's age , especially in children presenting with abdominal pain , jaundice , and palpable mass . to the best of our knowledge , we report the largest choledochal cyst in infancy .

bilateral shoulder dislocation are most commonly posterior type . these are most commonly due to seizure disorder and electrocution . to best of our knowledge there are only few cases of similar kind are reported in literature . we hereby report a interesting case of posttraumatic , bilateral anterior dislocation of shoulder without associated fracture in a 45 old women without any predisposing pathoanatomy . a 45-year - old women presented to casualty with sudden onset of pain and restriction of movement in both shoulders fallowing trauma . immediately post trauma she had severe pain and restriction of both shoulders . on examination arms were abducted and externally rotated . there was loss of round contour of shoulder with increased vertical diameter of axilla anteriorly . bilateral traumatic anterior shoulder dislocations are rare and are seen as a result of unique mechanism of injury . in our case patient bilateral shoulder dislocations are usually posterior type and are almost pathognomonic of seizure disorder or electrocution . though anterior dislocation of shoulder is commonest bilateral simultaneous dislocation is very rare[1 - 7 ] . we hereby report a case of posttraumatic , bilateral anterior dislocation of shoulder without associated fracture in a 45 old women . a 45-year - old women presented to lok nayak hospital , new delhi , india in august 2010 with sudden onset of pain and restriction of movement in both shoulders fallowing trauma . she had no history of seizure , epilepsy , previous shoulder dislocation or instability in other joints . on examination arms . there was loss of round contour of shoulder with increased vertical diameter of axilla anteriorly . radiological examination revealed bilateral anterior dislocation of the shoulders without any associated fractures(figure 2 ) . radiograph showing bilateral shoulder dislocation concentric reduction of bilateral shoulder joint achieved closed reduction done by milch technique after intraraticular lignocaine injection . majority of the bilateral shoulder dislocations are of posterior type most commonly seen during convulsion , electric shock or hypoglycaemic seizures . posterior type is common in these conditions due to violent contractions of the muscles of the shoulder girdle [ 8 - 10 ] . unlike bilateral occurrence of anterior shoulder dislocation is rare because of the fact that one extremity takes the brunt of the impact . to best of our knowledge only three cases of bilateral anterior dislocations are reported in literature . in two of the three cases reported were sequential , one sided followed by contra lateral side dislocation . in our case impact is same on both shoulders at the same time . the mechanism of anterior dislocation is forced extension , abduction and external rotation of the arm . in our case mechanism of injury mechanism of injury , systemic disease and associated fractures in various similar cases is depicted in table 1 . croswell and smith reported a case of bilateral anterior dislocation of the shoulder without any fractures in a bench - pressing athlete . in an unusual mechanism of injury weight on the bar forced his arms into hyperextension in the mid - abducted position . the humeral shaft gradually pivoted on the bench and the humeral heads were slowly dislocated interiorly by the weight of the bar . sandeep s and sudhir k reported a case of sequential bilateral anterior dislocation in which the left shoulder dislocated first due to trauma followed by atraumatic dislocation of the right shoulder . sreesobh k v et al reported a case where atraumatic right shoulder dislocation was followed by traumatic dislocation of the left . this type of dislocation involves a unique type of mechanism injury and in our case it was fall on pointed elbow causing forced extension . traumatic bilateral anterior dislocations without any pathologic lesion are very rare with only few cases reported in literature .

introduction : bilateral shoulder dislocation are most commonly posterior type . these are most commonly due to seizure disorder and electrocution . anterior shoulder dislocations occurring bilaterally without any predisposing factors are very rare . these types of injuries are due to trauma with a unique mechanism of injury . to best of our knowledge there are only few cases of similar kind are reported in literature . we hereby report a interesting case of posttraumatic , bilateral anterior dislocation of shoulder without associated fracture in a 45 old women without any predisposing pathoanatomy.case report : a 45-year - old women presented to casualty with sudden onset of pain and restriction of movement in both shoulders fallowing trauma . immediately post trauma she had severe pain and restriction of both shoulders . on examination arms were abducted and externally rotated . bilateral shoulder movements were painful and restricted . there was loss of round contour of shoulder with increased vertical diameter of axilla anteriorly . radiological examination revealed bilateral anterior dislocation of the shoulders without any associated fractures . closed reduction done by milch technique after intraraticular lignocaine injection . mri of bilateral shoulder showed no pathological lesion . both shoulders were immobilized with a shoulder immobilizer for three weeks.conclusion:most of the bilateral shoulder dislocations are posterior type seen in seizure disorders . bilateral traumatic anterior shoulder dislocations are rare and are seen as a result of unique mechanism of injury . in our case patient had a fall on her elbows causing forced extension . if diagnosed and treated promptly completely normal function of the shoulders can be restored .

maculopapular exanthema ( mpe ) is the most frequent clinical manifestation of nonimmediate allergic reactions due to drugs and t helper 1 ( th1 ) cytokines and cd4 ( + ) t cells have been shown to play an important role in its pathogenesis . pyrazinamide is used in the management of tuberculosis ( tb ) in combination with other drugs . the common side effects due to pyrazinamide are hyperuricemia ( gout ) , hepatotoxicity , nausea , vomiting , flushing , dysuria , arthralgia , and sideroblastic anemia . we hereby report a case of maculopapular rash due to pyrazinamide in a patient undergoing antitubercular treatment . we also established the causality , severity , and preventability of the suspected adverse drug reactions ( adrs ) . a 19-year - old patient of bersarai area , new delhi , belonging to a lower middle class family , visited a microscopic center situated in delhi government dispensary of bersarai area with complaints of cough with expectoration and fever , loss of appetite , and weight for the past 1 month . the sputa were examined as per revised national tuberculosis control programme ( rntcp ) by zeihl - neelson ( zn ) staining for acid - fast bacillus ( afb ) and were found to be negative . chest radiograph showed bilateral upper zone infiltration . on the basis of the above clinical examination and observation he was referred to the directly observed treatment ( dot ) center of his area for the initiation of category i antituberculosis therapy as per national rntcp guidelines according to his weight ( 40 kg ) . category i antituberculosis therapy includes isoniazid 600 mg ( 2 tablets ) , rifampicin 450 mg ( 1 capsule ) , pyrazinamide 1500 mg ( 2 tablets ) , and ethambutol 1200 mg ( 2 tablets ) . ( after the second day of therapy ) , the patient visited dot center with generalized maculopapular rashes all over the body and more on both shoulders and upper and lower limbs . a diagnosis of antitubercular drug - induced maculopapular rash was made by the medical officer . rashes were round in shape , raised from the body surface , appeared reddish in color , and hot on touch . therefore , pyrazinamide was stopped on the advice of the medical officer and other antituberculosis medicines were continued with the addition of oral antihistaminic and the patient was kept under close observation for evaluation . he tolerated isoniazid , rifampicin , and ethambutol but on inclusion of pyrazinamide , rashes reappeared in the same part of the body . he is on regular follow - up with disappearance of rashes and signs and symptoms of tuberculosis . we carried out the causality assessments as per the naranjo algorithm and preventability and severity assessments as per the hartwig scale . the causality assessment revealed a probable association ( naranjo score 7 ) between the adr and pyrazinamide . higher rates were found in elderly patients who are likely to be receiving multiple medications for long - term illnesses . maculopapular rashes consist of macules ( distinct flat areas ) and papules ( raised lesions ) . the rash is usually bright red in color and the skin may feel hot with burning sensation or itch . the whole of the skin surface may be involved , though the face is often spared . up to 5% of the patients receiving penicillin , sulfonamides , phenytoin , or gold , erythema multiforme has been reported in one patient following pyrazinamide administration for cutaneous tuberculosis related to a pleural fistula . daily antituberculosis treatment ( att ) was initiated in this patient with isoniazid , rifampicin , ethambutol , and pyrazinamide . after 26 days of therapy , maculopapular erythematous lesions appeared , and biopsy results confirmed the diagnosis of erythema multiforme . the rash disappeared with the discontinuation of all drugs , but reappeared when rifampicin and pyrazinamide were reintroduced 5 days later . the patient developed the rash on the third day ( after second dosing day of therapy ) after initiating antituberculosis therapy and disappeared after few days when the drug ( pyrazinamide ) was stopped . the causal relationship between the drug and the adr was found to be probable . generalized maculopapular skin rash was a common adr reported in an investigational trial of ofloxacin ( 800 mg a day ) and pyrazinamide ( 1500 mg a day ) . the management of such reactions needed withdrawal of the suspected drug and management of symptoms , if any . in this study , the suspected drug was stopped immediately following the adr and antihistamines were added to manage associated itching due to drug reaction , to which patient responded well . the severity assessment revealed the adr to be moderate ( level 3 ) , suggesting that the suspected drug should be withheld , discontinued , otherwise changed , and/or on antidote or other treatment is required . since this patient did not have any past history of skin reaction due to pyrazinamide or any other drugs , therefore this reaction was unpreventable . since pyrazinamide is a common drug used in tb management , and tb is also a common problem in countries like india , the dermatological manifestations due to pyrazinamide gain attention . upon occurrence of dermatological manifestations , the patients may become noncompliant , which is one of the common causes with other anti - tb drugs for treatment failure in tb therapy . although skin reactions due to pyrazinamide are not well reported , one should be suspicious of maculopapular rashes due to pyrazinamide also . upon occurrence , the suspected drug/(s ) should be stopped immediately and the patient should be managed symptomatically . the patients undergoing treatment on an outpatient basis should be counseled for the early recognition of dermatological manifestations .

pyrazinamide is a commonly used first - line antitubercular drug . gastric - related adverse drug reactions are common with pyrazinamide . dermatological manifestations due to pyrazinamide are rare . this study aimed find out the dermatological manifestations / adverse drug reaction ( adr ) due to pyrazinamide . we reported a case of maculopapular rash caused by pyrazinamide in a patient on antituberculosis treatment using structured questionnaires . the patient developed maculopapular rashes on receiving combination antituberculosis treatment . the rashes disappeared after stopping the suspected drug . the patient was rechallenged with pyrazinamide , which led to reappearance of a similar type of rash . the causality , preventability , and severity were assessed using the naranjo algorithm and hartwig scale . since pyrazinamide is a commonly used drug in tuberculosis and which is a common infectious disease in developing countries , with the similar reports , we can predict early case detection and can prevent the occurrence of similar reactions in future .

fever in returning travelers is a common problem and usually the diagnosis is made within a few days or the traveler recovers , . we report here two travelers who presented with fever two weeks after returning from a six week vacation in south america . over the following 18 months they presented with short attacks of fever , elevated crp and leukocytosis and a curious and key feature was , that they were completely synchronous both developing symptoms within an hour and presentation with the same laboratory findings of leukocytosis and elevated crp . after about a year it turned out that the couple used aroma oils ( nature and decouvertes fig . 1 ) in the home applied by a nebulizer placed in the middle of a table . the oils were not used during summer , also explaining the absence of symptoms during the summer months . since the use of the oils ended there has been no relapse in any symptoms . we are not aware of or have found any previous reports , in english or other languages , reporting similar events after the use of similar products . after traveling to south america for a six weeks tourist holiday the two patients returned home to denmark . ten days after returning they were both admitted to hospital with fever , muscle and joint pains and vomiting . from the 8 september 2012 to the 22 october 2012 they spent 3 weeks in peru , half a week in bolivia , half a week in chile and 2 weeks in brazil . they traveled by local busses , stayed at medium level hotels and were not ill at any time during their travels . prior to their travels they had received vaccines against hepatitis a and b , yellow fever and tetanus / diphtheria . table 1 shows the patients history and table 2 lists crp , white blood cell count and recorded rectal temperatures . day 0 is defined as the first day the couple was admitted to hospital , 10 days after the return from south america . there was no eosinophilia at any given time and the total ige remained normal throughout . approximately nine episodes occurred within the following 18 months , all attacks being identical in symptoms , duration and paraclinical results for both patients ( table 2 ) . initially the patients were tested negative for malaria and dengue fever . under the suspicion of a rickettsial infection they were treated with doxycycline 100 mg two times daily initially for one week . this was immediately afterwards repeated again for one week . after the third relapse , day 20 , the patients received 3 months doxycycline 100 mg two times daily plus moxifloxacin 400 mg 1 , as it was believed they previously had had a good effect . the patients went through an elaborate program of serological tests and test for nucleic acids of different pathogens , which is summarized in table 3 . through the whole course a wide range of tests were performed including heart echocardiography , pet - ct and an mri which were all normal . definition of travel associated disease is a patient who has crossed an international boarder within the past 10 years and presents for a presumed travel - related disease . it is not uncommon for travelers to report an illness associated with their travels ( 2070% ) , but only a small portion of these actually seek medical attention , , . a detailed medical history is a very important tool in correct diagnosis , including destinations , risk factors , previous medical history . incubation time is also important to keep in mind through the process ( table 5 ) , , . the geosentinel surveillance program has found that the most common causes of fever after traveling is malaria , dengue fever , enteric fever ( salmonella typhi ) and rickettsioses . initially malaria and dengue fever were excluded and the patients were treated with doxycycline under the presumption of a rickettsia or bartonella infection . when the fever attacks continued we excluded endocarditis due to coxiella burnetii ( q fever ) and looked for south american trypanosomiasis due to trypanosoma cruzi ( chagas disease ) , which can be transmitted orally through fresh fruit juice . we speculated that toxoplasma gondii was a possibility as t. gondii genotypes in south america are more pathogenic compared to europe , but only one of the subjects had antibodies at a low titer , not compatible with an acute infection . leptospirosis was also a diagnostic option , especially with a second phase of fever shortly after the first ; this was however also excluded by a negative serology . acute schistosomiasis and other parasites were ruled out as there was no eosinophilia or elevated total - ige in either patient . different centers were asked to assist with this case , including unit des rickettsies , france , porton down , uk and center for disease control and prevention ( cdc ) , united states . see table 3 , table 4 for list of all the test and results found on the patients . cytomegalovirus and epstein barr virus were also considered as they are a common cause to fever of unknown origin in adults . hiv and other sexually transmitted diseases were also screened for and these results were also negative . the patients were not tested for histoplasmosis as this rarely causes a prolonged course of disease in immunocompetent patients . throughout the long period of symptoms with this couple it became more and more apparent that it was not an infectious agent at play . the attacks were short lived and the crp and leukocytosis normalized within a few days . malignancy and inflammatory diseases were also ruled out as the cause of the recurrent fever . both patients had a slight dry cough and small , non - tender , lymph glands at several stations . one would expect the patients to react differently to the same infectious agent as their immune systems are different as they differ genetically . we , therefore , started searching for an agent they could both be exposed to in the home . all the symptoms seemed to occur during autumn , winter and spring during which the windows are closed . it is therefore believed to be repeated exposure to aroma oils ( nature and decouvertes ) that is the cause of the symptoms . since the exposure has stopped there have been no more events . for ethical reasons it has not been possible to test with expose . the oils used here were nebulized and we assume that the two patients inhaled a high concentration of the nebulized oils over a short time . this was actually recommended by the manufacturer and the oils and table top nebulizer was purchased together . as mentioned earlier , we are not aware of reports of any similar events after using other similar products . the investigational program was build up over time as new attacks continued to occur and suggestions from different centers which were consulted were followed up . the number of tests performed at different laboratories took time and emphasizes that a panel of analysis in returning travelers with continued symptoms but without a clear diagnosis should be developed . written informed consent was obtained from the patient for publication of this case report and accompanying images . a copy of the written consent is available for review by the editor - in - chief of this journal on request .

fever in returning travelers is a common problem and usually the diagnosis is made within a few days or the traveler recovers.we present two travelers who presented with fever two weeks after returning from a six week vacation in south america . over the following 18 months they presented with short attacks of fever , elevated crp and leukocytosis and the program for investigation became more and more elaborate . a curious and key feature was , that they were completely synchronous both developing symptoms within an hour and presentation with the same laboratory findings of leukocytosis and elevated crp . extensive and repeated tests were performed , at our facility and abroad . after a year it was discovered that the uses of aroma oils were associated with the symptoms . no similar case has been found to be reported previously.these cases emphasize that natural products are not inherently safe . the investigational program was build up over time as new attacks continued to occur and suggestions from different centers which were consulted were followed up . the number of tests performed at different laboratories took an extensive amount of time . these cases emphasize that a panel of analysis in returning travelers in which no clear diagnosis is found should be developed .

primary lymphoma of the parotid gland is an uncommon entity . predominantly ( 84 - 97% ) parotid lymphomas are non - hodgkin lymphomas ( nhl ) and most of them are of b - cell origin . there may be coexisting lymphadenopathy of the cervical region . however , to categorize them as a primary from the parotid , the first clinical manifestation should arise in the parotid gland . we report a case of primary nhl of the parotid gland in a 61-year - old male that was diagnosed on fine needle aspiration cytology ( fnac ) with further histological confirmation . a 61-year - old male presented with a painless swelling in the right parotid region for the last 1 year and right sided cervical swelling for the last 3 months . on examination , a mass of 5 cm 5 cm was identified in the parotid region . in addition , the patient had an enlarged right sided level v cervical lymph node . hematological investigations were within normal limits ( hemoglobin 11.8 gm / dl , total leukocyte count 6000/cumm and a differential count of 66% neutrophils , 28% lymphocytes , 4% eosinophils and 2% monocytes , platelet count 1.6 lakhs / cumm ) . his erythrocyte sedimentation rate was elevated with 58 mm at the end of the first hour . contrast - enhanced computed tomography ( cect ) revealed a lobulated mass of 7 cm 5 cm 2.5 cm arising from the deep lobe of the parotid . fnac was advised from both the parotid swelling and the enlarged right sided level v cervical lymph node . smears were moderately cellular and comprised of large atypical lymphoid cells with high n : c ratio , irregular nuclear contour , vesicular chromatin , and prominent nucleoli and scanty agranular cytoplasm . the background showed lymphoglandular bodies alongwith normal - appearing salivary gland acini and ducts [ figure 1a ] . subsequently , immunocytochemistry ( icc ) was performed on the papanicolaou - stained smears without destaining . these cells were positive for leukocyte common antigen ( lca ) , cluster of differentiation ( cd ) 20 ( inset ) and negative for cd3 and cytokeratin . in view of the icc findings , a diagnosis of high grade nhl favoring diffuse large b - cell lymphoma ( dlbcl ) was offered . ( a ) fnac of the parotid mass shows moderately cellular smears comprising of large atypical cells with scant cytoplasm , irregular nuclei , vesicular chromatin , and prominent nucleoli . background shows normalappearing salivary gland acini and ducts ( arrow ) ( mgg , 400 ) . inset : b - cell marker ( cd20 ) positivity of the tumor cells ( icc , 100 ) ( b ) cervical lymph node fnac shows similar cytomorphology ( mgg , 100 ) fnac from the cervical lymph node showed similar cytomorphology [ figure 1b ] . in view of the above findings , a diagnosis of high grade b - cell nhl favoring dlbcl involving the right parotid gland with secondary involvement of the level v cervical lymph node was made . excision biopsy of the cervical lymph node revealed diffuse effacement of the architecture with sheets of atypical lymphoid cells with vesicular chromatin and scanty cytoplasm . based on the above histological and ihc findings , a diagnosis of dlbcl was made . correlating with the clinical history and radiological findings , final diagnosis of a primary dlbcl of the right parotid gland with secondary involvement of level v cervical lymph node was made . the patient was treated with six cycles of rituximab - cyclophosphomide - hydroxydoxorubicin - oncovin - prednisolone ( r - chop ) chemotherapy . malignant lymphoma of the parotid gland is relatively rare and constitutes about 4 - 5% of extranodal lymphomas , and 1 - 4% of all parotid tumors . facial nerve paresis and associated cervical lymphadenopathy may be a feature as well . in the present case they may be associated with autoimmune diseases such as sjgren 's syndrome . in the present case , the lymphoma may originate from the intraparotid lymph nodes or from the parenchyma ( mucosa - associated lymphoid tissue [ malt ] ) or both . in view of this , lymphoma primarily affecting the parotid gland to refer to lymphoma affecting the parotid region . the differentials of malt lymphoma are lymphoepithelial sialadenitis ( lesa ) and warthin 's tumor . the cytology of lesa reveals a mixture of acinar cells , epithelial , and myoepithelial cells admixed with polymorphous lymphoid cell population . warthin 's tumor is considered a benign primary parotid gland neoplasm that is composed of a mixture of oncocytic cells , basal cells , and stroma that contains numerous lymphocytes usually arranged in papillary and cystic structures . by contrast , the diagnosis of non - malt high grade lymphomas is usually straightforward , as they have overt cytological atypia . the criteria for primary parotid lymphoma , as suggested by hyman and wolff , include the first clinical manifestation in the parotid gland , histologically involving the parotid gland parenchyma and malignant nature of the lymphoid infiltrate . in the present case , the parotid swelling developed earlier than the cervical lymph node and the parenchyma was infiltrated by malignant lymphoid cells , meeting all the criteria as suggested by hyman and wolf for primary lymphoma of the parotid . lymphoma of the salivary gland is highly chemo - radiosensitive and , therefore , a timely diagnosis is important . moreover , surgical management of parotid lymphoma patients carries a high risk of morbidity due to infiltrative nature of the neoplasm . the role of fnac thus becomes important in such a setting for an early definitive diagnosis .

primary lymphoma of the parotid gland is relatively rare and constitutes about 4 - 5% of extranodal lymphomas . the majority of them is non - hodgkin lymphoma ( nhl ) and is b cell in nature . we report a case of primary diffuse large b - cell lymphoma ( dlbcl ) of the parotid gland in an elderly male . the case was diagnosed on fine needle aspiration cytology ( fnac ) of the right parotid gland as high grade b - cell nhl and confirmed on histopathology as dlbcl . in correlation with the clinicoradiological findings , the case was diagnosed as primary parotid dlbcl . the case highlights the role of fnac as a timely and useful diagnostic tool .

for decades metal ceramic restorations have been considered the gold standard treatment for the fabrication of prosthetic crowns and fixed partial dentures ( fpds ) . ceramic materials with different processing routes were developed to play this role without the inherent disadvantage of a metal framework . at first , the main drawback with the use of all - ceramic crowns was bulk fractures due to the brittle nature of ceramics and the lower mechanical properties when compared with metal frameworks . the advent of computer - aided design / computer - aided manufacturing ( cad / cam ) technology allowed the possibility of working with high strength polycrystalline materials , shifting the problem to the veneering ceramics . this seems reasonable since the veneering ceramics present lower flexural strength ( 90 - 120 mpa ) compared with the yttria - stabilized tetragonal zirconia polycristals substructure ( 900 - 1200 mpa ) . this way , the use of glass - ceramic ingots for pressing veneering ceramics onto zirconia frameworks was proposed to reduce the prevalence of veneer chippings / fractures . however , attempts to improve the microstructure and mechanical properties of veneering ceramics did not result in increased reliability . in addition , identical chipping failure patterns were observed . despite the numerous advantages of cad / cam technology , it seems that at some point clinicians and laboratory technicians started paying less attention to basic principles of substructure design in fixed prosthodontics . this article describes an alternative technique for the customization of cad / cam frameworks through a dual - scan process . this procedure provides adequate porcelain support and thickness in a predictable manner with little additional effort and cost to both clinicians and laboratories . a 46-year - old woman , presented with a carious lesion under a metal onlay restoration on tooth number 16 requesting an esthetic restoration . she was advised of the available metal ceramic and all - ceramic options before selecting a zirconia - based all - ceramic crown . first , a full - contour waxing of the final restoration was made to guide all clinical and laboratory steps . the other uncut was used to fabricate the temporary restoration with bis - acrylic resin ( protemp 4 , 3 m espe , st . tooth preparation was carried out leaving 1.5 mm of space for the final restoration in the axial walls and 2.0 mm in the occlusal area [ figure 1 ] . the temporary restoration corresponded to the patient and dentist 's expectations functionally , biologically and esthetically . commonly cad / cam systems deliver a uniform substructure ranging from 0.3 mm to 0.6 mm in thickness . the software provides uniform substructure width , but the restorations end up with an extensive and non - uniform space for porcelain application over the zirconia framework , being more prone to chipping and cracking . in the technique described in this article , waxing cut back was performed to obtain uniform and adequate porcelain thickness [ figure 2 ] . this way , substructure thickness is not uniform , but the zirconia substructure provides support for the overlying porcelain . the only difference to a metal ceramic substructure is that there is no palatal / lingual collar since zirconia exposure to moist environments may be detrimental to its performance . hence , what needs uniformity in thickness is the porcelain and not the zirconia substructure . a dual - scan procedure was used to combine the datasets from the die with and without the waxed substructure . then , the wax pattern was sealed to the die , which was scanned a second time . the scanner ( cercon eye ; dentsply usa ) used the sub marginal data points to orient the two scans and merge the data sets . it works as a subtraction of the images : the die with the waxed substructure minus the die with the tooth preparation . the result of this image subtraction is the personified substructure for that specific case [ figure 3 ] . the merged file was transmitted to a milling facility and the coping was manufactured [ figure 4 ] . the porcelain veneering ( cercon ceram , dentsply , usa ) was completed [ figure 5 ] and the crown cemented ( relyx unicem , 3 m espe , st . paul , mn , usa ) after surface treatment with sandblasting and alloy primer application . this procedure works as if a subtraction of the images is made : the die with the waxed substructure minus the die with the tooth preparation . and the result of this image subtraction is the customized framework for that specific case ready to be milled from zirconia blocks milled zirconia substructure identical to the waxed substructure ceramic crown after porcelain application the chipping problem is the most frequent reason for failures in all - ceramic zirconia restorations , irrespective of the zirconia veneer system applied . veneer fracture rates are reported at 2 - 9% for single crowns after 2 - 3 years and at 3 - 36% for fpds after 1 - 5 years . implant - supported zirconia - based restorations revealed even higher rates at 8% for single crowns after 6 months and at 53% for fdps after 1 year . impaired proprioception and rigidity of osseointegrated implants associated with higher functional loads might further aggravate cohesive veneer fractures . persuasive literature exists pointing to thermal processing problems as a possible cause of residual stress and defects ( too rapid heating and cooling given the very low thermal conductivity of zirconia ) . however , chipping seems to be a phenomena not limited to zirconia restorations and also related to the design of the substructure . the design of the substructure especially with the launch of cad / cam generated zirconia restorations provided no support for the veneering ceramic . this lack of porcelain support may have contributed to the actual high numbers of chipping on fpds as opposed to failures on the interface between substructure and veneering alone as supposed earlier . the lack of a uniform layer of the veneering ceramic because of improper framework design has been discussed as a possible reason for chipping fractures . few clinical data on optimal design of zirconia - supported restorations have been published . with the introduction of cad / cam technologies in dentistry , excessive veneer layer thickness ( > 2.5 mm ) was created because of the uniform layer thickness of the copings for crowns and bar - shaped connectors for fdps . improved customized zirconia coping design derived from the conventional porcelain fused to metal technique has been recommended to provide adequate support for the veneering ceramic . a dual - scan procedure of the die and full - contour wax pattern has been merged to customize the desired framework . preliminary in vitro studies showed that cohesive fractures within the veneering ceramic could not be avoided with the improved support , but the size of the fractures decreased significantly and failure initiated at higher loads . hence , the effect of framework design modifications on residual stress states needs to be better elucidated . it allows for the individualization of the substructures in a predictable manner and possibly improves longevity of all - ceramic crowns since core and porcelain thicknesses can be controlled . this technique needs to be further studied in a controlled clinical trial to determine the effectiveness of substructure modification .

the use of bi - layered all - ceramic crowns has continuously grown since the introduction of computer - aided design / computer - aided manufacturing ( cad / cam ) zirconia cores . unfortunately , despite the outstanding mechanical properties of zirconia , problems related to porcelain cracking or chipping remain . one of the reasons for this is that ceramic copings are usually milled to uniform thicknesses of 0.3 - 0.6 mm around the whole tooth preparation . this may not provide uniform thickness or appropriate support for the veneering porcelain . to prevent these problems , the dual - scan technique demonstrates an alternative that allows the restorative team to customize zirconia cad / cam frameworks with adequate porcelain thickness and support in a simple manner .

a 53-year - old caucasian male was evaluated for bright red blood per rectum with colonoscopy . the patient 's past medical history was significant for end - stage liver disease due to hepatitis c and hepatitis b , asthma , and erythropoietic porphyria . the patient had prior episodes of hematemesis which required multiple sessions of esophageal variceal rubber band ligation treatment . the patient complained of bright red blood per rectum for a four month period without clinical evidence of active or massive bleeding . family history was notable for colon cancer in the patient 's father at age 60 . his medications included propanolol , lasix , aldactone , lactulose , thiamine , and folate . pertinent review of symptoms was negative for fevers , chills , weight loss , diarrhea , constipation , melena , weakness , or loss of consciousness . physical examination revealed a well - developed , well - nourished male in no acute distress appearing his stated age . vitals signs were as follows : blood pressure 108/58 mm hg , heart rate 66/min , respiratory rate 18/min , and temperature 36.9c . in addition , abdominal examination revealed a 2 2 cm umbilical hernia and a right - sided inguinal hernia . recent complete blood count prior to colonoscopy revealed a hemoglobin of 11.5 g / dl , hematocrit of 34.6% , and a platelet count of 36,000 with a mcv of 81.3 . the patient had positive serologies proving both infection with hepatitis b and c. the patient 's hepatitis c viral load was undetectable . recent abdominal ultrasound was notable for hepatomegaly , nodular and heterogeneous in consistency , massive splenomegaly , and ascites . colonoscopy was significant for internal hemorrhoids as well as cecal and proximal ascending colon varices without any endoscopic evidence of bleeding ( fig . reported cases of varices have been found in the stomach , small bowel , biliary tree , colon , rectum , and even the site of a surgical ostomy . the stomach and the rectum are the most common sites of extraesophageal varices and up to 30% of variceal bleeding may originate from an extraesophageal location . a history of abdominal surgery may predispose a patient to develop varices of the lower gastrointestinal tract [ 5 , 6 ] . other causes include biliary atresia , biliary sclerosis , congestive heart failure , superior mesenteric vein thrombosis , and inferior mesenteric vein thrombosis . a smaller number are of idiopathic origin . due to the patient 's history of esophageal varices as a result of portal hypertensive gastropathy this case of cecal and ascending colon varices is presented because of the uniqueness of this anomaly within the colon and most notably the cecum . in the literature , a total of seventeen cases ( including this one ) of cecal varices have been reported [ 2 , 5 , 6 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 ] . the cases of cecal varices that have been reported have all presented clinically with massive lower gastrointestinal bleeding necessitating urgent management with blood products and surgical or endoscopic intervention . to our knowledge this is the first case of cecal varices reported that has not resulted in massive lower gastrointestinal bleeding . he did not demonstrate any episodes of massive gastrointestinal bleeding requiring immediate intervention as in the previous cases reported . given the colonoscopic finding , we believe that the internal hemorrhoids and not the cecal varices may have contributed to his clinical presentation . by reporting our experience we emphasize that cecal varices are very rare and may also be present in stable patients without massive lower gastrointestinal bleeding . because of the paucity of data , the proper management of patients with colonic varices is unknown . treatment of cecal varices has included colectomy as well as transjugular portal - systemic shunt in cases of varices attributed to portal hypertension . there are no established guidelines for the treatment of extraesophageal varices due to the scarcity of cases reported . he is clinically stable and has no clinical evidence indicating a history of active bleeding from those varices . however , further study is needed to determine whether sclerotherapy , band ligation , or even surgical treatment is effective for bleeding associated with cecal varices . careful patient follow - up should also be instituted for those who are incidentally noted to have cecal varices but have not bled .

since the original description of colonic varices in 1954 [ n engl j med 1954;250:434 - 438 ] , fewer than 100 cases have been reported in the literature . particularly , even fewer cases of cecal varices have been reported . more than 75% of these cases have been due to portal hypertension . our objective is to contribute a rare case with an uncommon presentation to the medical literature . we present the case of a 53-year - old male with hepatitis c and hepatitis b liver cirrhosis who presented for outpatient colonoscopy . the indication for colonoscopy was bright red blood per rectum and iron deficiency anemia . a significant amount of varices were noted in the cecum and proximal ascending colon . no endoscopic evidence of colonic bleeding was noted . this is the first reported case of cecal varices not presenting with massive lower gastrointestinal bleeding .

twenty - eight post mortem lower eyelids ( 14 right , 14 left ) from 16 japanese cadavers were examined and divided into 2 groups . one group of 14 lower eyelids of 7 cadavers ( 7 right , 7 left ; 3 males , 4 females ) with an average age at death of 80.4 years ( range : 73~97 ) , were sagittally incised from the lower lacrimal punctum ( figure 1 ) . the other 14 lower eyelids of 9 cadavers ( 7 right , 7 left ; 4 males , 5 females ) with an average age at death of 76.3 years ( range : 69~95 ) were incised from the lower lacrimal punctum with 5 degrees lateral inclination to the sagittal plane ( figure 1 ) . all cadavers were registered in the cadaveric service of aichi medical university , with proper consent from our institutional review board and approval to use them for educational purposes and studies . all methods for securing human tissue were humane and complied with the tenets of the declaration of helsinki . in the first group , which underwent a strict sagittal incision , 10 canaliculi of 7 cadavers ( 6 right , 4 left ; male 5 eyelids , female 5 eyelids ) were interrupted at the halfway point of the vertical portion and so the distal part of the canaliculi were not included ( figures 2a , b ) . however , 4 canaliculi of 4 cadavers ( 1 right , 3 left ; male 1 eyelids , female 3 eyelids ) did include the whole length of the vertical portion . in the second group with 5 degrees laterally inclined incision , all specimens included the whole length of the vertical portion ( figures 3a , b ) . most vertical portions of the lower lacrimal canaliculus examined here demonstrated a laterally inclined course of approximately 5 degrees ( figure 4 ) . the course of the laterally directed vertical portion of the lacrimal canaliculus should be considered not only in lacrimal probing , syringing , and trephining but also in eyelid surgeries . the 5 degrees lateral inclination of the vertical portion significantly emphasizes the need of a laterally directed insertion and lateral lid traction during the passage of a lacrimal probe , syringing needle or trephine to accord with the true course of the vertical portion . as well , since the vertical portion takes its lateral course within 1 mm laterally from the lacrimal punctum ( whitnall 1979 ; hwang et al 2005 ) , surgeons , during eyelid surgery , need to recognize the area within 1 mm laterally from the punctum as being a zone with a potential for serious lacrimal canalicular injury . the vertical portion changes its direction medially because of the inward eyelid movement ( kakizaki et al 2005 ) caused by the contraction of horner s muscle . at this time , the closure of the vertical portion may become insufficient , because the relative angle of the muscle of riolan and the vertical portion of the lacrimal canaliculus becomes milder . to maintain the relative perpendicular angle to each other , although all the cadavers were in an eye - closed state , and the muscle tonus of the orbicularis oculi had been completely lost , this state would have been similar to the eye - opening state in live humans whose muscle tonus of the orbicularis oculi is little . as the lacrimal probing syringing and trephining is usually performed in eye - opening state , the present result is reasonable for the true clinical situations . first , as the study samples were all taken from elderly cadavers , we may not be able to apply the results to younger generations . confirming our results in a younger patient population through clinical procedures such as lacrimal probing is possible . our experience is that the lateral inclination of a probe usually enables easy lacrimal probing , even in young generations . second , as we did not examine samples of caucasians or blacks , we can not conclude that they have the same inclination . however , to date , racial variations have not been reported with regard to the lacrimal drainage system ; therefore , we may be able to speculate that they have the same inclination . in conclusion , most vertical portions of the lower lacrimal canaliculus are directed laterally at approximately 5 degrees . surgeons need to recognize the true course so as not to cause injury to the vertical portion of the lower lacrimal canaliculus . this anatomical view will be helpful for a better understanding of the lacrimal drainage system .

the nomenclature of each part of the lacrimal canaliculus , for example the vertical portion , does not always reflect the true course . since we have sometimes observed findings suggesting the so called vertical portion of the lower lacrimal canaliculus inclined laterally , we re - examined the course of the vertical portion . twenty - eight postmortem lower eyelids in 16 japanese were examined and divided into 2 groups . the first group was 14 lower eyelids of 7 cadavers . eyelids were incised sagittally from the lower lacrimal punctum . the second group was 14 lower eyelids of 9 cadavers ; these were incised from the lower lacrimal punctum with 5 degrees lateral inclination to the sagittal plane . in the first group , 10 canaliculi of 7 cadavers were interrupted at the halfway point of the vertical portion . four canaliculi of 4 cadavers included the whole length of the vertical portion . in the second group , all specimens included the whole length of the vertical portion . most vertical portions of the lower lacrimal canaliculus demonstrated a laterally inclined course of approximately 5 degrees , although some took a completely vertical course .

isolated nccm was first described in 1984 , but it only regained recognition in the last decade . now , it increasingly attracts scientific attention , especially because the condition has not been fully understood so far and is thus a subject of ongoing investigations on its pathology , development , clinical course , and therapy ( 1 ) . nccm is a primary genetic cardiomyopathy , caused by a defect in endomyocardial morphogenesis . as a result , the muscle of the ventricle is built out of trabeculae with intratrabecular recesses , giving a typical appearance of muscular bands in a spongy mashwork. this specific morphology is reminiscent of the myocardium during early embryogenesis . between the 4th and 18th week of cardiogenesis , a non - compacted structure is essential for the nutrition of the cells , since the muscle is being supplied primarily by diffusion of blood that flows in the intratrabecular spaces . when the angiogenesis of coronary arteries is not accompanied by a simultaneous regression of trabeculae , nccm is occurring ( 2 ) . mostly , they encode for sarcomere and cytoskeleton proteins , such as tafazzin ( taz ) , lim domain binding protein 3 ( ldb3 ) , -dystrobrevin ( dtna ) , lamin a / c ( lmna ) , etc . the exact prevalence is not known and it is assumed to bear a significant number of undiagnosed cases . in echocardiography , it is found in 1 of 2,000 studies . since the diagnosis is defined by structural features , which can only be evaluated by cardiac imaging , mostly transthoracic echocardiography . a diagnosis can be made in the presence of three factors : 1 ) a thickened left ventricular wall consisting of two layers with a maximum ratio of non - compacted to compacted myocardium > 2:1 at end - systole in pax , 2 ) color doppler evidence of flow within the deep intertrabecular recesses , 3 ) prominent trabecular meshwork in the lv apex or midventricular segments of the inferior and lateral wall ( 3 ) . various criteria systems have been described ; however , they all disembogue in the structural features and quantifications . cardiac mri is an advanced option for gaining a closer motion insight of the myocardium . it is especially useful if the morphology is overlapped with other cardiopathies ( 4 ) , due to its enhanced spatial resolution , improved tissue characterization , and lack of ionizing radiation ( 5 ) . the disease may remain silent along the entire life or appear unspecifically , depending to what extent the heart function is affected : dyspnea , fatigue , limbs edema , limited physical capacity , and exercise intolerance . furthermore , tachycardia has recently been found associated with nccm , leading to hypotension . syncopies have not been mentioned so far in possible signs of the disease but are plausible since it is a result of cerebral hypoperfusion . in advanced stages , the prognosis is difficult to predict and has to be individualized , but it can be geared to the resulting cardiac impairment grade . since there is no specific treatment so far , the essential approach is the early recognition of any cardiac dysfunction , prevention of complications and symptom - based therapy , including ace inhibitors , beta - blockers , and aspirin . more aggressive treatments such as surgical interventions have been reported in severe nccm cases ( 6 ) . in our case , the symptoms were still limited to situations where our patient is physically challenged . thus , she was advised to forego intensive sports and advised to have regular check - ups with a cardiologist ( including loop recording and tte ) . patient 's children have been advised to undergo a cardiologic check - up as well . its molecular genetic basis is not yet fully clear , and the same is true of its diagnosis , treatment , and prognosis . the author has not received any funding or benefits from industry or elsewhere to conduct this study .

a 41-year - old female was brought to the er after a presyncope and absence episode while riding a bike . she recalled no prodromi . her physical and neurological examinations were unremarkable ; past medical , family , substance , and travel history bland . she reported a constant tiredness , sporadic slight cephalgia , responsive to acetaminophen , and a recent syncope while jogging , resulting in a fall , circumstances of which she could not recall . non - compaction cardiomyopathy is a type of cardiomyopathy that was first described 25 years ago . its molecular genetic basis is not yet fully clear , and the same is true of its diagnosis , treatment , and prognosis . further study of these matters is needed .

cardiac perforation is a dreaded complication of transvenous pacemaker , cardiac resynchronization therapy defibrillator ( crt - d ) , and implantable cardioverter - defibrillator ( icd ) lead placement because of the potential for critical morbidity and mortality . we report the case of a 45-year - old woman affected by severe dilated cardiomyopathy who was admitted to our cardiology department for an upgrade of a previously implanted pacemaker . the patient received a vvi pacemaker implant for complete atrioventricular block 22 years earlier and an upgrade ( ddd mode ) 11 years later . leads were inserted through the left subclavian vein : ( a ) an active fixation double catheter in the right ventricle , ( b ) a bipolar active fixation lead in the right auricle ( fig . 1 ) , and ( c ) a bipolar lead introduced from the coronary sinus into the antero - lateral vein . the previously implanted pacemaker and one atrial lead were removed ( the old unipolar lead was left in the right ventricle ) and a crt device was connected to the new implanted leads . after satisfactory r - wave sensing ( > 5 mv ) and pacing thresholds ( < 1.0 v at 0.5-ms pulse width ) had been demonstrated , the patient underwent defibrillation threshold testing ( dft ) to ensure proper device function . sustained ventricular tachycardia and ventricular fibrillation were induced to make certain that the device was able to constantly sense , detect , and terminate arrhythmias with a shock at 25 j. during skin closure , the patient went into cardiac arrest with pulseless electrical activity . cardiopulmonary resuscitation maneuvers were performed immediately and echocardiography showed intrapericardial effusion that was partially drained . after 1 h , pulseless electrical activity persisted , the patient was declared dead , and resuscitation attempts were halted . the unexpected fatal outcome resulted in an allegation of medical negligence against the operating cardiologists . a board of physicians consisting of a forensic doctor and a heart surgeon conducted a post - mortem examination and analysis of the medical records . at autopsy , approximately 180 cm of blood and clots were found inside the pericardial cavity . careful inspection of the aorta showed a deep lesion , 2 mm in length , on the antero - lateral aspect of the vessel ( fig . 1 ) 2 cm above the valvular plane ( fig . the lead metal extremity was implanted at the base of the auricle on the medial wall . the right atrium was very thin , with an average thickness of 2 mm . histology showed abundant adipose tissue in its context , endocardial fibrosis , and numerous foci of inflammatory infiltration by various elements including lymphocytes and monocytes as well numerous plasma cells and eosinophils , clearly indicating myocarditis that was not recent . the ascending aorta was incised and approximately 2 cm above the noncoronary cusp , a pinpoint injury related to the lesion described on the aortic adventia ( fig . the pathogenetic mechanism underlying this patients death can be explained in the following manner : the active lead ( fig . 1 ) , positioned in the right atrium medial aspect , perforated the thin atrial wall and the lateral aspect of the aortic wall . after aortic perforation , the lead initially remained inside its wall , which explains why the device interrogation revealed satisfactory r - wave sensing ( > 5 mv ) and pacing thresholds ( < 1.0 v at 0.5-ms pulse width ) . the patient eventually underwent dft to guarantee proper device function . at this point , the active lead , positioned inside the aorta like a cork , slipped out , causing cardiac tamponade . in a review of relevant literature , we identified only five cases , , , , of atrial damage by a pacemaker lead with concomitant aortic wall perforation , as shown in table 1 . however , not all reports indicate whether the lead responsible for the cardiac laceration was active or passive . a board of physicians conducted a post - mortem examination and their findings did not support a direct allegation of medical negligence against the operating cardiologists who performed the icd implant . the board analyzed the guidelines for cardiac pacing and cardiac resynchronization therapy and considered the indications for the procedure performed on the patient to be correct . the increased heart size shifted the right atrial appendage closer to the adjacent lateral aspect of the aorta . at the same time , the remarkable thinness of the right atrium was conducive to wall perforation . the perforation was not necessarily a result of malpractice in electrode positioning , but was also caused by other factors such as the pressure exerted during cardiac contraction . moreover , since she was bearing a pacemaker , the patient could not undergo magnetic resonance imaging before the procedure to highlight the thicknesses of the cardiac wall . in addition , her pre - existing myocarditis undoubtedly reduced the chances of successful resuscitation . antonino m. grande , affirms on behalf of all co - authors that we have no conflicts of interest in connection with this article .

a 45-year - old woman with dilated cardiomyopathy was admitted for the upgrade of a previously implanted pacemaker . echocardiography showed intraventricular dyssynchrony and a low ejection fraction ( 0.35 ) . treatment with a cardiac resynchronization therapy defibrillator ( crt - d ) was selected and the device was implanted . crt - d interrogation revealed proper function . following procedure termination , the patient went into cardiac arrest and died despite resuscitation attempts . an autopsy revealed that the medial aspect of the right atrium was pierced by an active lead and that the aorta had a deep lesion , 2 mm in length , on its lateral aspect . we explain the probable pathogenesis of this patients death .

this injury is commonly associated with multiple lesions such as acetabular fractures , nerve palsy and femoral head fractures , due to its high energy trauma mechanism . bilateral hip dislocation is an extremely rare finding , with very few cases reported in literature [ 2 , 3 ] . most are related to acetabular or proximal femoral fractures , consisting of complex lesions , and are rarely are pure ligamentous injuries . in the diagnosis of a dislocated hip , prompt reduction must follow to prevent future complications like avascular necrosis ( avn ) [ 1 , 3 ] . the following case report consists in the presentation of a traumatic bilateral posterior hip dislocation without associated injuries . the patient is a 23-year - old female , with no comorbidities , who sustained bilateral hip pain after an automobile accident . at the time of the accident patient was driving her vehicle and the accident happened while the patient was driving and had her hips and knees in a flexed position , thus transmitting loads to the posterior aspect of the hips and forcing dislocation . only mild head trauma and severe hip pain was reported by paramedics at the time she was primarily attended . she was referred to the emergency room unable to move her legs and had slight hip flexion with internal rotation . physical examination revealed 20 hip flexion with 30 internal rotation , sensitivity and muscular strength were intact , pain was intensified when passive or active limb motion was attempted . antero - posterior radiographs were taken , diagnosing bilateral posterior hip dislocation with no evidence of associated fractures ( fig . 1 ) . computed tomography was realized to evaluate fractures and articular fragments and none were found ( fig . an hour after initial evaluation , the patient was admitted to the operating room , under general anesthesia , and closed reduction of both hips was performed without incidents . stability and range of motion was assessed , with normal findings ; control x - rays showed concentric hip reduction of both femoral heads ( fig . physical therapy was initiated the first week with quadriceps and gluteal muscles isometric exercises and passive hip movements . at second week after 6 weeks follow - up the patient achieved a complete hip range of motion and walked without assistance . figure1 : patient with hip flexion and internal rotation and x - ray with traumatic bilateral posterior hip dislocation.figure 2:ct - scan show bilateral posterior hip dislocation without evidence of fracture or articular fragments.figure 3:after closed reduction - hip abduction and external rotation . 1 : patient with hip flexion and internal rotation and x - ray with traumatic bilateral posterior hip dislocation . ct - scan show bilateral posterior hip dislocation without evidence of fracture or articular fragments . after closed reduction - hip abduction and external rotation . the classification of traumatic hip dislocation is based of dislocation and the degree of joint disruption including any associated joint fracture . three commonly used classifications systems are : the epstein classification of anterior hip dislocation , pipkin sub - classification an the thompson - epstein classification of posterior dislocation as in this case report . simple injuries are dislocations without associated fractures , whereas complex injuries include either acetabular or proximal femoral fractures . posterior dislocations represent the majority of these injuries , occurring due to an axial load of the femur , with the hip flexed and adducted . usually , the axial load is transmitted through a flexed knee , as in dashboard injuries [ 3 , 5 , 6 ] . anterior dislocations , in contrast , occur with the hip in abduction and external rotation , and the position of the femoral head can be either inferior or superior to the acetabulum . motor vehicle accidents are the leading cause of hip dislocations , followed by falls from significant heights ; sports related injuries have also been reported in literature [ 6 , 7 ] . bilateral simultaneous hip dislocations are an extremely rare injury , a reported 1.25% of all hip dislocations [ 3 , 68 ] . these injuries are rarely present as isolated dislocations , and often associate fractures , usually of the acetabulum . these acetabular fractures frequently relate to the type of dislocation , posterior dislocation with posterior wall and column fractures and anterior dislocation with anterior wall fractures [ 1 , 5 , 7 ] . a patient with a dislocated hip usually presents acute pain and evident deformity , with an inability to tolerate active or passive range of motion . of all , 95% of these patients have associated injuries , and for this reason an adequate trauma emergency evaluation must be realized in the moment of arrival , following the atls sequence of patient evaluation . a ct scan is also important , presence of intra - articular fragments needs to be discarded ; this imaging also functions as means of postoperative follow up [ 3 , 7 , 8 ] . soft tissue injuries such as avulsions of the labrum may be assessed by magnetic resonance imaging , but are not requested routinely . heterotopic calcifications , avascular necrosis and osteoarthritis can also be evaluated with magnetic resonance imaging as part of the patient follow up . treatment of hip dislocations can be either operative or nonoperative , but emergent reduction of the joint is of upmost importance . delay in reduction of dislocated hip results in avascular necrosis in around 26% of patients . early mobilization results in a prompt return to weight bearing , but the outcome and relationship with complications such as avascular necrosis is largely debated . closed reduction of a posteriorly dislocated hip implies general anesthesia and an adequate positioning of the patient . several techniques have been described , all of which include traction and counter - traction as part of the maneuver . post reduction imaging studies must be performed to assess the quality of the reduction and presence of associated injuries that may have been neglected initially . open reduction is performed in the case of an irreducible dislocation , a delayed presentation or articular fragments that make a concentric reduction impossible .

abstractbilateral traumatic hip dislocations are extremely rare . most of these are related to acetabular or proximal femoral fractures , consisting of complex lesions , and are rarely pure ligamentous injuries . posterior dislocation is the most frequent . some dislocations are accompanied by sciatic nerve palsy . the present case is a posterior bilateral hip dislocation with no other associated lesions , there are very few reports published with this clinical setting . the patient had good functional outcome .

the mitral valve is located far from midline sternotomy , but is very close to the interatrial groove . as our experience grew and we learned from embryology , we found that a more cental left atriotomy was advantageous , and possible by interatrial groove dissection using ultrasonic scalpel . under general anesthesia , midline sternotomy is performed and the ascending aorta is cannulated for arterial inflow , and the superior vena cava and right atrial appendage are cannulated for venous outflow . the fad pad covering the interatrial groove is dissected under cardiopulmonary bypass by ultrasonic scalpel . the dissection is carried out in the exact groove of sondergaard until accessing beneath the thin atrial septum with careful attention paid to the coronary sinus vein ( fig . the aorta is cross - clamped and a vertical incision is made onto the left atrium , leaving enough room for the atrial wall to close . using a self - retaining retractor , atrial tissue above the anterior mitral annulus this makes a tractile and stretched wide view of the mitral valve possible because the left atrium wall hook - up is optimal . this is quite different from the right side left atriotomy , which usually leaves a redundant left atrial wall to be hooked up . because the mitral valve is such a complex anatomic structure and the maneuvers involved in correcting a regurgitant valve may vary from the simple to the very complex , adequate exposure is an absolute requirement in every operative plan . the first critical step to standard valve repair is the complete and thorough development of the sondergaard plane reflecting the right atrium off the left atrium to the atrial septum . this was first described in the 1950s by the danish surgeon sondergaard to expose the atrial septum for noncardiopulmonary bypass treatment of atrial septal defects . regardless of claims from previous procedures , it should always be possible to dissect out the groove without significant difficulty . the complete and full development of the groove is crucial for obtaining adequate exposure of the mitral valve . with this technique , via blunt and sharp dissection with ultrasonic scalpel , we had no need for any other incision for mitral valve repair or replacement , whether for primary surgery or reoperation . this incision brings the surgeon very close to the mitral valve even in the most difficult anatomic situations . once the right atrium is dissected off the left atrium , a generous incision in the left atrium is made , avoiding the atrial septum . we have been adopting this incision and approach in 10 consecutive patients and have found that the aortic cross - clamp time decreased significantly from an average 75 minutes to 48 minutes compared to right - side left atriotomy patients . a more central incision onto the left atrium

abstractexposure of the mitral valve is of critical importance in mitral valve surgery . the mitral valve is located near the interatrial groove . as we accumulated experience and learned from embryology , we found that a more central left atriotomy was advantageous , and was made possible by interatrial groove dissection using an ultrasonic scalpel . this crucial finding prompted us to report the technique .

sertoliform endometrioid carcinoma of the ovary ( sec ) is an uncommon variant that bears histologic similarity to sertoli and sertoli - leydig cell tumors ( slts ) . recognition of this tumor is important as it is a well - differentiated , low - grade malignancy that displays a good prognosis when confined to the ovary . a 55-year - old postmenopausal female patient presented with mass per abdomen since 2 years and pain for 2 days . ultrasonography showed multicystic lesion measuring 12 cm 11 cm in the left ovary [ figure 1 ] . ultrasonography showing multicystic lesion measuring 12 cm 11 cm in the left ovary on gross examination , ovary weighed 550 g , measured 13 cm 11 cm 7 cm , showed solid and cystic areas on cut section [ figure 2 ] . microscopy showed round to solid tubules lined by pseudostratified columnar epithelium with elongated nuclei resembling sertoli tumor - like pattern along with conventional endometrioid tumor [ figures 35 ] . inset : outer surface showing blackish areas due to torsion microphotograph showing sertoliform endometrioid carcinoma showing tubules and tightly packed nests of tumor cells separated by fibrous stroma . individual tumor cells having vesicular nuclei , prominent nucleoli , and moderate cytoplasm ( h and e , 400 ) microphotograph showing foci of conventional endometrioid carcinoma ( h and e , 400 ) microphotograph showing foci of conventional endometrioid carcinoma and sertoli cell tumor - like pattern ( h and e , 200 ) immunohistochemistry ( ihc ) showed tumor cells strongly immunoreactive for epithelial membrane antigen ( ema ) , cytokeratin ( ck ) but negative for inhibin [ figures 68 ] , thus confirming the diagnosis of sec of left ovary . sertoliform endometrioid carcinoma showing cytokeratin positivity ( immunohistochemistry , 200 ) sertoliform endometrioid carcinoma showing epithelial membrane antigen positivity ( immunohistochemistry , 400 ) sertoliform endometrioid carcinoma showing inhibin negativity ( immunohistochemistry , 400 ) endometrioid carcinoma of ovary resembling sex cord - stromal tumor is a rare variant of endometrioid adenocarcinoma that focally looks like a sex cord - stromal tumor with sertoli , leydig , or granulosa cells . ordi et al . , in their study , reported that sec typically demonstrates the following characteristics , while slts do not : ( 1 ) presence of areas with usual pattern of endometrioid carcinoma , ( 2 ) presence of mucin at the apical borders of the tumor cells . other factors that may favor sec would include squamous or squamoid areas , well - developed cilia , the presence of endometriosis , or a concomitant adenocarcinoma of the endometrium . immunostains for alpha - inhibin is positive in most neoplastic sertoli cells but negative in the cells of endometrioid carcinoma , while positive ema and ck immunostains favor sec . despite histologic similarity misir and sur in their study report that in slts , the patient age tends to be younger , with an average age of 25 years and clinically , up to 50% of slt patients may exhibit endocrine manifestations . in contrast , sec occurs almost exclusively in postmenopausal women with an average age of 68 years . virilizing symptoms , though uncommon in endometrioid carcinoma , may be encountered in the sertoliform variant , thus compounding the diagnostic difficulty . in our case , usually , solid portion of endometrioid adenocarcinoma is considered as grade 3 according to the international federation of obstetrics and gynecologists grading system . sec should be considered as grade 1 ( well - differentiated ) , despite the presence of solid , sex cord - like proliferation as it carries good prognosis when confined to ovary . according to these suggestions , our case is graded as well - differentiated ( grade 1 ) based on the histological features of foci of conventional endometrioid carcinoma . the case is presented for its rarity . in view of relatively good prognosis of sec as compared to endometrioid tumor , identifying this variant by an extensive sampling of the specimen is mandatory .

sertoliform endometrioid carcinoma of the ovary ( sec ) is an uncommon variant that bears histologic similarity to sertoli and sertoli - leydig cell tumors . we report an interesting case of sec in a 55-year - old female with a left ovarian mass with torsion . histology revealed an sec , featuring foci of typical endometrioid carcinoma , and areas composed of uniform , small , hollow tubules lined by columnar cells with apical cytoplasm . tumor cells were strongly immunoreactive for ema and cytokeratin but negative for inhibin ; thus , confirming the diagnosis of sec ovary . recognition of this tumor is important as it is a well - differentiated , low - grade malignancy that displays good prognosis when confined to the ovary .

an 81-year - old diabetic male came to our hospital to seek medical attention for 2 weeks of productive cough , fever and chills , and a 10 kg weight loss . physical examination revealed a fatigued - looking man with mild respiratory distress . except for bilateral crackles in both lungs , more on the right side ; the remainder of the physical examination was unremarkable , including the oral cavity . complete blood count revealed mild anemia , however , the patient did not have leukopenia / neutropenia . a chest x - ray showed a right lung infiltrate ; thus , community - acquired pneumonia ( cap ) was suspected and the patient was admitted for treatment of cap . the patient was started on intravenous gatifloxacin 400 mg daily . however , the patient 's symptoms continued to deteriorate while on gatifloxacin . a chest computed tomography ( ct ) done on hospital day 3 , showed a right upper lobe cavitary lesion ( figure 1 ) . because of the cavitary lesion , pulmonary diseases more complicated than cap ( such as lung cancer or vasculitis involving the lung ) were suspected . the patient underwent a bronchoscopy on hospital day 4 , which was reported as unremarkable . the bronchial washing cytology examination did not reveal malignant cells , but it showed scattered elongated bacilli ( figure 2 ) . within 24 hours the bronchial washing culture grew the same gram - negative bacillus , methicillin - sensitive staphylococcus aureus , viridans streptococcus and group b streptococcus . the gram - negative bacillus was eventually identified as ls at the north dakota state laboratory ( figure 3 ) . blood cultures were drawn - once before starting antibiotics and three more times during treatment ; however , none of these cultures grew organisms . because of the clinical unresponsiveness to gatifloxacin , on hospital day 6 , the antibiotic was changed to intravenous vancomycin 1.5 g daily and piperacillin - tazobactam ( pip - tazo ) 3.375 g every 8 hours . figure 1computed tomography - scan of the chest shows a cavitary lesion in the right upper lobe . computed tomography - scan of the chest shows a cavitary lesion in the right upper lobe . magnification 400. papanicolaou stain of the bronchial washing reveals the elongated bacilli ( arrow ) . the pneumonia was suspected to be caused by one of the above gram positive cocci and the ls was considered an oral contaminant ; therefore , after a 9-day course of vancomycin and pip - tazo , vancomycin was continued but pip - tazo was discontinued on hospital day 15 . on hospital day 19 , after reconsidering ls a remotely possible pathogen of the pneumonia , we added oral amoxicillin 500 mg / clavulanate 125 mg every 8 hours to the vancomycin regimen . while on vancomycin and amoxicillin - clavulanate , the patient began to have spiked fever , rigors and increased dyspnea . on hospital day 23 , a chest radiograph indicated worsening of the pneumonia . pip - tazo was continued for 4 weeks , followed by 2 weeks of intravenous aqueous penicillin g 2 million units every 8 hours . the patient continued to do well and has had no recurrence of pneumonia to date . ls is an anaerobic gram - negative bacillus that resides in the oral cavity and usually does not cause disease . bacteremia , endocarditis , and hepatic abscess caused by leptotrichia buccalis have been reported in patients with neutropenia , hematologic malignancies and in bone marrow transplant recipients . mucosal disruptions such as oral mucositis , esophageal ulcers , or diverticulitis are possible risk factors for infected patients . by gram stain alone , ls can be confused with other oral commensals , such as the elongated gram negative fusobacteria sp , capnocytophaga sp or even with the gram positive lactobacillus sp as ls can sometimes be gram - variable . the ls in this case report was identified by gram stain and phenotypic analysis , but not by gene sequencing because the north dakota state laboratory was not equipped to perform the test . one case is cavitary pneumonia with bacteremia caused by l. buccalis in an immunocompromised host . the other case is a severe pneumonia caused by ls in an apparently immunocompetent male patient . our patient has type 2 diabetes mellitus , but he is not immunocompromised from other underlying diseases or from immunosuppressive agents ; therefore , i believe this is the first case report of cavitary pneumonia caused by ls in an immunocompetent patient . although the patient had good dental hygiene without dental caries , gingivitis , or oral mucosa lesions ; the oral cavity could still be the portal of entry in this patient 's pneumonia . pneumonia caused by ls could be underreported because ls is usually considered a normal oral flora and is difficult to identify . therefore ; if ls grows from bronchoalveolar lavage or lung tissue , a clinician should consider it one of the etiologic agents causing pneumonia . due to the rarity of human infection caused by ls , the duration of treatment for pneumonia caused by this bacterium is not clear . in our case , because the pneumonia recurred after we administered pip - tazo for only 9 days , we aggressively gave the patient 4 weeks of piptazo followed by 2 weeks of intravenous aqueous penicillin g. although there are no clinical and laboratory standards institute ( clsi ) breakpoints for ls , penicillins are considered the drugs of choice . thus , the patient continued to be clinically stable while on either pip - tazo or penicillin g. we postulate that the pneumonia recurred while on amoxicillin - clavulanate was likely due to the oral agent at a subtherapeutic level in the lung tissue . therefore , we do not advocate treating pneumonia caused by ls with an oral -lactam antibiotic .

leptotrichia species ( ls ) is an anaerobic gram negative bacillus in the bacteroidaceae family and part of the normal human oral flora . it is rarely pathogenic , but occasionally causes diseases in immunocompromised hosts . i am reporting a case of cavitary pneumonia caused by ls in an immunocompetent host .

a woman in her 30s with acquired immunodeficiency syndrome ( aids ) diagnosed by the fourth - generation hiv testing , with a cd4 count of 3 ( 1% ) cells / mm , presented with weight loss and cachexia . she was started on lamivudine , zidovudine , and nevirapine and then developed hypersensitivity rash and hepatitis . at that time , she also had pancytopenia ( white blood cells 2,950/l , neutrophils 79% , lymphocytes 13% , hemoglobin 9.6 g / dl , hematocrit 29% , platelets 325,000 10/l ) and intra - abdominal lymphadenopathy . she was treated initially for presumptive disseminated mycobacterial infection with isoniazid , rifampicin , pyrazinamide , ethambutol , and clarithromycin . she improved clinically , and her antiretroviral regimen was restarted after three weeks with lamivudine , tenofovir , and efavirenz . later on , she had gained weight , and laboratory results showed the resolution of pancytopenia and hepatitis . two months later , the antibiotic regimen was readjusted to ciprofloxacin , clarithromycin , and streptomycin injection ( for one month ) according to the susceptibility , and her clinical improvement was remarkable . two months after the initiation of antiretroviral therapy ( art ) , she had an hiv viral load of < 40 copies / ml and a cd4 count of 1 ( 2% ) cell / mm ; however , she had recurrent transaminitis . she was given prednisolone 20 mg / day with a tapered dose for five weeks for the treatment of hepatitis possibly due to immune reconstitution inflammatory syndrome . five months after the initiation of art , her cd4 count was 13 ( 3% ) cells / mm , with an hiv viral load of < 40 copies / ml . she developed cytomegalovirus retinitis and received intravitreal ganciclovir injection and oral valganciclovir for six weeks . she then developed abdominal pain , with a palpable large suprapubic mass , for three weeks . a computed tomography ( ct ) scan of the abdomen showed a large lobulated mass ( 8.5 10 cm ) with the epicenter at the right adnexa and with the invasion of the uterus , right ovary , right lateral wall of the sigmoid colon , appendix , superior wall of the urinary bladder , and right distal ureter , causing right ureter obstruction and a moderate degree of right hydronephrosis . multiple matted lymph nodes were present along the mesenteric root and in the para - aortic , aortocaval , and retrocaval regions . focal circumferential wall thickening of the left side of the jejunum caused moderate intraluminal narrowing , with evidence of small bowel obstruction ( fig . she underwent exploratory laparotomy ; intraoperative findings revealed a large pelvic mass with nodular surface and with dense adhesion to the omentum , the anterolateral wall of the uterus , the bladder , and the sigmoid colon . only partial resection of the omentum and a biopsy of the mass at the anterior aspect of the uterus were performed . acid - fast and gomori methenamine stains of the tissue specimen were negative ; however , microbial culture was not sent for laboratory analysis . her hospital course was complicated by wound infection / dehiscence , with the formation of an enterocutaneous fistula . she suffered from abdominal pain as well as malnutrition due to the nature of the unresectable residual mass and the enterocutaneous fistula . she was discharged home on palliative care , with antimicrobial therapy , and was lost to follow - up . disseminated m. simiae infection presented with pancytopenia , hepatitis , and lymphadenopathy , similar to mycobacterium avium infection in an aids patient.1 effective antimicrobial treatment of m. simiae included rifampicin and ciprofloxacin . our patient had an abnormal host immune response to infection , malakoplakia , which occurred after m. simiae infection , a condition that has not been previously described in the literature . however , we were unable to demonstrate a direct correlation due to the lack of microbiological data from tissue specimens . malakoplakia has a gross appearance of round , oval , or mushroom - shaped yellowish structures.2 histologically , there is a proliferation of histiocytes with abundant granular eosinophilic cytoplasm ( known as von hansemann histiocytes ) that contain the pathognomonic michaelis gutmann bodies.2 these bodies are round to oval in shape and are visible as deeply basophilic structures , which are typically described as target - like and are periodic acid schiff stain - positive and calcium - positive.2 pathogenesis of malakoplakia is associated with the lysosomal dysfunction of macrophages in the intracellular killing process of ingested organisms and/or a defect in elimination , resulting in the accumulation of partially degraded bacteria within the cytoplasm and phagolysosomes of histiocytes , forming michaelis gutmann bodies around the undigested bacteria.2 the most common infectious etiologies described are bacteria , such as escherichia coli , rhodococcus equi , pasteurella multocida , and mycobacterium tuberculosis.211 malakoplakia often occurs in immunocompromised patients , such as those with aids , organ transplant recipients ( mostly solid organ transplants ) , and in one case a patient who had undergone stem cell transplantation.5,1114 malakoplakia affecting a major organ could result in various organ dysfunctions . one of the most serious adverse events reported was irreversible renal allograft dysfunction due to malakoplakia in a renal transplant recipient with e. coli infection , pyelonephritis , and bacteremia.14 in an allogeneic stem cell transplant recipient , pleural malakoplakia caused by r. equi , presenting with a large tumor mass , required a lobectomy.12 in patients with residual disease , successful results have been reported by treating the underlying infection , reducing immunosuppression , improving the bactericidal activity of monocytes with a cholinergic agonist , such as bethanechol , and administering a multivitamin supplement.4,15,16 this is the first report of malakoplakia occurring after m. simiae infection .

malakoplakia in an acquired immunodeficiency syndrome ( aids ) patient with disseminated mycobacterium simiae infection presented with a large pelvic mass that caused organ dysfunction from mimicking a tumor . malakoplakia is a rare , chronic granulomatous abnormal host response toward infectious agents , presenting as a tumor - like lesion . this is the first report of pelvic malakoplakia after disseminated m. simiae infection in an aids patient .

postoperative complications of blepharoplasty range from skin changes to vision - threatening emergencies.1 some of these complications occur early in the postoperative period , such as retrobulbar hemorrhage , infection , and eyelid hematoma . other complications occur later in the postoperative period , such as eyelid malposition , strabismus , scar and over- and under - resection of skin or orbital fat.1 there has been a case report of persistent lateral hooding after upper lid blepharoplasty that has been treated with lacrimal gland repositioning.2 to the best of our knowledge , we report the first case of lacrimal gland fistula after upper lid blepharoplasty . she had a history of repeated lid swelling for almost 3 years before the age of 10 years . the patient underwent bilateral blepharoptosis repair at 12 years of age , re - operation on the right lid at the age of 18 years , and laser resurfacing of periocular region at the age of 20 . on examination , the patient had bilateral blepharoptosis ( margin reflex distance of + 2.5 mm on the right and + 2 mm on the left side ) , dermatochalasis with mild lateral hooding , and medial and preaponeurotic fat protrusion . upper blepharoplasty included skin - orbicularis muscle flap excision , medial fat excision , and partially preaponeurotic fat excision through a hole in the center of the septum . she had an uneventful postoperative follow - up with the exception of lateral hooding of the right lid . an elliptical lateral hooding excision was performed 2 months after upper blepharoplasty . at 1 week post - hooding excision , there was wound dehiscence with clear watery drops discharging from the wound [ figure 1 ] . post - lateral hooding excision right upper eyelid wound dehiscence and watery drops show a fistulous tract from lacrimal gland to the wound the patient was scheduled for repair of the wound and repositioning of the lacrimal gland . there was a tract from the lacrimal gland to the skin of the upper eyelid at the site of wound dehiscence . the lacrimal gland was repositioned into the lacrimal gland fossa using 4 - 0 prolene suture . the postoperative course was uneventful out the last visit at 6 months after repair [ figure 2 ] . last follow up ( 6 months ) after repair of the wound and repositioning of the right prolapsed lacrimal gland despite the facile nature of the blepharoplasty , high patient expectations can make this procedure quite challenging to the surgeon . in order to attain a good result and avoid patient dissatisfaction , the surgeon must perform a careful history and physical exam and address specific patient complaints and expectations.3 blepharochalasis is a rare eyelid disorder characterized by exacerbation and remission of painless edema , eventually leading to atrophy of the periorbital skin . these episodes of eyelid swelling usually become less frequent with age , and eventually most cases enter a relatively quiescent stage . ptosis is a common finding in blepharochalasis ; however , the levator function is preserved.4 surgical management should be performed during the quiescent phase of blepharochalasis to avoid recurrent bouts of lid swelling leading to further ptosis and lid atrophy . surgeons advocate that blepharochalasis symptoms should be quiet for 6 - 12 months before surgical treatment is contemplated.4 our case did not report any exacerbation for at least 15 years prior to the recent operation . a prolapsed lacrimal gland occasionally occurs due to atrophic changes in the septum in patients with blepharochalasis . in cases of frank preoperative prolapsed lacrimal gland , a blepharoplasty and suspension of the lacrimal gland should be performed.4 our patient did not present with a prolapsed lacrimal gland preoperatively . she only had post - blepharoplasty lateral hooding on the right side without a palpable lacrimal gland . we assume that blepharoplasty and consequently right - side hooding excision pulled an undetected partially prolapsed lacrimal gland inferiorly into the wound and resulted in the formation of a fistula . a similar case has been reported with hooding after blepharoplasty due to lacrimal gland prolapse ; however there was no history of blepharochalasis or postoperative wound dehiscence and fistula.2 repositioning of the lacrimal gland to the right side was successful without postoperative sequelae out to 6 months postoperatively . the procedure can complex especially in the context of chronic inflammatory conditions.5 this case report illustrates an uncommon post - blepharoplasty complication in patients with blepharochalasis . surgeons who perform blepharoplasty should have a high index of suspicion for lacrimal gland herniation in the presence of lateral hooding specially in patients with a history of blepharochalasis .

to report the first case of lacrimal gland fistula after upper eyelid blepharoplasty for blepharochalasis . standard upper blepharoplasty and the hooding excision were performed in a female with blepharochalasis . the patient developed a fistulous tract with tearing from the incision few days after hooding excision . fistula excision and lacrimal gland repositioning were performed . there were no complications after the repositioning procedure ( 6 months follow up ) . prolapsed lacrimal gland and fistula formation can occur after upper blepharoplasty hooding excision .

cross sensitivity is defined as sensitivity to one substance that renders an individual sensitive to other substances of similar chemical structure . cross sensitivity has been reported commonly among various lactam antibiotics and sulfonamides . for the diagnosis of cross sensitivity , various methods like prick test , oral challenge test is considered as the most reliable . due to broad antimicrobial spectrum , frequently reported adverse reactions with fluroquinolones are nausea , abdominal pain , diarrhea , dizziness , restlessness , headache , depression and somnolence or insomnia . . immediate type of reactions are more common than delayed type of reactions . frequently reported immediate hypersensitive reactions are urticaria , anaphylactic shock , rashes and erythema . ciprofloxacin and norfloxacin are the most common drugs causing hypersensitivity reactions , while levofloxacin is the least common with incidence of 1 per million population . however , we could not find a case of immediate hypersensitivity reaction with levofloxacin or case report of cross sensitivity between ciprofloxacin and levofloxacin . here with , we report a case of cross sensitivity between ciprofloxacin and levofloxacin for immediate type of hypersensitive reaction . seven years old male child ( 24 kg weight ) admitted in paediatric ward , sir takhtsinhji general hospital , government medical college , bhavnagar , gujarat , india with complaints of fever , headache , vomiting and abdominal pain for 7 days . fever was of high grade , intermittent and associated with chills , which was followed by colicky abdominal pain . he had a past history of similar attacks two times , at that time pain was relieved after oral treatment . various investigations like hemoglobin , total and differential wbc count , platelet count , esr , urine and stool examination were normal . as per surgical opinion conservative treatment was started with ciprofloxacin infusion ( 20mg / kg / day in two divided doses ) , inj . metronidazole ( 30mg / kg / day , in three divided doses ) , inj . five minutes after starting infusion of ciprofloxacin , patient developed itching followed by rashes at the site of infusion . on dermatological examination , reaction started from the site of injection and spread upward involving whole arm up to chest . infusion was stopped immediately and injection dexamethasone ( 0.5mg / kg ) was given along with injection chlorphenaramine maleate ( 0.1mg / kg ) . levofloxacin ( 10mg / kg / day ) was added in treatment in place of ciprofloxacin from next day under close observation . immediately after levofloxacin infusion , itching and rashes in arm appeared so , infusion was withdrawn . ceftriaxone ( 100mg / kg / day ) injection added in the place of fluoroquinolones along with metronidazole . it was moderate in severity according to modified hartwig and siegel 's scale and non preventable as per modified schumock and thornton scale . all fluoroquinolones have similar core structure ( 4-oxo-1 , 4-dihydroquinoline ring ) with fluorine atom attached at position 6 except , first generation quinolone , nalidixic acid . norfloxacin , a second generation fluroquinolone , is the result of replacement at c-7 methyl side chain with piperazine group , while replacement of n-1 ethyl group of norfloxacin with cyclopropyl group generates ciprofloxacin . levofloxacin , third generation flouroquinolone , has modification of piperazine group with methyl group at c-7 . although changes in position 1 , 7 and 8 generated different fluoroquinolones , core structure remains same . 54.5% immediate type of reactions to fluoroquinolones are ige mediated and occurs due to binding of ige to 7 position of core structure of fluoroquinolones . patient had a past history of two similar attacks , at that time he might be treated with fluoroquinolones , which may resulted in production of ige antibodies . as a result , treatment with fluoroquinolones this time might have caused the reaction . ciprofloxacin and levofloxacin have similar core structure ; this may be the reason for the cross sensitivity in this case . it is advisable to avoid other fluoroquinolones when hypersensitivity reaction detected to one and should be shifted to other class of antimicrobials . overall risk of adverse effects especially hypersensitivity reactions with fluoroquinolones is low but due to widespread use , such rare hypersensitivity reactions should be kept in mind . improvement in diagnostic tests like detection of ige antibodies , cellular tests and skin test may help in preventing such adverse drug reactions .

seven years old male child ( 24 kg weight ) diagnosed as a case of sub acute appendicitis treated with ciprofloxacin , immediately developed multiple erythmatous papules . reaction subsided after withholding ciprofloxacin and treatment with dexamethasone and chlorpheneramine maleate . it was developed again when treated with levofloxacin and subsided after withdrawal . ige binding at 7th position of core structure of fluoroquinolones likely to be the mechanism . as all the fluoroquinolones have similar core structure , hypersensitivity to one may have cross sensitivity to other fluoroquinolones . it is advisable to avoid other fluoroquinolones and switch over to other group of antibiotics when hypersensitivity to one occurs .

female genital tuberculosis ( fgtb ) is common in developing countries and associated with significant morbidity in the form of menstrual dysfunction , infertility , tubal block , peritubal adhesions , intrauterine adhesions , and perihepatic adhesions . fallopian tubes are involved in about 90% of the cases with findings such as congested tubes , tubercles , or caseous nodules on them , peritubal adhesions , and tubal blockage at various levels such as cornual block , mid - tubal block , multiple tubal blocks , or fimbrial block , hydrosalpinx , pyosalpinx , and tubo - ovarian masses . a 25-year - old woman with family history of pulmonary tb was presented to the gynecology outpatient department with primary infertility and oligomenorrhea of 3 years . on examination , vaginal examination demonstrated anteverted uterus with fullness and induration in both fornices . on investigations , hysterosalpingogram which was already performed by the referring doctor for infertility before diagnosis of fgtb was made showed bilateral tubal block . endometrial aspirate performed in premenstrual phase showed positive polymerase chain reaction based on the amplification of the 240 bp region of the mpt 64 gene using primers mpt 1 ( 59-tccgctgccagtcgtcttcc-39 ; nt 460479 ) and mpt 2 ( 59-gtcctcgcgag tctaggcca-39 ; nt 700681 ) with equipment using amplitron thermocycler ( barnstead / thermolyne ) , it also tested positive for culture for mycobacterium tuberculosis using mycobacterium growth indicator tube-960 . , there were findings of tb in the pelvis in the form of multiple tubercles on tubes and uterus , bilateral hydrosalpinx with beading of tubes , and few caseous nodules . when methylene blue dye was injected in the uterus , the fallopian tubes became distended and blue colored with beading looking with alternate constrictions and dilatation looking - like blue pythons diagnosis of genital tb was made , and she was started on anti - tb therapy . her postoperative period and follow - up were uneventful . left fallopian tube with distension , multiple ballooning , and blue coloration with fimbrial block showing blue python sign although gold standard in diagnosis of fgtb is demonstration of mycobacterium tuberculosis either on microscopy or culture of endometrial biopsy or presence of epithelioid granuloma on histopathology , they are positive in only few cases leading on to missing of diagnosis in many cases . polymerase chain reaction on endometrial biopsy is a sensitive and rapid method for detecting mycobacterium dna ( mpt 64 gene ) but can be false positive and may not be able to differentiate between infection and disease . gene xpert has recently been introduced in both pulmonary and extra - pulmonary tb as a sensitive and specific method , but its data in fgtb are very sparse . imaging modalities such as ultrasound , computed tomography scan , magnetic resonance imaging , and positron emission tomography scan have main role in tubo - ovarian masses . hysterosalpingography is generally avoided in a suspected case of fgtb due to risk of flare up of the disease but may demonstrate tubal block and other tubal patterns , especially in advanced case . hysteroscopy is useful in endometrial disease and may show pale looking endometrium with endometrial cavity being partially or completely obliterated by adhesions of varying grade ( asherman 's syndrome ) which may involve ostia . however , laparoscopy is the most reliable tool to diagnose fgtb , especially for tubal , ovarian , and peritoneal disease . there can be tubercles on peritoneum or tubes , tubo - ovarian masses , caseous nodules , encysted ascites , various grades of pelvic adhesions , hydrosalpinx , pyosalpinx , beaded tubes , tobacco pouch appearance , and inability to see tubes due to adhesions . other authors have also found laparoscopy very useful in diagnosis and management of genital and peritoneal tb with the advantage of avoidance of laparoscopy which is more traumatic and hazardous especially in a case of abdominopelvic tb . in a case of fgtb , if there is no cornual block , there is a partial or complete fimbrial block with multiple blocks at various parts of tube as is typical of fgtb , dye enters the tube but remains in various parts of tubes with alternate dilatation and constriction making the tube look like a blue python as happened in the present case . the new sign case is easily diagnosed by gynecologists during routine laparotomy performed for infertility patients with suspected fgtb and can aid in its diagnosis

female genital tuberculosis ( fgtb ) is an important cause of infertility in developing countries . various type of tb salpingitis can be endosalpingitis , exosalpingitis , interstitial tb salpingitis , and salpingitis isthmica nodosa . the fallopian tubes are thickened enlarged and tortuous . unilateral or bilateral hydrosalpinx or pyosalpinx may be formed . a new sign python sign is presented in which fallopian tube looks like a blue python on dye testing in fgtb .

porokeratoses are a group of cutaneous entities of unknown etiology , characterized by disordered epidermal keratinization and , at histological examination , by the presence of coronoid lamella , a column of parakeratotic cells extending through the stratum corneum . disseminated superficial porokeratosis ( dsp ) is a distinct form of porokeratosis that , clinically , is characterized by numerous , small , superficial keratotic papules with verrucoid aspects , an atrophic center and peripheral keratotic border . only 7 cases of this association have been described in the literature ( table 1 ) . we report a eighth case of this type and an atypical immunohistochemical method used to characterize the amyloid substance . a 76-year - old woman was referred to our department in june 1999 . she reported a 20 years history of an erythematous and squamo - papular eruption located on the extremities , without itching or pain . the patient had noted a progressive extension of these lesions in the last year and an exacerbation of the lesions during the summer . upon physical examination , numerous papules with a size of less than 15 mm , an atrophic center and hyperkeratotic borders were distributed on the neck , chest and extremities . palms , soles , mucous membranes and nails were spared , and the patient denied any family history of similar skin lesions . we examined a skin biopsy that revealed atrophy of the epidermis with hyperorthokeratosis and presence of homogeneous and compact cornoid lamella of keratin , typically oriented sideways with respect to the epidermal tissue ( fig . 1 a ) . the superficial dermis showed a slight chronic inflammatory infiltrate with vascular ectasia and edema . moreover , in the papillary dermis a compact deposit of a cellular eosinophilic material suspicious for amyloid was observed ( fig . 1b ) . to demonstrate the epithelial origin of the amyloid , we performed two immunohistochemical stainings by using specific moabs to pan - cytokeratin ae1-ae3 . interestingly , there was a striking difference between the two results : in fact , the acellular deposits , mentioned above , were negative for ckae1-ae3 , whereas a strong immunoreactivity was demonstrated for ck5 , supporting a specific origin of the amyloid substance from the basal cell layer of the epidermis ( fig . in 1937 , andrews first described dsp and introduced this term to indicate a clinical variant of mibelli 's porokeratosis . later , chernoski and freeman proposed a possible actinic etiology of this dermatosis and coined the term disseminated superficial nowadays , this term is generally accepted in european dermatological literature , and this definition is based on clinical and histological findings . dermatological manifestations are typically confined to sun exposed areas , with actinic induction and exacerbations . in the literature , two types of localized cutaneous amyloidosis ( lca ) are described : primary lca ( macular amyloidosis and lichen amyloidosis ) , which is not associated with other dermatoses or systemic involvement , and secondary lca , which is associated with inflammatory , hamartomatous or neoplastic skin disorders . the mechanism by which dsp induces dermal amyloid deposits is not clear , but piamphongstant et al . first suggested that this process can derive from degenerated epidermal keratinocytes . we hypothesize that a mutant keratinocyte clone is responsible for induction of porokeratotic lesions , because these necrotic epidermal keratinocytes ( colloid bodies ) might be transformed into amyloid by dermal macrophages and fibroblasts . immunohistochemical staining has shown an overexpression of p53 protein in porokeratotic lesions ; this is a tumor suppressor protein , an important gatekeeper and effector of the cell cycle . mutations of the p53 gene in all forms of porokeratosis , also in dsap , create a permissive state of uncoordinated cell cycling , and predispose cells to death . in our case , the lack of systemic involvement led to our hypothesis of a secondary dermal deposition of amyloid proteins and the existence of a close relationship between these two processes . this hypothesis was confirmed by positive staining with congo red and immunohistochemical staining with the anticytokeratin moabs strongly positive for ck5 , just below the epidermal porokeratotic zone in close proximity to the cornoid lamella . this cytokeratin is , in fact , strongly represented in the basal cell layer and these results indicate that the dermal deposits were amyloid originating from the epidermis ( type ii keratin ) . nowadays , more studies are necessary to clarify the exact mechanism that leads to secondary deposition of dermal amyloid in porokeratotic disease and the frequency of this association . we also suggest utilization of moabs for ck5 as a first - line target in these conditions , encouraged by the immediate and strong positivity of this protein in immunohistochemical studies , whereas pan - cytokeratin at first produced negative results .

only 6 cases with an association of disseminated superficial porokeratosis with dermal amyloid deposits are reported in the literature . we present the case of a 76-year - old woman who presented with a disseminated superficial porokeratosis . histological examination revealed amyloid deposits in the upper dermis , which were typed with routine he stains , congo red stains and anticytokeratin antibodies ( ae1-ae3 and ck5 ) . positive staining with congo red and , moreover , with ck5 ( a cytokeratin strongly represented in the basal cell layer of the epidermis ) indicates an epidermal origin of this protein .

hemangiomas are common lesions of the head and neck area , and they predominantly originate from the lips , tongue and buccal mucosa . the capillary type is lobulated and it lacks a capsule , it is purplish in color and it infiltrates the involved glands . microscopically , solid masses of cells and multiple anastomosing capillaries that replace the acinar structure of the gland are seen . the cavernous type is formed by dilated blood vessels or sinusoids lined by endothelium ( 1 ) . cavernous hemangioma of the external auditory canal ( eac ) and tympanic membrane is rare , but this lesion is more common in the eac . according to the previously reported cases ( 2 - 8 ) , cavernous hemangioma seems to most frequently arise in the posterior portion of the eac . however their incidence in the tympanic membrane is very rare and there have been only 4 such case reports in the literature according to a medline search from 1970 to 2008 ( 9 - 12 ) . we report here the 5th case of isolated cavernous hemangioma that was limited to the tympanic membrane and we review the relevant literature . a 49-year - old man patient was referred to our department for evaluation of right - sided pulsatile tinnitus that he had experienced for the previous 2 years . the patient denied having hearing loss or vertigo and he had no evidence of facial palsy . endoscopic examination of the eac revealed a red - colored soft non - pulsatile mass that occupied part of the tympanic membrane ( fig . the tympanic membrane was partially obscured by the mass and it was not clearly mobile . a pure tone audiogram ( pta ) showed high frequency mild sensorineural hearing loss in the right ear at 3,000 - 8,000 hz ( fig . 2 ) . temporal bone computerized tomography ( ct ) showed an isolated soft tissue mass just lateral to the tympanic membrane . a tympanomeatal flap was elevated to reveal a mass with involvement of the tympanic membrane . the mass was spongy on palpation , and it was removed en bloc with little bleeding . the defect of the tympanic membrane was reconstructed by type i tympanoplasty and using a piece of fascia taken from the temporal muscle . pathologic examination of the specimen showed a well - demarcated nodular lesion composed of dilated vascular spaces lined by a single layer of flat endothelial cells ( fig . the postoperative endoscopic examination showed an intact state of the neo - drum with good ventilation . in 1983 , andrade et al . ( 10 ) reported the first case of cavernous hemangioma limited to the tympanic membrane in a 59-year - old man with a contralateral ear infection . according to the previously reported cases , cavernous hemangioma seems to most frequently arise in the posterior portion of the eac . conductive hearing loss , tinnitus and aural fullness may be present . in the present case , the differential diagnosis includes aural polyp , glomus tumor , arteriovenous malformation , granulation tissue and carcinoma of the eac . a temporal bone ct scan is the first choice for the evaluating the location and size of the lesion and middle ear involvement . the majority of the previously reported cases showed a normal middle ear . only 1 case reported by magliudo et al . ( 8) had cavernous hemangioma that initially involved the middle ear space through a small perforation in the tympanic memrane and the tumor had reached the handle and the short process of the malleus . for an advanced lesion , diagnostic angiography should be obtained to identify the blood supply to preoperatively assess the safety and feasibility of performing embolization ( 5 ) . surgical treatment with en bloc excision is generally curative , and preoperative embolization is usually not necessary except for an advanced lesion . the endaural approach is generally used . in this case , we removed the mass by the postauricular approach due to the size of the mass .

cavernous hemangioma seems to most frequently arise in the posterior portion of the external auditory canal . however , they rarely occur in the tympanic membrane . a 49-year - old male patient was referred for evaluation of right - sided pulsatile tinnitus that he 'd experienced for the previous 2 years . temporal bone computerized tomography showed an isolated soft tissue mass just lateral to the tympanic membrane . there was no evidence of bony erosion or middle ear invasion . the patient underwent excision of the mass using a postauricular approach . the mass was removed en bloc and the defect of the tympanic membrane was repaired by tympanoplasty type i. there was no recurrence after 1 year of follow - up .

a subset of patients with myositis have unique autoantibodies . this has informed and altered our approach to the diagnosis and management of the inflammatory myopathies . the identification of myositis - specific autoantibodies ( msa ) and myositis - associated autoantibodies ( maa ) is important because they are associated with specific clinical phenotypes , and may guide the physician in terms of treatment planning . this study reports the experience and clinical utility of the extended myositis antibody ( ema ) panel across a range of specialties in a large teaching hospital . cork university hospital ( cuh ) is the largest university teaching hospital in ireland , and is a multi - specialty tertiary referral centre serving a population of 1.1 million . we conducted a retrospective review of the electronic and paper records of all patients who had serum samples tested for msa and maa from april 2014 -mar 2015 . euroline autoimmune inflammatory myopathies immunoblot was performed at university hospital galway . this assay uses membrane strip antigen testing to detect anti- : mi2 , tif1 gamma , mda3 , nxp2 , sae1 , ku , pm- scl 100 , pm - scl 75 , oj , ej , jo-1 , pl-7 , pl-12 , scl 70 , centromere a , centromere b , rna pol iii , fibrillarin , nor 90 , th / to , ku , pdgfr and ro-52 . the use of additional investigations ( electromyography , mri , muscle biopsy , ct thorax ) and laboratory results , including creatine kinase and autoantibody profile , were documented . we reviewed the utility of the assay in clarifying diagnosis , directing the investigative pathway and selecting the appropriate treatment . twenty two patients ( mean age : 55 , sd:15 ) had an ema panel sent during the study period . departments across the hospital included respiratory medicine ( n=8 , 36% ) , rheumatology ( n=5 , 23% ) , neurology ( n=4 , 18% ) , and other ( n=5 , 23% ) . additional investigations performed depended on the clinical picture but included cardiac or musculoskeletal mri ( n=8 , 36% ) , ct thorax ( n=14,64 % ) , muscle biopsy ( n=7 , 32% ) and emg ( n=6 , 27% ) . these autoantibodies were ana ( n= 10 , 45% ) , ena ( n=4 , 18% ) , anti - ro ( n=3 , 14% ) , anti - la ( n=1 , 5% ) , anti - dsdna ( n=1 , 5% ) and p- anca ( n=1 , 5% ) . of the 17 patients who had a ck recorded , six ( 27% ) were elevated . investigations and outcomes of patients with a positive ema panel are shown in table 2 . patient 3 was a 35-year - old woman who presented in acute heart failure , nyha ii . she had an elevated troponin ( 600s ) and ck ( 1787 ) , yet had a normal cardiac mri and transthoracic echocardiogram . ema panel was positive for anti pm - scl 75 and anti pm - scl 100 antibodies , providing evidence that her cardiac failure was secondary to an autoimmune process . her antibody profile resulted in first line treatment with rituximab , avoiding use of cyclophosphamide in a young woman who intended to start a family . one - month post rituximab infusion her dyspnoea had resolved and both her ck and troponin levels had normalised . patient 4 presented with a parietal stroke , and had a ck of 1539 on admission . ema panel was positive for anti - pl7 , resulting in a diagnosis of anti - synthetase syndrome . consequently , ct thorax and pulmonary function tests were performed , as well as onward referral to a respiratory physician . treatment with a combination of rituximab and steroids has halted the progression of her dyspnoea , and has lead to a resolution of her weakness . patient 6 tested positive for anti - tif1 gamma , and as a result has an annual ct - thorax abdomen and pelvis screening for an occult malignancy . two were treated with steroids alone , three received steroids in combination with rituximab , and one received steroids followed by azathioprine and then mycophenylate mofetil . ema panel is entering standard clinical practice but is not yet a routine tool in the investigation of idiopathic inflammatory myopathy ( iim ) in all centres . diagnosis of autoimmune myopathy was previously dependent on muscle biopsy , emg and radiological investigations . with the advent of the ema panel use of the panel avoided an invasive procedure ( muscle biopsy ) in two patients . antibodies are detectable early in the disease course , and are specific for autoimmune myopathy the ema panel was diagnostic in 27% of patients , when traditional testing had not been definitive . a positive ema panel is of significant clinical utility in facilitating decisions on appropriate investigations . patient 6 in our study has entered a cancer surveillance programme after testing positive for anti - tif1 gamma ; an antibody associated with a significantly increased risk of malignancy . in anti - synthetase patients 4 and 5 in our study had dyspnoea on presentation , and features of ild on imaging . all patients diagnosed with anti - synthetase syndrome should have a high resolution ct thorax and pulmonary function tests performed . onward referral to a respiratory physician , as was the case for patients 4 and 5 in our study , should be considered . autoimmune myopathies are important to identify as they often respond to immunosuppression . in our study all patients with a positive ema panel ( n=6 , 27% ) experienced symptomatic improvement on receiving immunosuppressants . a positive panel in patient 3 provided evidence for use of rituximab , as opposed to cyclophosphamide . in addition to myositis , a constellation of clinical features have been described in inflammatory myopathies , including dyspnoea , raynaud s phenomenon , polyarthritis , fever and weight loss . ild may precede the occurrence of overt myositis in up to 20% of cases , and is estimated to result in an excess mortality of up to 50% . the multisystem nature of autoimmune myopathy means patients need collaborative input from different medical specialities . ongoing involvement of these physicians is particularly important ; all of whom need to be familiar with the diverse clinical presentation of iim . this study illustrates the value of the ema panel in defining a heterogeneous patient population into clinicoserological phenotypes , thus guiding treatment pathways . furthermore , it highlights the diversity of these presentations , the need for multi - speciality input and serves to heighten awareness among clinicians of the diagnostic use of extended myositis antibody testing in these cases .

the discovery of unique autoantibodies has informed and altered our approach to the diagnosis and management of the inflammatory myopathies . this study reports the initial clinical experience of use of the extended myositis antibody ( ema ) panel in the largest university teaching hospital in ireland . we conducted a retrospective review of all patients who had serum samples tested for myositis specific antibodies and myositis associated antibodies from april 2014 to march 2015 . a positive ema panel was of significant clinical utility in facilitating decisions on appropriate investigations , and need for onward referral to other physicians . furthermore , this paper highlights the diversity of possible presentations of idiopathic inflammatory myopathy with subsequent need for multi - speciality involvement , and serves to heighten awareness among clinicians of the diagnostic use of extended myositis antibody testing in these cases .

pica actually refers to the latin word for magpie , a bird thought to have odd feeding habits . incidence of pica has also been linked to iron and zinc deficiency . whether the pica is an eating disorder or obsessive compulsive disorder is still controversial . the patient was a 27 year old young indian lady living in the baruipur with no previous psychiatric or medical history . patient complained of an unusual sensation in her abdomen for the last 3 months while her parents gave a history of habitual consumption of nails for the same duration . the patient was a single child to her parents and she was living in the suburbs since her birth . she had attended public schools and had completed her graduation on arts from a college in kolkata . soon after her marriage , she expressed sexual aversion with history of dyspareunia . within a month of her marriage , she found it extremely difficult to live in her in - laws house and preferred to stay at her parental home , although her parents repeatedly insisted her to go back to her in - laws residence . depressed and also admitted that for the last 3 months , she was having a sense of hopelessness and worthlessness over her future days . vanished. she temporarily enjoyed the gritty and spiky sensation of nails along her throat , but at night she used to have a vague abdominal sensation which she described to be funny. according to her mother , she did not want to go back to live with her husband . on close interviewing of the patient , initially her response to same- sex relationship was guarded but later it was found that she was actually having such a relationship with another lady of her age for the past 3 years . she also accepted that she used to consume glass piece occasionally since last 3 years but never disclosed them to anyone . she had once had an x - ray of her abdomen done 2 years back due to complaints of low back pain which was inconclusive ; perhaps glass beads which are radiolucent were missed . no further investigation was done . the patient was decently dressed in a pastel shade salwar with a boyish cut hairstyle . on mental state examination on interviewing the husband , it was known they had never had any sexual intercourse since marriage . on routine examination , possibility of iron deficiency anemia , serum ferritin abnormality and zinc deficiencies were ruled out . x ray of abdomen showed multiple radio opaque pointed objects in her gut ( ingested iron nails ) . the patient had been nurtured in an overprotected environment by her parents in her early childhood days . however immediately following her high school , she had to take many responsibilities of the family all of a sudden because of loss of job of her father . she was more bonded to her mother and in the elderly , often avoided her father because he was alcoholic . the cultural , psychodynamic , and physiological aspect of this case is a unique one . the patient 's initial experimentation with ingestion of glass beads , though in a very remote sense , may be culture based considering the fact that the practice of ingesting such beads and sharp metallic objects has been reported . considering the fact that the patient was initially nurtured in an overprotected environment by her parents and suddenly being exposed to a comparatively hostile situation where she found a discouraging picture of the members of the opposite sex from that of her alcoholic father , the patient 's inclination toward same - sex relationship can be explained . psychologically , the inclination of the patient toward same sex relationship and her unfamiliarity and uneasiness with the members of the opposite sex led to marital dissatisfaction . also , since the patient had to take many burdens of her family at an age which may be considered quite an early one considering the indian standards and her socioeconomic profile , this probably has caused her to resort to some , harsh robust means like ingestion of nails which may have been an act to depict her her intelligent quotient as measured by wechsler 's adult intelligence scale had been found to be 98 which fall in normal age . as in most cases of pica , she initially did not come with a complaint of pica . rather , she had reported to the hospital with complaints of unusual sensation in her abdomen . the present case presented a unique conglomeration of cultural , psychodynamic , and physiological determinants which together contributed to the expression of pica in comparison with previous reports and reviews . however , an interesting fact in this case is the age of the patient which is quite unusual for pica to occur . this perhaps necessitates further research in dealing with the epidemiology and other aspects of this quite rare but interesting disease entity .

pica has been considered as the ingestion of inedible substances or atypical food combinations . pica has been reported widely in pediatric age group and often found to be co existing with obsessive compulsive or major depressive disorder . reports of pica in elderly age group are relatively uncommon and rarely does it have an adult onset . in this article we present a case of adult onset pica . a young lady with unusual sensation in her abdomen was found to consume iron nails over years and there was history of dyspareunia since her marriage three months back . on query it was known that the lady is having same sex relationship over years . there unique conglomeration of cultural , psychodynamic and physiological determinants which together is responsible for this unusual habit of this lady . moreover the onset of the disease at a late age and different psychodynamic issues make the case all the more interesting . whether the pica is an eating disorder or obsessive compulsive disorder is still controversial . pica has been mentioned in diagnostic and statistical manual iv tr . the present case report warrants the need to look into this entity more closely with regards to its occurrence and etiology .

chronic cough is a frequent presenting complaint to not only the general practitioner but also the otolaryngologist and respiratory specialist . diagnosis can be difficult , especially in primary care , but empirical treatment for common causes such as asthma , chronic obstructive pulmonary disease , gastro - oesophageal reflux disease , postnasal drip or drug - induced cough ( most commonly secondary to angiotensin converting enzyme inhibition ) is recommended when there are no associated worrying symptoms , and is successful in the majority of cases . symptoms that may signify a rarer but more serious cause for chronic cough include the copious production of sputum , fever , sweats , unintentional weight loss , haemoptysis , clubbing , hoarseness and marked dyspnoea . here we highlight a unique cause of chronic cough : the initial symptom of lymphoma compressing the brainstem . a 69year - old gentleman presented to his general practitioner with a 3-month history of progressive unsteadiness . on further questioning he also volunteered that these symptoms had been preceded by a chronic cough , which had no obvious precipitant , and which had failed to resolve . on examination he had truncal ataxia , to the point where he had difficultly remaining upright when walking , and exhibited gaze - evoked first - degree nystagmus in his left eye on looking to the right . the patient had suffered an episode of labyrinthitis 10 years earlier , and so he was prescribed pharmacological therapy aimed at a presumptive diagnosis of a second episode . successive trials of prochlorperazine , betahistine , cyclizine and arlevert were not noted to have any positive effect on the patient 's dizziness and he was referred for outpatient neurological opinion . by the time of specialist review , the patient 's symptoms had worsened and he now described periods of severe nausea , with progression of his ataxia to the point where he was unable to stand unaided . he had also developed an area of hypoesthesia on the left side of his face . eye movements were adjudged to be markedly abnormal with dysmetric horizontal saccadic movements in addition to his ongoing nystagmus . his right vestibular ocular reflex was reduced and his left upper limb movements lacked coordination . gadolinium - enhanced magnetic resonance imaging ( mri ) of the brain revealed a homogenously enhancing , sharply demarcated 13 mm diameter lesion in the left posterior and lateral midbrain extending into the inferior cerebellar peduncle , with some adjacent oedema , suggestive of primary lymphoma ( see fig . 1 ) . stereotactic brain biopsy confirmed diffuse large b - cell lymphoma ( dlbcl ) and subsequent bone marrow biopsy and computer tomography ( ct ) imaging showed no evidence of lymphoma at any additional anatomical site . figure 1:gadolinium - enhanced t1-weighted mri images ( panes lettered with coronal ( a ) , axial ( b ) and sagittal ( c ) sections ) showing a homogenous enhancement of a clearly demarcated lesion in the left posterior and lateral midbrain with extension into the inferior cerebellar peduncle . gadolinium - enhanced t1-weighted mri images ( panes lettered with coronal ( a ) , axial ( b ) and sagittal ( c ) sections ) showing a homogenous enhancement of a clearly demarcated lesion in the left posterior and lateral midbrain with extension into the inferior cerebellar peduncle . the patient embarked on idaram ( idarubicin , cytarabine , methotrexate , dexamethasone ) chemotherapy followed by two rounds of high - dose methotrexate mono - therapy . imaging following treatment showed radiological remission and this is also seen on recent scans , now 2 years later ( see fig . 2 ) . while the symptoms of cerebellar dysfunction have largely abated since discharge , the patient has , however , been continually troubled by his cough . figure 2:compares initial t1-weighted gadolinium - enhanced mri images ( seen in panes on left hand side : a , c and e ) with those taken following completion of treatment ( panes on right : b , d and f ) . radiological remission is shown in the post - treatment images . compares initial t1-weighted gadolinium - enhanced mri images ( seen in panes on left hand side : a , c and e ) with those taken following completion of treatment ( panes on right : b , d and f ) . radiological remission is shown in the post - treatment images . otolaryngology assessment , including symptom review , medication history , endoscopic evaluation and chest x - ray , was unable to elucidate any cause for cough other than the presumed effects of the medullary lesion . empirical trials of nasal steroids , nasal saline washes , high - dose proton pump inhibitor and inhaled bronchodilators were all unsuccessful . coughing is a protective motor reflex under both voluntary and involuntary regulation , integrating physiological , psychological and social inputs . studies in experimental animals have shown that vagal afferents signal peripheral sensory stimulation from the airways and lungs , and project centrally to the nucleus tractus solitarius ( nts ) with further projections between the nts and the medulla and pons . that such projections constitute a regulatory brainstem cough centre is a concept that has been extensively pursued ; attempting to determine the specific central pathways involved in the initiation and regulation of the efferent arc of the cough reflex . the aetiology of chronic cough has been considered in a similar manner to the aetiology of chronic pain , with elements of both peripheral and central sensitization . most of the common causes of chronic cough , and notably the classically described diagnostic triad of asthma , gastro - oesophageal reflux disease and postnasal drip , arise primarily through the activation of peripheral vagal afferents , but with probable central maladaptation contributing to hyper - sensitization to peripheral stimuli . pathological impingement of the dorsal medullary region of the brainstem is recognized as a neurogenic cause of chronic cough in cases of patients with chiari i malformations and cough may be the initial symptom . however , to our knowledge , this is the first reported case of a lymphoma around the brainstem with chronic cough as one of the first symptoms . it is difficult to be certain as to the mechanism of chronic cough induced by this lymphoma . it may be that the expanding mass of the dlbcl compressed nearby nerve efferents serving the cough reflex , leading to aberrant hypersentization . however , dlbcl can be an aggressive lesion and has been reported to present with focal neurology from local invasion into nerves . it is possible , therefore , that the chronic cough in this case was caused by tumour infiltration into nerves rather than compression . although clearly cerebral lymphoma is a very rare cause of chronic cough , we believe that clinicians in primary and secondary care should consider rare central causes of chronic cough in cases where neurological symptoms or signs are present , or in cases where empirical treatment for the more common causes of cough fails to provide symptomatic relief .

we report a rare case of chronic cough as the presenting symptom of a primary brainstem lesion . a 69-year - old gentleman presented with chronic cough followed by onset of progressive truncal ataxia , incoordination and nystagmus . contrast - enhanced imaging showed a midbrain lesion extending into the cerebellum , confirmed as lymphoma by stereotactic brain biopsy . the patient was successfully treated with chemotherapy , but his cough persists . we present this case to highlight the possibility of a brainstem lesion as a rare central cause of chronic cough , and suggest that the diagnosis is considered in those with concomitant neurological symptoms .

mycobacterium tuberculosis isolates from all culture - positive tb patients in seattle and king county , washington , during 20032004 were genotyped by spacer oligonucleotide typing and mycobacterial interspersed repetitive unit methods . we included patients who had an isolate that matched the outbreak strain or who had a social link to an already included patient . patient medical records were reviewed , and infectious periods were calculated . for sputum smear positive patients , the infectious period extended from 3 months before symptom onset or the first positive smear ( whichever was earlier ) until 2 weeks after the start of appropriate tb treatment or until the patient was placed into isolation or produced consecutively negative smears . for sputum smear negative patients , the infectious period extended from 1 month before symptom onset , the start of appropriate tb treatment , or the date that the patient was isolated ( whichever was earlier ) , until 2 weeks after the start of appropriate tb treatment or until patient isolation ( 7 ) . we interviewed patients to learn their contacts , activities , and locations frequented while they were contagious . additional contacts were found by outreach workers and a disease intervention specialist from the east - african community who was hired to work in the neighborhoods frequented by the patients . while in these neighborhoods , outreach workers and the disease intervention specialist recruited persons seen with patients or their contacts to be evaluated for tb and latent tb infection . contact activities , specifically those related to illicit drugs , were observed or self - reported . friends were defined as contacts of patients who spent time within a close - knit network of young men who exhibited similar marijuana - using behavior . other contacts were defined as the families and relatives of patients and those who were named but were not closely associated with this network . contacts received a tb evaluation including a tuberculin skin test ( tst ) to detect infection . patient 1 was first evaluated in december 2003 , when a chest radiograph suggested pulmonary tb ( i.e. , upper lobe cavitary infiltrate ) . however , only clarithromycin was prescribed , and the patient was lost to follow - up . he was again seen in an emergency room in april 2004 after the infection evolved into bilateral extensive pulmonary tb . eight patients were born in east africa ; a median of 13 years ( range 622 ) had passed since their arrival in the united states . patient 5 was a white woman who received illicit drugs from patient 1 . * unknown for 1 patient . patients were symptomatic and had findings indicating infectiousness : all had pulmonary tb , 7 had cavitary disease , and 8 had sputum that tested smear - positive for acid - fast bacilli . consecutive chest radiographs indicated progression to cavitary disease in < 75 days weeks in 3 patients and < 121 days in another patient . table 2 shows the dates of clear chest radiographs interpreted as normal and the first chest radiographs showing disease . while contagious , patients stayed in various locations , including cars , for most of the day . a single - bedroom apartment occupied by at least 1 patient while he was contagious was regularly visited by 2 other patients . numerous members of the friend network slept there on any given night , and many others would regularly visit during a 10-week period beginning in april 2004 ( figure ) . the occupants nailed boards over the apartment windows to conceal activities , primarily marijuana use , from outsiders . all reported frequent " hotboxing , " the practice of smoking marijuana with others in a vehicle with the windows closed so that exhaled smoke is repeatedly inhaled . considerable overlap in infectious periods was noted , which highlights the potential for simultaneous contact with multiple contagious patients . at least 31 ( 57% ) friend contacts spent time at the 1-bedroom apartment . after those with a past positive tst result were removed , 14 ( 64% ) of 22 screened friends and 6 ( 23% ) of 26 other contacts had a positive tst result . the risk for a positive tst result was 2.8 greater among friends than among other contacts ( 95% confidence interval = 1.36.0 ) . twenty - nine ( 54% ) friend contacts self - reported or were observed hotboxing . among the friends who reported or were observed hotboxing , 11 ( 79% ) of 14 who received a tst had a positive result . risk factors for tb include birth in a country with high tb prevalence ( 2 ) and incarceration ( 8) . although most patients in this outbreak were foreign - born and had histories of incarceration , genotyping results and epidemiologic findings suggest that tb was transmitted recently in the community rather than before immigration or during incarceration . frequent marijuana use has been reported among tb outbreak patients ( 9 ) and was the behavior linking these patients together . creative sharing of marijuana has been described recently as a factor for m. tuberculosis transmission . in australia , sharing a water pipe ( i.e. , " bong " ) was linked to transmission ( 10 ) . " shotgunning " refers to inhaling smoke from illicit drugs then exhaling it directly into another 's mouth ( 11 ) and was associated with m. tuberculosis transmission among a group of exotic dancers and their contacts ( 12 ) . this investigation noted that a similar activity , hotboxing , might have contributed to transmission . one patient with smear - positive cavitary disease reported daily hotboxing with friends , often for most of the day . in addition , marijuana smoking might induce cough , creating an ideal environment for transmission . many friends stayed and used marijuana at the single - bedroom apartment during the height of the outbreak . furthermore , by nailing boards over the windows , ventilation was limited , creating an environment similar to that of hotboxing . although progressive primary tb by nature is thought to be due to recent transmission , progressive primary tb with cavitation is uncommon ( 13 ) . however , frequent marijuana use and the setting of intense exposure may have played a role . in addition , poor nutrition and unhealthy lifestyles might have predisposed these young men to more rapid progression of disease . while no laboratory investigation to assess genetic susceptibility or strain virulence was conducted , these factors might have also contributed to the development of cases . this outbreak resembles an outbreak reported among regular patrons of a neighborhood bar ( 14 ) . both were fueled by a highly infectious source patient who spent extended amounts of time indoors with 1 group of persons who regularly used substances ( i.e. , alcohol or marijuana ) . the result in both situations was a higher than expected incidence of tb disease and latent tb infection . in the outbreak reported in this article , however , the substance of choice was illicit and further complicated the control of this outbreak . patients ' illicit drug activities promoted a reluctance to name contacts at risk and locations frequented . efforts had to revolve around meeting these young patients at times and locations convenient to the group . then after gaining the groups ' trust , outreach workers successfully found and screened contacts . many successful screenings took place on street corners and in parking spaces throughout the community . often outreach workers were successful only after spending hours driving throughout the community searching for patients and contacts . alternative strategies to name - based contact investigations may become increasingly critical to tb control as tb recedes further from the general population , yet persists within smaller guarded groups ( 15 ) .

matching mycobacterium tuberculosis isolates were noted among 11 young tuberculosis patients socially linked through illicit drug related activities . a large proportion of their friends , 14 ( 64% ) of 22 , had positive tuberculin skin - test results . the behavior of " hotboxing " ( smoking marijuana inside a closed car with friends to repeatedly inhale exhaled smoke ) fueled transmission .

intraspinal juxtafacet cysts ( commonly , synovial or ganglion cyst ) can be an uncommon cause of lower and radicular pain , neurogenic claudication , and cauda equina syndrome2,9,10,13 ) . spinal synovial cysts generally arise from a degenerated facet joint and often presenting as a gradual onset of back pain and potentially , chronic progressive radiculopathy is a much less common occurrence , and patients may present with acute onset of symptoms3,13 ) . we describe a case in which hemorrhage into a right l2 - 3 facet synovial cyst caused acute back pain and radiculopathy and required surgical excision . a 72-year - old - woman with a past history of hypertension presented to the clinic with acute back pain radiating to the anterior surface of the right thigh and leg . the right straight - leg - raise test elicited pain in the leg at 30 which was exacerbated further by ankle dorsiflexion . magnetic resonance imaging ( mri ) revealed the presence of a cystic formation in the right l2-l3 facet joint with arthritis compressing the l3 right root and the dural sac ( fig . , the mri displayed a hyperintense abnormality on t1-weighted images and hypointense on t2-weighted images consistent with hemorrhage . the posterior elements from l1 to l3 were exposed and right l2-l3 laminectomy and facetectomy were performed . a brownish mass was found in continuity with the right l2-l3 facet joint adherent to the dural sac . the extradural hematoma was visualized and evacuated , effectively decompressing the right 2 nerve root and sacral sac . microscopically , there was a synovial cyst with synovial cell lining , neovascularization , hemosiderin microdeposits , and blood ( fig . one month after the operation , the patient had no symptoms in her lower back and leg . spinal synovial cyst can be defined as a soft tissue mass located extradurally along the medial border of a degenerated facet joint , especially l4 - 5 level1,4,8,11,12 ) . these cysts are filled with clear or xanthochromic fluid and have a synovial - like epithelial lining with a demonstrable connection to a joint capsule3,11 ) . if the synovial cell lining and the communication with a joint capsule are absent , the cyst is classified as ganglion11 ) . intraspinal synovial cysts are rare but have been documented with increasing frequency because of the improvement of neuroradiological imagings4 ) . an mri is considered a good modality for diagnosis . on mri , cysts appear as well - circumscribed , epidural mass lesions that are hypo- and hyper - intense on t1 and t2 weighted images , respectfully . bleeding into a synovial cyst may cause a sudden expansion of the cyst leading to compression of the epidural space result in root compression symptoms . hemorrhage into a synovial cyst results in an acute increase in pain and radicular symptoms , including neurological deficits12 ) . hemorrhagic presentation can be caused by anticoagulation treatment , trauma , disc herniation , vascular anomaly , and neoangiogenesis in the cyst4 ) . in addition , high vascularization of the cyst can cause hemorrhage in spite of minor trauma or just spinal instability . synovial cyst hemorrhagic events in some cases can occur despite the absence of trauma or coagulopathy13 ) . as a result , other yet to be determined risk factors for synovial cyst hemorrhage occurrence exist prompting the need for more studies . non - hemorrhagic synovial cysts are occasionally treated by percutaneous aspiration with successful resolution of symptom6 ) . the injection of corticosteroid agents into the facet joint may be an treatment option . despite conservative treatment , recurrence of cyst with symptoms the natural history of spinal synovial cysts is unknown , but spontaneous remissions of a synovial cyst on follow up imaging with improved symptoms have been reported5 ) however , pain can be treated with steroid injections and bed rest in substantial number of patients . we report of a case in which hemorrhage into a right l2 - 3 facet synovial cyst causing an acute back pain and radiculopathy . treatment by resection of the cyst and evacuation of the hematoma led to complete neurological recovery . synovial cysts of the lumbar spine are an uncommon cause of back and radicular pain . rarely , these cysts can go through hemorrhagic transformation by trauma , anti - coagulopathic state , or for unknown reasons . hemorrhage into a cyst is an uncommon occurrence that can lead to acute symptom . in most cases , surgery is the treatment of choice for symptomatic hemorrhagic synovial cysts . surgery involving the evacuation of the hematoma and/or cyst can result in complete resolution of acute symptoms and neurologic deficit .

synovial cysts of the lumbar spine are an uncommon cause of back and radicular pain . these cysts most frequently present as back pain , followed by chronic progressive radiculopathy or gradual onset of symptoms secondary to spinal canal compromise . although less common , they can also present with acute spinal cord or root compression symptoms . we report of a case in which hemorrhaging into a right l2 - 3 facet synovial cyst caused an acute onset of back pain and radiculopathy , requiring surgical excision .

the unfortunate complication of a colotomy resulting from a colonoscopic polypectomy can be disastrous . using the versatility of laparoscopic surgery we report a case of cecal perforation , in a 70 year old female , following colonoscopic polypectomy that was treated successfully with laparoscopic application of an endo - gia linear stapler . four months following staple resection of her cecal perforation , the patient is asymptomatic and has undergone a repeat colonoscopy without any sequelae . laparoscopic techniques can be safely applied in the treatment of colonic perforations following therapeutic polypectomies . a suggested management algorithm is provided highlighting the role laparoscopy may provide in selected patients . colon perforation during therapeutic colonoscopy has been estimated to occur at a rate of approximately 0.1% to 0.3% . perforation may occur through a variety of mechanisms , including energy application at the polyp site , direct mechanical trauma from the tip of the endoscope , lateral pressure from the side of a bowed endoscope , and pneumatic injury from overinsufflation . several case reports advocating laparoscopic repair of colonic perforation following colonoscopy have been described in the literature . in our present case report , we discuss the single application of an endoscopic linear stapler to close the perforation while preserving colonic lumen integrity in the presence of endoscopically placed mucosal clips . a 70-year - old caucasian female with a past medical history significant for sigmoid diverticulosis and previous benign polyps of the colon was found to have a lipomatous lesion in the ascending colon during routine surveillance colonoscopy . a complete resection of the polyp was performed using a 15-ml saline injection with india ink lift and a hot snare . after resection , the mucosal biopsy site was larger than anticipated , and it became difficult to maintain insufflation . the procedure was halted secondary to progressive abdominal pain and the clinical suspicion of a colonic perforation . the perforation was confirmed with the presence of substantial free air on radiographs . in this rare situation of early discovery and hemodynamic stability , the patient was taken immediately to the operating room for surgical repair . in the operating theater , pneumoperitoneum was established with a veress needle , and a 5-mm camera with an optical access trocar was placed in the left lower quadrant . of note , the abdominal pressure at initial access was measured at 10 mm hg , consistent with pneumoperitoneum from insufflation through the colotomy . a 5-mm anterior perforation of the cecum was clearly visible with an area of circumferential coagulation extending approximately 2 mm ( figure 1a ) . the cecum was noted to be adhered to the side wall , and hence one additional 5-mm port and one 10-mm port were placed for mobilization in the midline . the ports were placed in the umbilicus and the suprapubic position . after a sufficient segment of the cecum was freed , a single 30 silk stitch was placed to provide temporary closure and to provide a handle for elevation of the cecum . an endo - gia 60-mm blue load was applied to preserve the lumen of the cecum while incorporating the entirety of the defect including the area of coagulation necrosis ( figure 1b ) . the suture line incorporated healthy pink mucosa with preservation of the colonic lumen ( figure 1c ) . the patient had an uncomplicated recovery with discharge on hospital day 4 . at the 4-month follow - up , the patient is without any complaints , tolerating a regular diet , and at baseline health . she has had an interval surveillance colonoscopy without any abnormalities and a well - healed polypectomy / surgical site . a : anterior cecal perforation with surrounding circumferential area of necrosis and endoscopically placed clips ( white arrows ) . b : endo - gia 60 mm stapling with careful preservation of colonic lumen with incorporation of the entirety of the defect and area of coagulation necrosis . colotomy that occurs during colonoscopy is a specialized subset of colon perforations for a variety of reasons . to begin with , in many cases the patient is under the care of a physician in a monitored setting with established intravenous access . although there may be some question as to the precise benefits of mechanical preparation in elective colorectal surgery , in the emergent setting it may be advantageous in minimizing the amount of gross spillage . in addition , the endoscopist may provide localization of the suspected injury , allowing for more directed treatment . it has been advocated by some that colonoscopic polypectomy should be performed in conjunction with laparoscopy when a particularly difficult or suspicious polyp is anticipated . in keeping with the success of previous case reports and advances in minimally invasive techniques , we propose an active role of laparoscopy in complex colonoscopies as outlined in figure 2 . perforations , depending upon their size , may be treated with direct suturing , staple resection or segmental resection . our approach to this cecal perforation was fundamentally the same as that for an appendectomy . the technical skills involved in the procedure are well within the capabilities of practicing general surgeons , but several technical tips merit further comment . in this case , primary repair with intracorporeal suturing was technically possible . however , it seemed ill advised , given the visible zone of coagulation necrosis surrounding the defect . in regards to the surgical stapler , it would have been technically easier to place a single firing across the cecum rather than 2 angled applications ; however , 2 factors suggested multiple stapling : the endoscopically placed clips may have interfered with proper staple line creation , and a single firing raised the concern for luminal compromise . it may be argued that in this case the procedure was effective , but that minimally invasive techniques are not broadly applicable . however , in the analogous situation of perforated appendicitis , there have been multiple studies in which laparoscopy does not have an increased rate of complications over open laparotomy . conceptually , it seems there is little harm in starting with a diagnostic laparoscopy and proceeding based on the nature of the injury and the experience of the surgeon . many general surgeons will be confronted with the problem of colon perforation during colonoscopy . in the case presented here , we modified the established technique of laparoscopic appendectomy to the specific problem of cecal perforation and have demonstrated the feasibility of such an approach .

background and objectives : the unfortunate complication of a colotomy resulting from a colonoscopic polypectomy can be disastrous . using the versatility of laparoscopic surgery , we aim to provide a treatment algorithm for these colonoscopic perforations.methods:we report a case of cecal perforation , in a 70 year old female , following colonoscopic polypectomy that was treated successfully with laparoscopic application of an endo - gia linear stapler.results:four months following staple resection of her cecal perforation , the patient is asymptomatic and has undergone a repeat colonoscopy without any sequelae.conclusions:laparoscopic techniques can be safely applied in the treatment of colonic perforations following therapeutic polypectomies . a suggested management algorithm is provided highlighting the role laparoscopy may provide in selected patients .

bednar tumor or pigmented dermatofibrosarcoma protuberans ( dfsp ) is a rare , aggressive , cutaneous tumor that constitutes 1 - 5% of all dfsps . a 40-year - old female presented with right shoulder swelling present since few months and gradually increasing in size . a complete excision was performed without any prior fine needle aspiration cytology or needle biopsy . the tumor was just beneath the skin , fairly circumscribed , non - capsulated extending into the subcutaneous fat and measuring 5 4 4 cm . grossly , focal areas of pigmentation were noticed towards the periphery of the tumor ( fig . the tumor was a spindle cell lesion abutting the skin and invading the underlying subcutaneous fat . 2 ) . on higher magnification , the spindle cells were uniform appearing with a moderate amount of eosinophillic cytoplasm and plump nuclei with tapering edges . the pigment - laden cells showed coarse brown black pigment obscuring the nuclei and positivity for masson - fontana stain . it is considered to be a tumor of intermediate grade and very infrequently reported in the literature . it is seen in young to middle - aged adults in the fourth decade with very occasional cases in the pediatric age group . the preferred sites are the shoulder region as in our case as well as the trunk , extremities and the head and neck . grossly , the tumors have been described to be multilobular with pigmentation , irregular surface and firm nodules growing deep within the subcutaneous tissue . in this case , the cut surface of the fairly circumscribed tumor showed trabeculations and peculiar pigmentation in the subepidermal portion , which has not been described in the literature . it has to be distinguished from other pigmented cutaneous spindle cell lesions like pigmented neurofibroma , psammomatous melanotic schwannoma , neurocristic cutaneous hamartoma and desmoplastic malignant melanoma . however , our case had classic features of dfsp , and the above lesions were ruled out on histopathology itself . neuro - ectodermal differentiation or melanocytic colonization are the two proposed theories for histogenesis for the bednar tumor . it has also been reported in association of dermal melanocytosis ( nevus of ito ) , and based on the immunohistochemistry , the cell of origin is thought to be a neuromesenchymal cell . bednar tumor can rarely undergo malignant transformation in form of fibrosarcoma with repeated recurrences and distant metastasis . hence , a close follow - up of these cases is always necessary , which is being done in our case . to conclude , though rare , it is important for the histopathologists to be aware and recognize this unusual entity and distinguish it from other pigmented spindle cell lesions . ethics committee approval and patient consent has been obtained . published research complies with the guidelines for human studies and animal welfare regulations . the patient has given informed consent , and the study protocol has been approved by the institute 's committee on human research .

bednar tumor is an uncommon variant of dermatofibrosarcoma protuberans . also known as pigmented dermatofibrosarcoma protuberans , this tumor is of intermediate grade . it is seen in adults and has a predisposition to affect the shoulder region . we report a rare case of bednar tumor in a 40-year - old female patient . the diagnosis of bednar tumor must be considered while reporting pigmented subcutaneous spindle cell lesions .

this clinical case report described a three - month - old mongrel dog that had the urethral orifice opening 3 cm from the tip of the penis and lacked a completely formed preputial sheath . urinary incontinence stopped soon after surgery , suggesting that it had been probably due to an ascending urethritis . hypospadias is a congenital developmental anomaly of the external genitalia in male animals and man , in which the external urethral orifice is on the ventral surface of the penis rather than at the tip of the glans . to a variable extent in individual cases , there is failure of fusion of the urogenital folds and incomplete development of the penile urethra . the urethra may open at any level on the ventral surface of the penis somewhere between the normal location and the ischiatic arch or on the surface of the perineum . in severe cases , lesions such as failure of the two halves of the scrotum to fuse , underdevelopment or absence of the penis , and failure of the urethra to close in the perineal area the urethral meatus may be located along the scrotal raphe . in one report hypospadias was seen in association with unilateral renal agenesis , whilst in another report there was underdevelopment of the penis , fusion failure of the urethra , prepuce and scrotum . other abnormalities associated with hypospadias are retained testicles , bone or anorectal defects , umbilical hernia , hydrocephalus , and urinary incontinence . there are relatively few reports of this condition in the veterinary literature but this may not reflect the true prevalence of the condition . its aetiology is unclear ; it may be due to inadequate production of androgens by the foetal testes or to inadequate numbers of androgen receptors on the urethral folds . this case report describes a three - month - old mongrel dog that had hypospadias , unilateral retained testicle , urinary incontinence , and absence of a fully formed preputial sheath which has not been described before . a three - month - old mongrel dog , weighing 12 kg , was admitted to the veterinary hospital with urinary incontinence and an exposed and dry penis that appeared unsightly ( figure 1 ) . the referring veterinarian had informed the owner that the dog had severe paraphimosis and that the penis could not be readily replaced into the preputial sheath . on physical examination , there was urine scald of the skin on the ventral abdomen and urinary incontinence . the preputial sheath was absent from the bulbus glandis to the tip of the penis ( figure 2 ) but the dorsal mucosa of the incompletely formed sheath was present on the ventral abdominal wall . the dog was anaesthetised using intravenous thiopentone ( 10 mg / kg ) and intubated ; anaesthesia was maintained with halothane and oxygen . a lateral incision was made in the mucocutaneous junction on either side of the midline up to the bulbus glandis . after reconstruction of the preputial sheath , a paramedian celiotomy was performed to locate and to remove the intraabdominal testicle . a small , nodular , vestigial testicle , 0.5 cm in diameter , was found and removed . the animal was kept for observation and it was noticed that urinary incontinence had resolved after 14 days . the surgical wounds had healed and the cosmetic appearance was satisfactory ( figure 3 ) . the prepuce has been reconstructed , leaving a wide preputial orifice . this report describes a case of hypospadias in a three - month - old dog with an incompletely formed preputial sheath . the cause of hypospadias is not known ; it is thought that the affected foetus may secrete inadequate quantities of testosterone or that there may be inadequate conversion of testosterone to dihydrotestosterone in the target tissues of the urogenital sinus and external genitalia . a spectrum occurs from mild hypospadias , in which the urinary orifice is located in the glans penis to severe hypospadias where the orifice is at the penoscrotal junction , scrotum , or the perineum . in the dog , exposure of the male foetus to progesterone or anti - androgens during gestation , especially between day 30 and day 44 of pregnancy , may be responsible . severe genetic defects including cryptorchidism , absence of the scrotum , bifid scrotum , and persistent mllerian structures have been observed in dogs . in many cases in animals and man , surgical correction is usually not attempted because the urethra cranial to the abnormal orifice is deficient . in the present case although the uretheral opening was 3 cm from the tip of the penis , it was patent up to the urinary bladder . one author unsuccessfully attempted to reconstruct the urethra and , subsequently carried out amputation of the penis whilst others recommended the removal of the open prepuce , partial penile amputation and prescrotal or perineal urethrostomy and castration as the treatment for hypospadias . when there are severe urethral defects , excision of the external genitalia and urethrostomy is the treatment of choice . in the present case the preputial sheath was easily reconstructed to cover the exposed penis . it was not necessary to modify the preputial orifice since , after surgery , the preputial opening created was sufficiently large and long enough to allow outflow of urine without scalding the ventral abdominal skin . it is thought that initially the penis appeared dry because of exposure to the air and it was soiled whenever the dog lay down . this must have caused irritation and probable ascending urethritis from bacteria entering through the abnormally located urethral opening . urinary incontinence without neurological deficits is a frequent observation in dogs with hypospadias . in the present case it is presumed that the constant penile irritation and ascending urethritis could have been responsible for the urinary incontinence . in some animals penile and preputial disorders may be associated with pain , incontinence or reluctance to breed . in the present case , the owner was not interested in breeding the dog , and was satisfied with the outcome of surgery . it is concluded that in the less severe cases of hypospadias an attempt should be made to surgically reconstruct the affected parts . although the cause of hypospadias is not known , affected dogs should not be used for breeding purposes .

this clinical case report described a three - month - old mongrel dog that had the urethral orifice opening 3 cm from the tip of the penis and lacked a completely formed preputial sheath . it was presented to the clinic with an exposed penile shaft that was dry . the dog had urinary incontinence that was not of neurological origin . it also had unilateral agenesis of the right testicle . the preputial sheath was successfully reconstructed . urinary incontinence stopped soon after surgery , suggesting that it had been probably due to an ascending urethritis . a large preputial opening was left because of the location of the urethral opening .

however , studies have shown that surgery is best done during normal working hours . given the pressures on theatre use it is important to be able to assess the retinal detachment and to ascertain the urgency of planning surgical intervention . one of the most important features is the involvement of the macula and fovea that is macula on or macula off . in cases of macula - off retinal detachments , macula - on retinal detachments , however , should have their surgery expedited , the main concern being the conversion to a macula - off situation which has a much poorer visual prognosis . the assessment of rhegmatogenous retinal detachments is multifactorial ; in an otherwise normal eye visual acuity is an easy measure of macula involvement with the 6/60 patient being macula off and 6/6 macula on . similarly the onset of symptoms and the age of the retinal detachment is important , as chronic detachments can be more stable and surgery can be safely delayed . also the extent of detachment and position of the retinal break can also help predict the progression of an acute macula on retinal detachment . however , in certain situations the macula - on or macula - off question is not easily answered ; visual acuities may be misleading ; examination of the detachment may be difficult due to poor views often due to vitreous hemorrhage and chronicity may be difficult to ascertain in patients with vague histories . high - speed spectral domain optical coherence tomography ( oct ) offers a noninvasive tool to evaluate retinal microstructural changes in a number of eye pathologies . newer systems using spectral domain calculations have improved data acquisition speeds compared with conventional time - domain oct equipment allowing much greater axial resolution . given the greater resolution a number of characteristic changes seen in retinal detachment have been observed . in this paper , we discuss two cases where spectral domain oct and an understanding of the histological changes have enabled a clearer diagnosis and planning of treatment . our first case is a seventy - five - year - old gentleman who presented with a vague history of blurred vision for six weeks . visual acuity was 6/24 and examination revealed a pseudophakic inferotemporal macula - off retinal detachment . the reduction in visual acuity was thought to be secondary to vitreous haemorrhage as biomicroscopy assessment showed the detachment stopping inferior to the macula ( figure 1 ) . to confirm the macula status , contrary to the biomicroscopy examination ( figure 1 ) , this revealed a macula - off retinal detachment ( figure 2 ) . changes seen in the oct scan were characteristic of an old retinal detachment with the presence of intraretinal cysts , undulation of outer retinal layers , and the hyper - reflectivity in the photoreceptor layer ( figure 2 ) . secondary to these oct findings , the surgical session was deprioritised and performed five days later . the surgical repair consisted of a three - port pars plana vitrectomy with perfluoropropane tamponade and cryotherapy . subsequent spectralis oct one year following the retinal detachment shows restoration of normal retinal morphology with resolution of the intraretinal cysts , flattening of the retinal layers , and no hyperreflectivity seen ( figure 3 ) . our next case was a fifty - year - old myopic female who presented on a friday with a several - month history of floaters and visual distortion described as microstructural analysis of the macula was performed using a heidelberg spectralis oct scan which confirmed a macula - off retinal detachment ; however , the oct scan revealed that the fovea was bisected by this detachment ( figure 4 ) . moreover , the macula microstructure seen in the oct scan showed no retinal folds or hyperreflectivity present near the fovea . indeed , the only morphological retinal detachment changes observed which indicated any chronicity were small intraretinal cysts present peripherally away from the fovea ( figure 4 ) . given the oct findings , she was treated as a macula - on retinal detachment patient , and surgery was expedited such that an emergency theatre session was organised within 24 hours on a saturday morning . the surgical repair was a three - port pars plana vitrectomy using a sulphur hexaflouride tamponade and cryotherapy . after subsequent cataract surgery , vision had returned to 6/6 with normal oct findings ( scan not shown ) . the morphological changes seen in retinal detachment have previously been evaluated by oct and are becoming clearer with newer systems using spectral domain calculations , which have improved data acquisition speeds to ~40 000 a - scans per second allowing much greater axial resolution to approximately 3.5 m tissue resolution . the transformations seen in retinal detachment include intraretinal cyst formation , intraretinal separation , and undulation of outer retinal layers [ 3 , 4 ] . the disruption of the photoreceptor inner and outer segment junction in macula - off rhegmatogenous retinal detachments is also seen both preoperatively and postoperatively [ 6 , 7 ] . murine models comparing histology and oct confirm these findings and also highlight the hyperreflectivity in the photoreceptor layer which may represent a cellular immune infiltration or misalignment of the photoreceptor layer . these changes were all seen in our first case ( figure 2 ) proving that the retinal detachment had been present for a period of time prior to arrival in our unit and enabling appropriate de - prioritisation within a busy vitreoretinal service . in our second case , in which the fovea was bisected by a retinal detachment , time of onset was in some doubt . retinal thickness of the detached retina has been shown to be time dependent initially thickening then thinning with time [ 8 , 9 ] ; however the subfovea thickness was normal when scanned suggesting a recent event along with the absence of any intraretinal cysts , retinal undulations , and hyper reflectivity of the photoreceptor layer ( figure 4 ) . onset of retinal detachment is of importance , as experimental retinal detachments in cats have shown that although alterations in the outer nuclear layer occur after 1 hour , progressive loss of photoreceptors continues up to 1330 days , with limited atrophy in cat retinas detached 3 to 7 days . however , patients that have no tomographic structural changes presumably due to recent foveal involvement have better clinical prognosis . this is most likely secondary to less atrophy and death of the photoreceptors which has histopathologically been shown to be present in prolonged detachment of the retina [ 1014 ] . finally , the height of retinal detachment , which appears to affect the formation of multiple cystic cavities in the detached inner and outer neuronal layers , correlates with poor visual outcome [ 15 , 16 ] . all of these features when taken into account suggested a good prognostic outcome for our second patient and hence prompt surgery resulting in an excellent visual recovery ; an outcome that could have been considerably poorer if surgery had been delayed and fovea atrophy had occurred . the morphological changes in retinal detachment seen in oct scans give prognostic factors pertaining to visual outcome and thus help anticipate surgical outcomes . this paper has shown the two scenarios where surgical prioritisation is reversed , that is , from macula on to macula off and secondly , from macula off to macula on . in our first case , a chronic detachment was identified by oct and allowed planning within the department for higher priority operations to take place . conversely , the lack of subfoveal morphological changes in our second case led to the conclusion that the detachment was recent and prognosis good , thus surgery was expedited . we suggest that if any doubt regarding the status of the macula exists , a routine noninvasive oct should be performed to help clarify the situation prior to surgery .

regmatogenous retinal detachments need prompt intervention particularly when macula is on . unfortunately this is not always easy to ascertain clinically and the chronicity of the event is often muddled in patient 's histories . developments in optical coherence tomography ( oct ) have allowed high - resolution axial scans which have enabled the characterisation of retinal changes in retinal detachments . in this paper , we show the changes in retinal morphology observed by spectral domain oct and how this can be used to plan appropriate surgical intervention .

symptomatic adrenal adenoma usually presents with systemic symptoms . depending on the function of the adenoma , the patient can present with pheochromocytoma - like symptoms ; primary hyperaldosteronism and cushing syndrome ( weight gain , weakness , depression , and bruising ) . a 41 year - old lady presented with multiple metatarsal and phalangeal fractures of the both feet without significant injury . laproscopic r adrenalectomy was performed and histological study confirmed adrenal cortical adenoma with adjacent cortical atrophy suggestive of a functioning adenoma . symptomatic adrenal adenoma usually presents with systemic symptoms . depending on the function of the adenoma , the patient can present with pheochromocytoma - like symptoms ( palpitations , sweating , headache , abdominal pain and labile hypertension ) ; primary hyperaldosteronism ( hypertension and unprovoked hypokalemia ) and cushing syndrome ( weight gain , weakness , depression , and bruising ) . we reported a case of bilateral adrenal adenoma who presented with multiple insufficiency fracture of both feet . a 41 year - old lady presented to us with fracture of the base of her left fifth metatarsal after an inversion sprain ( fig 1 ) . she had sudden onset of left forefoot pain eight months later without any preceding injury . radiographs showed fracture of her left second metatarsal with callus formation ( fig 2 ) . radiographs showed old fractures of bases of bilateral fifth metatarsals , proximal phalanges of bilateral third toes , left second metatarsal with callus formation and crack fracture of the proximal phalanx of the left fourth toe ( fig 3 ) . she was newly diagnosed to have diabetes mellitus but the sensation of the feet was still intact . overnight dexamethasone suppression test ( ondst ) was 660nmol / l which was non suppressible . the serum cortisol reached peak level of 699 in low dose short synacthen test ( ldsst ) and the acth level was less than 1.6 . computed tomogram showed bilateral adrenal adenoma ( fig 4 ) . in order to plan for surgery , the functionality of the adenomas needed to be determined . radiographs showed old fractures of bases of bilateral fifth metatarsals , proximal phalanges of bilateral third toes , left second metatarsal with callus formation and crack fracture of the proximal phalanx of the left fourth toe . multiple metatarsal and phalangeal fractures of the foot of different ages without any significant injury are uncommon . this lady is a housewife and not involved in any activity that can contribute to fatigue fracture . dexa scan was performed as insufficiency fracture was suspected and the result confirmed the presence of osteoporosis . overnight dexamethasone suppression test ( ondst ) , urine cortisol level , low dose short synacthen test ( ldsst ) and the acth level was compatible with adrenal cushing syndrome . adrenal adenoma is a cause of cushing syndrome that is surgically treatable . the presence of adenoma in both adrenal glands needs further study of the functionality of the tumours for surgical planning . histological study confirmed adrenal cortical adenoma with adjacent cortical atrophy suggestive of a functioning adenoma . the clinical significance of this report is that it demonstrates the multiple phalangeal and metatarsal fractures can be the sole presentation of adrenal adenoma . causes of secondary osteoporosis should be investigated in pre - menopausal patient with insufficiency fracture of the feet .

introduction : symptomatic adrenal adenoma usually presents with systemic symptoms . depending on the function of the adenoma , the patient can present with pheochromocytoma - like symptoms ; primary hyperaldosteronism and cushing syndrome ( weight gain , weakness , depression , and bruising).case report : a 41 year - old lady presented with multiple metatarsal and phalangeal fractures of the both feet without significant injury . dexa scan showed evidence of osteoporosis . investigations showed that the picture was compatible with adrenal cushing syndrome . computed tomogram showed bilateral adrenal adenoma . adrenal cortex scintigraphy with np-59 scan showed hyperfunctioning right adrenal adenoma . laproscopic r adrenalectomy was performed and histological study confirmed adrenal cortical adenoma with adjacent cortical atrophy suggestive of a functioning adenoma . post - operatively , she was put on hydrocortisone replacement and recovered well.conclusion:adrenal adenoma can present with insufficiency fractures of the feet .

warfarin is the most commonly used oral anticoagulant both for prophylaxis as well as treatment for arterial and venous thromboembolic conditions . it has a narrow therapeutic index and is metabolized by hepatic cytochrome p450 2c9 enzyme ( cyp2c9 ) . various polymorphisms exist in the gene that code for cyp2c9 , of which * 1 is the wild form expressing normal activity . amongst the variants , * 2 and * 3 are more common than the rest and exhibit lesser enzymatic activity . the anticoagulant effect of warfarin is measured by prothrombin time - international normalized ratio ( pt - inr ) . warfarin exerts its anticoagulant effect by reducing the regeneration of active vitamin k through inhibition of vitamin k epoxide reductase , encoded by vitamin k epoxide reductase complex subunit 1 gene ( vkorc1 ) . increased risk of deranged inr or bleeding has been reported in patients harboring mutant form of either cyp2c9 or vkorc1 . this case report describes a patient on low - dose warfarin , who succumbed to a subdural hematoma and was found to have polymorphic cyp2c9 ( * 1/*3 ) . a 49-year - old male patient , who was a known case of superior sagittal sinus thrombosis for the past 8 years on tablet warfarin 2.5 mg every night , was brought to the hospital unconscious . the patient was non - compliant in not following up with his physician nor did he check his pt - inr levels regularly for the past 1 year . the patient 's relative , however , maintained that the patient was regular in taking the prescribed dose of warfarin ( 2.5 mg ) every night . on physical examination , he was found to be unconscious ( glasgow coma scale score of 3 ) , pulse was feeble and the blood pressure was not recordable . a plain computed tomography ( ct ) brain revealed 24.2 mm hyperdense cresentic subdural hematoma in left fronto - temporo - parieto - occipital region with subfalcine extension and midline shift [ figure 1 ] . his pt - inr was found to be 9 [ normal ( in the absence of anticoagulation therapy ) is 1 and it has to be maintained between 2 and 3 for an appropriate therapeutic effect for warfarin ] . fresh frozen plasma was initiated at a dose of 15 ml / kg / day . blood ( 5 ml ) was collected from the patient and genotyping was done for both cyp2c9 and vkorc1 . cyp2c9 was found to be polymorphic for * 3 ( * 1/*3 ) and vkorc1 was found to be wild - type ( normal ) . plain ct brain revealing 24.2 mm hyperdense cresentic subdural hematoma ( red arrow ) in left fronto - temporo - parieto - occipital region pharmacogenetics is the study of genetic influence on inter - individual variation in the response of a drug . pharmacogenetics has been shown to be of value in detecting an optimal response as well as in preventing adverse reactions following drugs such as warfarin , clopidogrel , and phenytoin . warfarin is metabolized by cyp2c9 and over 30 polymorphic alleles have been identified in the gene coding for cyp2c9 . the wild form of the gene is * 1 and the most common polymorphic forms include * 2 and * 3 . allelic frequency of cyp2c9 * 2 and * 3 in various populations including indians were found to be in the range of 0 - 20% . a recent systematic review concluded that in comparison to the patients with cyp2c9 * 1/*1 genotype , the cyp2c9 * 1/*2 , cyp2c9 * 1/*3 , cyp2c9 * 2/*2 , cyp2c9 * 2/*3 , and cyp2c9 * 3/*3 patients required warfarin doses that were 19.6 , 33.7 , 36.0 , 56.7 , and 78.1% lower , respectively . similarly , in the vkorc1 gene , two alleles have been identified in vkorc1 namely g and a. patients with gg ( wild type ) are resistant requiring a higher dose of the drug whereas aa are sensitive requiring a lesser dose and ga requires intermediate . a randomized clinical trial has also shown that pharmacogenetic - based dosing of warfarin was associated in maintaining the pt - inr levels within the acceptable range for a longer time than by the standard dosing . united states food and drug administration ( fda ) has recently modified the warfarin labeling by suggesting cyp2c9 and vkorc1 genotyping before initiating the drug to ensure warfarin safety . our patient had a fatal subdural hematoma despite low - dose ( 2.5 mg hs ) of warfarin . he had massively deranged inr of 9 , was brought in with glasgow coma scale of 3 . the * 1/*3 variant cyp2c9 explained this serious adverse event with low - dose warfarin . the * 3 variant is less than 5% as active as the wild type whereas * 2 retains around 12% of the activity . a recent indian review of oral anticoagulant use revealed lack of proper laboratory facilities , with irregular pt - inr monitoring in 25% patients and deranged inr values in a high proportion of patients . pharmacogenetic testing needs to be done only once and hence can be considered cost - effective . studies have shown that along with age , gender , body weight , cyp2c9 and vkorc1 polymorphisms account for almost 64% of the variability in the dose of warfarin required for an individual . hence , by doing these genetic testing a priori to the treatment initiation , there is a high possibility that the patients may receive an appropriate dose than just by routine practice . even , several international algorithms for predicting dose of warfarin have been developed with these predicting factors of which , gage 's and wadelius algorithms have been shown to be the most accurate . recently , an algorithm for indian patients has also been developed , which was found to be more accurate , sensitive and significantly reduced the risk of overestimation of dose of warfarin . this algorithm can be used for predicting the appropriate dose of warfarin in our population with the genotype details of cyp2c9 and vkorc1 . to conclude , this case highlights the necessity of adopting the practice of routine genetic testing for cyp2c9 and vkorc1 while initiating warfarin therapy .

warfarin is the most common and cheap oral anticoagulant currently used in clinical practice . a high inter - individual variation is seen in the response to warfarin . recently , pharmacogenetics has gained importance in managing patients on warfarin , both in predicting the optimum required dose as well as in decreasing the risk of bleeding . this case report is a description of a 49-year - old patient who had a lethal subdural hematoma with low - dose warfarin . he was subsequently found to have cyp2c9 gene polymorphism ( * 1/*3 ) . this case report stresses the importance of pre - prescription assessment of genetic analysis for those initiated on warfarin .

spinal epidural abscess ( sea ) is a rare severe infection and accounts for 2.53.0 cases per 10,000 admissions in general hospitals . several sea risk factors , such as injection - drug use , diabetes mellitus , and several illnesses have been identified , however , no report has described sea associated with abortion . we report a case of post - abortion sea caused by the bacteroides fragilis group , which was managed successfully with antibiotics and surgical drainage . a 30-year - old woman was transferred to our hospital for fever and posterior cervical pain . two weeks before admission , dilation and curettage ( d and c ) under intravenous anesthesia was performed for incomplete abortion . after d and c , her obstetrician prescribed ampicillin ( 750 mg / day ) , however , she took them only one day . three days later , she developed posterior cervical pain , which worsened and caused difficulty in flexing and extending the neck . ten days after the operation , she was admitted to the emergency department of another hospital . physical examination revealed body temperature of 39.3c ; blood pressure , 147/91 mmhg ; pulse , 101 beats / min ; and respiratory rate , 18 breaths / min . she experienced severe pain and stiffness in the neck . neither abdominal nor pelvic signs were seen . her leukocytes were elevated to 8100/l and the c - reactive protein ( crp ) was positive . physical examination revealed mild weakness in the right upper limb [ medical research council ( mrc ) grade 4 ] ; deep tendon reflexes were slightly decreased . the liver - function test results and electrolyte levels were normal ; however , leukocytes had elevated to 8400/l , and the crp level had elevated to 9.87 mg / dl . she was seronegative for hepatitis b virus surface antigen , and hepatitis c virus and human immunodeficiency virus ( hiv ) antibodies . analysis showed elevated cell count ( 266/l ) and total protein concentration ( 1,311 mg / dl ) . chest radiography revealed normal results , and a transthoracic echocardiography showed no vegetation on the valves . magnetic resonance imaging ( mri ) of the cervical spine showed infectious spondylodiscitis at the c5/c6 level and anterior epidural abscesses at c4-th3 with spinal cord compression [ figure 1 ] . sagittal t2-weighted image shows an anterior epidural mass associated with c5 spondylodiscitis at the c4-th3 level intravenous injections of ampicillin ( 12 g / day ) and ceftriaxone ( 4 g / day ) , and hyperbaric oxygen therapy were administered . seven days after admission , weakness in the right upper and lower limbs increased ( mrc grade 2 ) and the numbness extended to the right lower limb . mri showed enlarged epidural abscesses and increased spinal cord compression ; therefore , urgent orthopedic surgery was performed . after the anterior discectomy of c5 and c6 , pus was drained . since pus culture revealed b. fragilis group , oral metronidazole ( 1 g / day ) the weakness in her limb muscles were improved ( mrc grade 4 ) , and the numbness gradually resolved after the operation . in most causes of sea , at least one predisposing factor such as intravenous drug use , diabetes mellitus , invasive spinal procedures , penetrating spinal trauma , and immunosuppressive therapy is present ; however , d and c for abortion as one of the risk factors for sea has not reported previously . bacteremia occurs in approximately 5% of the patients who undergo d and c , and progresses to intrauterinal abscess in most cases . however , some forms of extrauterinal abscess after d and c , such as anaerobic breast abscess , septic arthritis and thigh abscess caused by -haemolytic streptococci , psoas abscess caused by staphylococcus aureus and bacterial sacroliliitis and gluteal abscess caused by streptococcus agalactiae have been rarely reported . these manifestations were not associated with any underlying diseases , and most of them eventually required surgical drainage . although the role of antimicrobial prophylaxis in postprocedural pelvic inflammatory disease prevention has been extensively discussed , there is no evidence supporting the use of antibiotics for subacute bacterial endocarditis prophylaxis . a large variety of causative agents for sea have been indentified ; however , two - thirds of the causes of pyogenic bacteremia were caused by staphylococcus aureus , and only 2% of the cases were caused by anaerobic bacteria . the b. fragilis group , which includes organisms such as b. fragilis , b. ovatus , b. thetaiotaomicron , b. uniformis , and b. vulgatus , contains anerobic bacteria . these bacteria can produce a wide variety of infections with a tendency to form abscesses . female genital tract infections caused by these bacteria include bacterial vaginosis , endometritis , and postsurgical obstetric and gynecologic infections . on the other hand , except for extragenital procedures involving d and c , endodonic procedure and vertebroplasy might be one of the risk factors of anerobic spondylodiscitis , which were resulted from hematogenous spreading from a distant primary site of infection . in our case , d and c for incomplete abortion might have caused transient bacteremia resulted in spondylodiscitis and hematogenous sea . micro - organism in vagina might reach cervical discs through hematogenous spreading from pelvic venous plexus ( batson 's plexus ) and cause spondylodiscitis and sea in early days . the patient did not have any risk factors other than the d and c procedure . early imaging could play a crucial role in the treatment of sea , and a careful history taking and physical examination could allow detection of the infectious source . although the use of prophylactic antibiotics in intrauterinal procedures and in cases of incomplete abortion is controversial at present , further clinical trials to assess the necessity of antibiotic prophylaxis after d and c for abortion is necessary should be considered .

spinal epidural abscess ( sea ) is a rare infection complicated in patients who have some risk factors such as injection - drug use , diabetes mellitus , and several illnesses . however , no case of sea associated with abortion has been reported . here we report a case of sea in a 30-year - old woman after dilation and curettage for incomplete abortion . the diagnosis of sea was done by mri and pus was drained after the cervical discectomy . bacteroides fragilis group was cultured from the aspirated pus sample . the patient responded to surgical drainage and antibiotics .

in 2007 , a 14-year - old male killer whale at a marine park in san antonio , texas , usa , died suddenly without notable premonitory signs . on gross examination , mild multifocal meningeal hyperemia and petechial parenchymal hemorrhage focally extensive tan discoloration and fibrosis were present in the right accessory lung lobe with associated hemorrhage and congestion . tissues fixed in 10% buffered formalin were processed routinely and stained with hematoxylin and eosin for histologic examination . inflammatory lesions of the central nervous system were focused in gray matter of the medulla oblongata , pons , mesencephalan , and cerebellum . blood vessels demonstrated mild to moderate acute necrosis and lymphocytic and contained plasmacytic and neutrophilic infiltrates within vascular walls . encephalitis was characterized by perivascular lymphocytes and fewer plasma cells expanding the virchow - robbins spaces . predominant lesions in the lungs were areas of chronic and active abscessation amid a focally extensive area of mixed inflammation and fibrosis . they were characterized by central ulcerations with necrosis and a mixed inflammatory infiltrate surrounded by variable fibrosis and a rim of epithelial hyperplasia . changes in spleen , lymph node , and kidney included acute edema , congestion , and vascular dilation . conventional diagnostic assays were performed for aerobic , anaerobic , and fungal microbes in liver , lung , kidney , cerebrospinal fluid , and brain . the final diagnosis was fulminant peracute bacteremia and septicemia secondary to a primary viral infection associated with nonsuppurative encephalitis . published etiologic considerations for cetacean nonsuppurative encephalitis include morbillivirus and protozoal infections ( 2 ) . a dna microarray with highly conserved sequences from > 1,000 viruses was selected to screen for known and novel viruses ( 3 ) . total rna was extracted from brain tissue and hybridized to a microarray as described ( 4 ) . analysis of the resulting hybridization pattern demonstrated a strong hybridization signal to many oligonucleotide probes on the microarray from the family flaviviridae , in particular to wnv . consensus reverse transcription pcr primers ( 5 ) targeting wnv were used to confirm the microarray results . hq610502 ) yielded a sequence with 99% nt identity and 100% aa acid identity to wnv strain ok03 ( genbank accession no . eu155484.1 ) , a strain originally identified in oklahoma , usa . to further support a wnv diagnosis , we performed immunohistochemical staining on brain tissue . rockville , md , usa ) with peroxidase - tagged goat antirabbit immunoglobulin g ( dakocytomation , carpinteria , ca , usa ) bridge and 3-amino-9-ethylcarbazole ( dakocytomation ) as the chromogen . this staining demonstrated abundant wnv antigen within the cytoplasm of a small number of neurons and glial cells and in fewer macrophages in the brain tissue ( figure ) . brain specimen from killer whale ( orcinus orca ) with west nile virus infection that died at a marine park , san antonio , texas , usa , 2007 . neurons and glial cells demonstrate abundant intracytoplasmic west nile viras antigen . we evaluated wnv exposure within the same cohort , as well as a geographically distant cohort of whales by using serologic testing . all testing was performed at the same laboratory by using a standard plaque - reduction neutralization test . in this assay , a 90% neutralization cutoff was used ( 6 ) . a 90% plaque - reduction titer serum from the affected whale and 5 cohort killer whales from the same marine park in san antonio as well as 5 whales housed at another facility in orlando , florida , usa , were evaluated . in each facility , the animals have regular contact with each other . the facilities are geographically separated so the animals do not have exposure to those in the other park . all 6 animals from texas had 90% plaque - reduction titers > 10 , ranging from 40 to 80 . these findings broaden the known host tropism of wnv to include cetaceans in addition to previously known pinnipeds . although we can not definitively attribute the cause of death of this whale to wnv , the observed lesions are consistent with those caused by wnv in other animals . the serologic results demonstrate that subclinical infections can occur and that exposure can be variable . both bexar county , texas , and orange county , florida , have had wnv in wildlife since 2002 . mosquito management practices are similar in both facilities and have been expanded since this diagnosis . differences in wnv prevalence or mosquito numbers may have played a role in the different serologic results . health evaluations of free - ranging and captive cetaceans should include wnv serology to assess exposure rates . this report focuses on killer whales , but the loafing behavior ( stationary positioning at the water s surface ) is commonly seen in many coastal dolphins , thereby increasing the likelihood of mosquito bites and exposure to wnv . serologic screening of bottlenose dolphins ( tursiops truncatus ) from the indian river lagoon demonstrated wnv titers ( 7 ) . as with many species of birds and mammals , wnv infection carries a risk for zoonotic transmission . until the implications of this infection in marine mammals are better understood , biologists and veterinarians working with cetaceans should consider this possibility . potential viral shedding can occur through the oropharygeal cavity and feces as well as through blood and organs during necropsies . finally , our study demonstrates the broad applicability of using panviral microarray - based diagnostics . even though pcr diagnostics are well developed for wnv , the agent was not initially considered as a potential pathogen in this species . panviral microarray can be used not only to identify novel viruses but also to detect unsuspected agents .

in 2007 , nonsuppurative encephalitis was identified in a killer whale at a texas , usa , marine park . panviral dna microarray of brain tissue suggested west nile virus ( wnv ) ; wnv was confirmed by reverse transcription pcr and sequencing . immunohistochemistry demonstrated wnv antigen within neurons . wnv should be considered in cases of encephalitis in cetaceans .

gastrointestinal stromal tumors ( gist ) represent less than 1% of all primary tumors of the gastrointestinal tract ( git ) and commonly affect patients in the middle and older age groups with equal gender distribution . . their sub mucosal location can produce local obstructive symptoms , particularly when arising in the esophagus or the small intestine . most gists usually present with vague upper abdominal pain , fullness or gastrointestinal ( gi ) bleeding . sometimes they are found incidentally during barium studies , endoscopy or abdominal scans performed for other reasons . a 40-year - old man presented with a history of rectal bleeding for two days , with a reported loss of more than 1 liter of fresh blood . despite intensive fluid management with whole blood and normal saline , colonoscopy was not attempted because of continuous bleeding . tc-99 m labeled red blood cell ( rbc ) scintigraphy [ figure 1 ] showed accumulation and movement of tracer in the mid - abdomen , corresponding to the region of the jejunal loops . a diagnosis of active gastro - intestinal bleed ( likely to be from the jejunum ) was made , and the patient taken up for exploratory laparotomy . a polypoidal exophytic mass about 4 cm in size was found in the jejunal wall , 40 cm from the duodeno - jejunal junction . the mass along with the adjoining jejunal loops was resected , followed by end - to - end anastomosis . gross examination of the specimen [ figure 2a and b ] showed a polypoidal mass ( measuring 3.2 cm in its largest dimension ) arising from the serosal aspect , producing a bulge in the central part of the mucosa , which was ulcerated and could be the possible source of bleeding . microscopic examination [ figure 2c ] showed a well - circumscribed tumor in the submucosa . the mass showed a spindle cell tumor arranged in long interlacing fascicles with characteristic perivascular arrangement of tumor cells . the tumor cells were moderately pleomorphic with mitotic rate of 2/50 high power field ( hpf ) . following surgery , the patient made a satisfactory recovery and was discharged on the seventh post - operative day . dynamic scintigraphy using tc-99 m labelled red blood cells showing accumulation and movement of tracer in the mid - abdomen ( arrows ) the resected specimen from the jejunum ( a and b ) a polypoidal mass arising from the serosal aspect , with an ulcerated bulge in the central part of the mucosa ( c ) microscopic examination shows a spindle cell tumor arranged in long interlacing fascicles with perivascular arrangement of tumor cells ( d ) immunoperoxidase staining showing cd-117 positivity gists are derived from cajal cells or their precursors and most commonly occur after the age of 50 years in the stomach ( 60% ) , jejunum and ileum ( 30% ) , duodenum ( 4 - 5% ) , rectum ( 4% ) , colon and appendix ( 1 - 2% ) , and esophagus ( < 1% ) , rarely as apparent primary extra - gastrointestinal tumors in the vicinity of the stomach or intestines . the role of labeled rbc scintigraphy in acute gastro - intestinal bleeding is well - established and its application in rare cases has also been reported . however , few authors have discussed the role of tc-99 m rbc scintigraphy in discovering a bleeding gist . surgery is the primary treatment of choice for patients with localized or potentially resectable gist lesions and is recommended if bleeding is present . the scottish intercollegiate guidelines network ( sign ) recommends that nuclear scintigraphy should be considered to assist in localization of bleeding in patients with significant recent lower gi hemorrhage . although colonoscopy is the initial diagnostic modality of choice to localize the bleeding site for patients presenting with acute severe hematochezia , it is difficult when patients present with profuse bleeding . in these situations arteriography or labeled rbc scintigraphy is warranted . as a non - invasive investigation that requires no specific patient preparation , there is also no need of any iodinated contrast that may cause contrast - nephropathy in acutely ill - patients . since the risk of malignancy was low in our patient , he has been kept under follow - up with no adjuvant treatment . this report highlights the advantage of this non - invasive investigation in hemodynamically unstable patients by detecting and localizing the site of an active gi bleed .

the role of 99m - technetium labeled red blood cell ( rbc ) scintigraphy in acute gastro - intestinal bleed is well - established . the authors report a case of a bleeding gastrointestinal stromal tumor ( gist ) incidentally discovered on tc-99 m rbc scintigraphy .

retinitis pigmentosa ( rp ) was defined as a group of hereditary disorders that diffusely involve photoreceptor and pigment epithelial function characterized by progressive visual field loss and abnormal electroretinogram . rp also has changes at vitreoretinal interface , such as vitreoretinal traction and preretinal membrane , which causes macular disorders such as cystoids macular edema and macular hole . we report a case of vitreoretinal traction syndrome associated with rp , which spontaneously resolved after posterior vitreous detachment ( pvd ) . a 81-year - old woman presented with bilateral loss of peripheral vision and defective dark adaptation 15 years before . the best - corrected visual acuity was 20/28 and 20/28 in the right and left eyes , respectively . slit - lamp examination showed bilateral retinal arteriolar narrowing and intraretinal pigment deposition ( bone spicule pigmentation ) in both eyes ( figure 1a ) . the visual fields of the patient were markedly constricted to the central 10 in both eyes ( figure 2 ) . on june 2 , 2006 , retinal detachment was detected in the nasal periphery in the right eye ( figures 1b and 1c ) . at that time , pvd was confirmed with weiss ring ( figure 3 ) . fluorescein angiography showed a hyperfluorescence due to atrophy of retinal pigment epithelium but no active leakage in the detached area ( figure 1c ) . the detached area was within the area where the visual field was lost , and we observed a clinical course of this eye without any treatment . retinal detachment was kept localized and not extended . five months later , pvd further progressed beyond the arcade area , accompanying retinal hemorrhage around the detached retina . in this case , an eye with rp was complicated with tractional retinal detachment , possibly associated with increased vitreoretinal traction secondary to the incidence of pvd . the retinal detachment was limited within the area where the visual field was lost and was not progressive . eight months later , spontaneous resolution of tractional retinal detachment resulted from progression of pvd , which was suggested by transiently increased traction - related retinal hemorrhage . eyes with rp may have abnormal liquefaction of vitreous and altered distribution of vitreoretinal adhesion . the incidence of pvd in the posterior pole might result in the focal remarkable traction in the peripheral retina and tractional retinal detachment . eight months later , the retinal detachment was resolved because local vitreoretinal traction was weakened with extention of pvd beyond the arcade vessels . rp has vitreomacular traction - related pathologic events involving vitreomacular traction syndrome , cystoids macular edema , and macular hole.13 such changes in this patient with rp might result in a tractional retinal detachment . rani et al4 reported that a 50-year - old man underwent a tractional retinal detachment involving the posterior pole with a taut posterior hyaloids surface . in this study , vitrectomy was not performed because the patient opted for no surgical intervention in view of poor prognosis . on the other hand , it was reported that vitrectomy was effective for vitreous opacity,5 cystoid macular edema,6 exudative retinal detachment,7 and macular holes8 in rp . however , regarding the indication to perform a vitrectomy , the balance of the expected gain in visual function and usual risks of vitrectomy and rp - related risks , such as phototoxicity worsening of visual field loss , and cystoid macular edema . it might be a choice that a patient with tractional retinal detachment in rp is observed without surgical treatment , if the detached area is not progressive .

we present a case of retinitis pigmentosa with vitreoretinal traction - associated retinal detachment . the retinal detachment was detected in the nasal periphery . no retinal breaks and no active vascular leakage were observed by fundus scopy and fluorescein angiography , respectively . however , 8 months later , the tractional retinal detachment was spontaneously resolved with posterior vitreous detachment .

metastases to the breast from extramammary malignancies are rare and account for 0.43% of all breast malignancies . lymphoma , melanoma , sarcoma , lung carcinoma and ovarian tumor are common extramammary primary malignancies that metastasize to the breast [ 1 - 3 ] . cases of metastasis from primary colorectal neoplasm to the breast is extremely rare . in searching medline and embase including non - english literature , and handsearching the references , we describe the features of metastatic breast cancer from adenocarcinoma of the colon in a 63-year - old woman . a 63-year - old woman was referred to a breast surgeon due to a mass with suspicious metastatic axillary lymph node discovered incidentally on positron emission tomography - computed tomography for follow - up study after colon cancer surgery without other metastatic lesion ( fig . four years ago , she underwent a subtotal colectomy for stage t3n1m0 sigmoid colon cancer with colonic obstruction and subsequent chemotherapy with 5-fluorouracil and leucovorine . two years after the operation , a 2.8 cm sized solitary metastatic lung nodule on the right middle lobe was identified on chest computed tomography for follow - up study for which she underwent pulmonary wedge resection , followed by chemotherapy with oxaliplatin and 5-fluorouracil . physical examination revealed a 2 cm sized firm nodule in the lower inner quadrant of the right breast without evidence of axillary or supraclavicular lymphadenopathy . the mass was not identified on mammography , but ultrasonography showed 1.1 cm sized mass in the lower inner quadrant of the right breast without enlarged lymph node in the ipsilateral axilla . serum tumor markers were all in normal ranges ( carcinoembryonic antigen 4.2 ng / ml , carbohydrate antigen 15 - 3 6.2 u / ml ) . subsequent magnetic resonance imaging showed 1.8 cm sized ill - defined irregular shaped enhancing mass in the lower inner quadrant of the right breast and enlarged lymph node at the ipsilateral axilla compatible with a nodule suspicious for metastasis . breast specific gamma imaging using tc - mibi showed a lesion with increased uptake in the lower inner portion of the right breast with no lesion uptake in the axilla . the tumor cells in the breast mass were cuboidal to columnar with eosinophilic cytoplasm and pseudostratification , compatible with the features of adenocarcinoma of colorectal origin ( fig . immunohistochemical study revealed the following results : tumor cells were strongly positive for cdx2 , cytokeratin ( ck)20 and negative for ck7 , thyroid transcription factor-1 , estrogen receptor , progesterone receptor , c - erbb2 ( fig . 4 ) . these findings are consistent with metastatic adenocarcinoma of the colon or rectum . primary breast cancer is one of the most common malignancies and leading cause of death from malignant disease in women . a malignant mass in the breast can easily be misinterpreted as breast cancer and inappropriate surgical intervention could be carried out . although there are few instances of breast metastasis from colorectal malignancy , correct diagnosis is crucial to avoid unnecessary surgical intervention . generally , metastases to the breast from extramammary malignancies are characterized by rapidly growing , mobile masses that are easily palpable but do not cause overlying skin or nipple retraction , or bloody nipple discharge . core biopsy allows histological assessment of the tissue , helping in identifying the origin of the carcinoma . on the histologic finding , the periductal and perilobular location of the tumor with the absence of in situ ductal carcinoma in the surrounding breast specimen favor a metastasis . immunohistochemistry enables a more confident differentiation between a primary breast adenocarcinoma and a colorectal adenocarcinoma metastasis to the breast . for our patient , diagnosis was finally reached after immunohistochemistry finding . the majority of colorectal carcinomas are usually ck7-negative and ck20-positive , while the majority of primary breast carcinomas is ck7-positive and ck20-negative . and the positive immunostaining for cdx2 is a highly sensitive and specific marker of colorectal carcinoma . our case showed these findings as well ; tumor cells were positive for cdx2 , ck20 and negative for ck7 . recently , some authors have suggested that the expression of ck20 can be identified in primary breast cancer in contrast to the conventional immunohistochemistry results . so , clinicians and pathologists should examine multiple immunohistochemistry markers and combine them as an approach for final diagnosis . according to recent reports , the management of metastatic breast mass from colorectal adenocarcinoma should be diagnostic and palliative . advocated that surgical excision should be avoided in the view of short life expectancy and risk of seeding to the skin . suggested that excisional biopsy was usually appropriate and provided adequate local control . if the diagnosis for the breast mass is unclear , unnecessary intervention , for example mastectomy or lymph node dissection of axilla , can be performed . therefore , when breast mass is identified incidentally on radiologic study during follow - up after operation for colorectal carcinoma , clinicians should keep the possibility of breast metastasis from colorectal adenocarcinoma in mind . additionally , periodical examination of the breast after colorectal cancer surgery may be helpful to detect the metastasis early .

a 63-year - old woman was referred to a breast surgeon with a breast mass discovered incidentally during follow - up study after colon cancer surgery . invasive adenocarcinoma was revealed on core needle biopsy . wide excision of the breast including the tumor was performed . on standard histological examination the tumor showed features of moderately differentiated adenocarcinoma . the immunohistochemistry study revealed positive results for cytokeratin ( ck)20 and cdx2 , but negative for ck7 . these are typical characteristics for colon cancer . considering her history of subtotal colectomy for sigmoid colon cancer , it is presumable that the mass in the breast was of colonic origin , and it was an extremely rare case of metastasis to the breast from primary colorectal neoplasm . although the instance is rare , clinicians should keep the possibility of breast metastasis from colorectal cancer in mind for early and correct diagnosis .

a 41-year - old woman with a history of liver cirrhosis presented with recurrent portal hypertension and bleeding from esophageal varices . three months prior to this admission , a tips was created with a 10 mm 7 cm wallgraft ( boston scientific , natick , ma ) . however , the stent 's anticoagulation was inadequate ( international normalized ratio [ inr ] : 1.02 - 1.21 ) after placement . the initial attempts at recanalization of the completely occluded stent via the transjugular approach were unsuccessful . therefore , a percutaneous transsplenic approach guided by ultrasound was used to gain entry to the splenic vein and portal vein for catheterization of the occluded stent . to perform the ultrasound guided puncture of the splenic vein through the spleen , we used a skater introducer set ( angiotech , pbn medicals , denmark ) . a 22-guage chiba needle was introduced into the splenic vein , then a 0.018 inch guide wire was placed through the chiba needle into the splenic vein , and a 6-fr dilator system was placed over the wire . we then exchanged the 0.018 inch guide wire with a 0.035 inch guide wire , followed by a 5-fr introducer sheath and a 4-fr kmp catheter ( cook medical inc . , however , residual thrombosis of the stent was noted in combination with occlusion of the hepatic vein ( fig . so , we performed catheter - directed intrathrombus thrombolysis with urokinase ( taiwan green cross co. , taipei , taiwan ) . in brief , the distal end of a 4 fr rc1 catheter ( cook medical inc . , bloomington , in ) with home - made side holes ( n = 10 , made by needle - sticks into the distal 5-cm segment of the catheter ) was wedged into the thrombosed tips stent . then , catheter - directed intrathrombus thrombolysis with a solution of 48 vials of urokinase ( 60,000 u / vial ) in 500 ml normal saline at a drip rate of 21 ml / hr ( i.e. , 2 vials / hr = 2,000 u recanalization was performed in the following day and a wallstent endoprosthesis ( 14 mm 9 cm , boston scientific , natick , ma ) was placed , resulting in the successful opening of the shunt ( fig . the catheters were removed and the transsplenic tract was embolized with 4 serial coils ( mwce-35 - 8/4-tornado , cook medical inc . , she is doing well five months after discharge and an ultrasound study indicates that the stent remains patent . transjugular intrahepatic portosystemic shunt is now considered the procedure of choice for accessing the portal system for management of refractory variceal bleeding . certain complications , including a high reintervention rate due to occlusion of the stent , have raised concerns regarding its application ( 2 , 3 ) . yet recent reports have suggested that using a covered prosthesis improves the tips patency to longer than two years ( 4 , 5 ) . although a covered stent had been employed in the patient presented here , thrombosis occurred early ( i.e. , within 3 months ) . transjugular intrahepatic portosystemic shunt failure requires recanalization of the stent or placement of serial or parallel stents ( 6 , 7 ) . neointimal hyperplasia at the ends of the stent may completely occlude the outflow hepatic venous tract ( 1 ) and this can make reintervention difficult . as in this case , an alternative route is needed when attempts at recanalization via a conventional transjugular approach fail . the splenic vein drains directly to the portal vein , and its size and straight route make this approach an ideal option ( 5 , 8) . the transsplenic approach has been used for other indications , as reviewed by tuite et al . ( 8) , but we are unaware of any other report of using the percutaneous transsplenic approach to recanalize an occluded tips stent . additionally , we believe that the overnight infusion of a thrombolytic agent contributed to successful recanalization in our case ( 9 ) . concerns regarding post - procedural splenic bleeding and the reported difficulty some clinicians have experienced negotiating the pathway have limited the use of the transsplenic route . a cautious , image - guided approach reduces the risk of splenic hemorrhage ( 5 , 8) and obliteration of the transsplenic tract with coils upon completion of the procedure also assists in preventing post - procedural bleeding ( 8) . no significant splenic bleeding occurred in our patient , despite her liver impairment , during or following the intervention . subtherapeutic anticoagulation ( inr < 2 ) probably contributed to the thrombosis and subsequent occlusion in this patient . there is no routine anticoagulation protocol to follow for tips patients . we prescribe anticoagulation for only the patients with tips dysfunction ; however , there is no reference available for the effectiveness of this regime . as a general rule for preventing thromboembolism , chronic oral anticoagulant therapy with a vitamin k antagonist is administered and the dose is adjusted to achieve an inr of 2.0 to 3.0 ( 10 ) . in summary , we have reported on a rare case of using the transsplenic approach for recanalization of a tips stent by thrombolysis . transsplenic entry to the portal system is useful in recanalizing a stent that is difficult to reach through the usual transjugular route .

a 41-year - old woman with liver cirrhosis had recurrent portal hypertension and bleeding from esophageal varices due to complete occlusion of a previously inserted transjugular intrahepatic portosystemic shunt stent . because recanalization of the stent by the transjugular approach was unsuccessful , ultrasound - guided entry to the splenic vein and portal vein was used . after catheter - directed intrathrombus thrombolysis , successful opening of the stent was achieved and a stent was placed . we herein report a rare case in which thrombolysis and recanalization of a tips stent were performed via a percutaneous transhepatic approach .

in the current issue of critical care , de prost and colleagues evaluated the impact of protective and injurious ventilation strategies on lung neutrophil distribution and activation in sheep 2 hours after endotoxin infusion . the protective ventilation strategy consisted of 8 ml / kg tidal volume and titration of positive end - expiratory pressure ( peep ) to achieve a plateau pressure of 30 cm h2o . peep was not applied in the injurious ventilation protocol , and tidal volumes of 14 to 18 ml / kg were delivered . positron emission tomography ( pet ) imaging and n-[nitrogen]-saline infusion were performed to evaluate regional lung perfusion and shunt fraction . cellular metabolic activities were measured by using an f - fluorodeoxyglucose ( f - fdg ) infusion protocol , and lung leukocyte infiltration was determined by histological analysis . their results show that protective ventilation was associated with better gas exchange and lower shunt fraction in dependent lung regions prior to endotoxin infusion . endotoxin infusion worsened gas exchange in both groups , but less so in sheep receiving protective ventilation . protective ventilation also attenuated f - fdg uptake and phosphorylation after endotoxin infusion , particularly in dependent areas of the lungs . the authors conclude that a protective ventilation strategy that optimizes alveolar recruitment and minimizes alveolar distension may mitigate neutrophil activation in the lung , particularly in dependent areas , during early experimental acute lung injury ( ali ) . use of the sheep model in this study had several advantages . with a size comparable to that of the human , the ovine model allowed an analysis of physiological endpoints that are clinically relevant and permitted the application of ventilation strategies that are similar to those used in clinical practice . the tidal volumes and peep applied in the protective strategy are similar to those advocated for use in the clinical setting . alternatively , a study by takeuchi and colleagues described a method of pressure - volume curve analysis to identify the most appropriate peep during mechanical ventilation in an adult sheep model of ali , which may further optimize lung protection and would be interesting to apply in the model used by de prost and colleagues . the endotoxin infusion model has the advantage of being reproducible and easily titrated and is known to induce neutrophil accumulation in the lungs . however , the protocol appears to have induced minimal histological evidence of lung injury as indicated by lung injury scores of 0 to 1 ( on a scale of 0 to 4 ) in both groups . given the lack of demonstrable lung injury at the histological level , it is possible that differences in normal lung recruitment and hemodynamics contributed to the dissimilarities in gas exchange and shunt fraction observed between groups . however , changes in alveolar - capillary integrity that were not detectable by light microscopy may also be present early after endotoxin infusion and could have contributed to the observed gas exchange perturbations . evidence indicates that dysregulated inflammation and the inappropriate accumulation and activation of leukocytes , especially neutrophils , contribute to the pathogenesis of ali . furthermore , investigators have postulated that protective ventilation strategies decrease regional lung inflammation in subjects with ali . the assessment of cellular metabolic activity by using pet imaging and f - fdg infusion along with the evaluation of lung perfusion and ventilation , as performed by de prost and colleagues , is an informative , non - invasive approach that provides useful data regarding regional differences in cellular metabolic rate . the technique may have practical utility since studies have shown that evaluation of f - fdg uptake may be valuable in predicting respiratory failure and evaluating therapy in clinical and experimental models of ali [ 8 - 10 ] . the present study extends previous reports by documenting the impact of ventilation strategies on regional metabolic activity and neutrophil accumulation early during the course of ali . although differences in cellular metabolic activity were observed between groups , neutrophil accumulation in the lungs was not different when sheep receiving protective or injurious ventilation were compared . the authors interpret that finding as possibly being indicative of increased neutrophil activation in sheep receiving injurious ventilation . that conclusion is based , in part , on previous studies that showed neutrophil activation to be the primary factor contributing to increased f - fdg uptake and phosphorylation during ali . however , as noted by the authors , it is unclear whether the alterations in f - fdg uptake and phosphorylation observed in their analysis are entirely specific for neutrophils . it is possible that the metabolic rates of other leukocyte and non - leukocyte cell populations were affected . evidence indicates that alterations in macrophage , epithelial , and endothelial cell functions occur during ali . further studies are needed to identify the cell populations that are affected and the functional importance of the observed alterations . overall , the study by de prost and colleagues provides new insights into the impact of ventilation strategies on regional cellular metabolic activity during early ali . more investigation is needed to extrapolate their findings into the clinical setting and determine the functional importance of their findings . it is hoped that the group will perform follow - up studies to determine whether the observed changes in regional leukocyte activation are predictive of progressive respiratory failure and pulmonary injury as well as better define the specific cell populations involved . 18f - fdg : f - fluorodeoxyglucose ; ali : acute lung injury ; peep : positive end - expiratory pressure ; pet : positron emission tomography .

protective mechanical ventilation is currently accepted as a key strategy for the management of acute lung injury ( ali ) and its most severe form , acute respiratory distress syndrome . the study by de prost and colleagues in the current issue of critical care provides new insights into the impact of ventilation strategies on pulmonary function , gas exchange , and regional cellular metabolic activity during early ali in sheep . the group reports that a protective ventilation strategy may attenuate neutrophil activation in dependent lung regions during early experimental ali . this is an innovative report that provides the basis for further study .

in this issue of critical care , lesur and colleagues report the differential profile of stress response in septic and non - septic patients . adrenocorticotropic hormone ( acth ) and acth / cortisol ratio were lower whereas baseline cortisol , procalcitonin ( pct ) , and stromal cellderived factor-1-alpha ( sdf-1 ) were higher in septic patients than in non - septic patients . furthermore , a probability score incorporating acth , cortisol , and pct by multivariate logistic regression analysis predicted sepsis better than sepsis score or pct did . the response of hypothalamic - pituitary - adrenal ( hpa ) axis to the sustained stress of severe illness has been the focus of many studies in recent years . in healthy subjects , cortisol secretion by adrenal cortex is regulated by acth secretion by the pituitary , which in turn is regulated primarily by hypothalamic secretion of corticotropinreleasing hormone ( crh ) , whereas cortisol inhibits both acth and crh production through a negative feedback [ 2 - 4 ] . however , hpa stress response during sepsis is much more complex and is poorly defined . plasma cortisol levels may be low , normal , or high in sepsis but nonetheless inadequate to control the inflammatory response and meet the elevated metabolic demand . this effect is termed relative adrenal insufficiency ( rai ) , also known as critical illness - related corticosteroid insufficiency ( circi ) . other factors are involved in the hpa stress response during sepsis . in rodent models , arginine vasopressin ( avp ) was shown to increase endogenous adrenal acth secretion . apelin , a neuropeptide originating from paraventricular and supraopitc nuclei , acts on hpa axis regulation by releasing crh and acth and by reducing avp . copeptin , a 39-amino acid glycopeptide , makes up the pre - pro - vasopressin molecule together with neurophysin ii and avp and serves as a surrogate marker to assess avp plasma concentrations in septic shock . in normal rats , the chemokine sdf-1 and its receptor colocalize with avp in magnocellular neurosecreatory neurons , resulting in an inhibition of avp - induced release . the dissociation of acth and cortisol levels in late phase ( lasting many days to weeks ) , which is different from that of the acute phase ( hours to a few days ) of an illness , indicates that alternative pathways not mediated by acth are involved . limited clinical studies prove that the dehydroepiandrosterone ( dhea ) level is very low in septic shock , whereas its sulphate and the cortisol / dhea ratio might be prognostic markers and signs of exhausted adrenal reserve in critical illness . tissue resistance to corticosteroid action may also play an important role in sepsis and can be caused by either defects in the corticosteroid receptor or postreceptor alterations and may not be defined accurately based on plasma cortisol levels . despite the uncertainty of the definition and diagnostic criteria , clinical studies show that patients with rai are at a significantly higher risk of hospital mortality and this has been the driver for corticosteroid replacement therapy in severe sepsis / septic shock [ 2 - 4 ] . this is not surprising upon review of the aforementioned complexity and unknowns of hpa stress response . furthermore , it must be acknowledged that the decision to treat with stress - dose corticosteroids is based on clinical criteria rather than on the inconclusive results of adrenal function tests . the dissociation of acth and cortisol levels observed in the study is more compatible with neuroendocrine characteristics of prolonged critical illness , although the authors claimed to include patients within the first 24 hours of admission . the clinical significance of the predictive model is hindered by the unavailability of acth or cortisol measurements at the bedside and by the fact that the predictive value of sepsis score or pct has not been consistently validated in clinical trials . despite all of these limitations , the study by lesur and colleagues undoubtedly expands our understanding of the complex neuroendocrine network regulating hpa stress response in human sepsis . we believe that further investigation into the mechanism is warranted before we plan a successful strategy for corticosteroid replacement in sepsis . acth : adrenocorticotropic hormone ; avp : arginine vasopressin ; circi : critical illness - related corticosteroid insufficiency ; crh : corticotropin - releasing hormone ; dhea : dehydroepiandrosterone ; hpa : hypothalamic - pituitaryadrenal ; pct : procalcitonin ; rai : relative adrenal insufficiency ; sdf-1 : stromal cell - derived factor-1-alpha .

the hypothalamic - pituitary - adrenal ( hpa ) axis response in sepsis remains to be elucidated . apart from corticotropin - releasing hormone , adrenocorticotropic hormone , and cortisol , many other neuroendocrine factors participate in the regulation of hpa stress response . the hpa response to acute and chronic illness exerts a biphasic profile . tissue corticosteroid resistance may also play an important role . all of these add to the complexity of the concept of relative adrenal insufficiency ' and may account for the difficulty of clinical diagnosis and for the conflicting results of corticosteroid replacement therapy in severe sepsis / septic shock . the study by lesur and colleagues expands our understanding of the mechanism , and further study of hpa stress response is warranted .

a 65-year - old man without any symptoms was referred for an operation on the aortic root and ascending aortic aneurysm . he had undergone a total correction of tetralogy of fallot ( tof ) and aortic valve replacement with mechanical valvular prosthesis 22 years earlier ( in 1987 , at the age of forty - three ) . in august 2009 , a huge aneurysmal dilatation ( 9 cm ) of aortic root and proximal ascending aorta , severe tricuspid regurgitation , and good left ventricular ejection fraction were found on echocardiography . computed tomography showed a huge saccular aneurysm of the aortic root and proximal ascending thoracic aorta ( fig . 1 ) . the patient underwent a bentall procedure involving a composite conduit with a mechanical valvular prosthesis . the operation also included pulmonary valve replacement using a bioprosthesis , tricuspid annuloplasty , subaortic pannus resection , maze procedure , and coronary artery bypass surgery ( saphenous vein to proximal right coronary artery ) . because the right coronary artery orifice was deviated to the left side and was close to the aortic annulus , coronary artery bypass surgery was chosen despite availability of the button technique . after the operation , there was the microscopic finding of cystic medial degeneration in the ascending aortic wall ( fig . he was discharged on his 37th postoperative day . at follow - up , he remains well 17 months after this latest procedure . in 1997 , dodds and colleagues described the first series report about progressive aortic regurgitation and aortic root dilatation after complete , uncomplicated repair of tof . a long - standing volume overload of the aortic root may cause aortic root dilatation in adults with a repaired tof . the right to left shunt through the ventricular septal defect in unrepaired tof increases the volume overload effect on the aortic root . another causative mechanism for progressive aortic root dilatation intrinsic histological changes were found in tof patients ; including medionecrosis , fibrosis , cystic medial necrosis , elastic fragmentation and elastic lamellae disruption . our patient had initial reparative surgery at the age of forty - three in 1987 . the phrase of the enlarged aorta was described in the record of the first operation . , we did not know why correction was not performed in the initial operation . in 2002 , an operation for ascending aortic aneurysm was recommended , but he refused any surgery at that time . the pathologic conditions of aortic root and ascending aorta in repaired tof are no longer a benign problem . in recent reports , these are the first reports of aortic dissection in tetralogy of fallot . after an initial curative operation for tof , meticulous monitoring of aortic root and ascending

surgical repair of the tetralogy of fallot is one of the most successful operations in the treatment of congenital heart diseases . we report the case of a 65-year - old man who had an aortic valve replacement at the time of complete repair of the tetralogy of fallot at the age of forty - three . he subsequently had progressive aortic root and ascending aorta dilation to 9 cm . the aortic root and ascending aorta replacement was done using a composite valve - graft and was performed along with other procedures . thus , meticulous follow - up of aortic root and ascending aorta after corrective surgery for tetralogy of fallot is recommended following initial curative surgery .

the spinocerebellar ataxias ( scas ) are a group of genetically heterogeneous neurodegenerative disorders characterized by cerebellar , pyramidal , extrapyramidal , cognitive , and peripheral nerve dysfunction in variable combination . dystonia can be encountered in a small subset of patients with sca , but task - specific dystonia ( writer 's cramp ) is extremely rare and has been reported only in association with stray cases of sca types 6 , 7 , and 14 worldwide . we report a rare case of genetically confirmed sca type 1 ( sca1 ) with a prominent and disabling writer 's cramp . a 48-year - old , nonalcoholic , male police officer , presented to us with 2 years history of very slowly progressive dysarthria and a mild gait imbalance noticed only during running and marching . for the past 4 months , he also complained of an abnormal posturing and stiffness of his right hand while writing . there was no abnormal posturing of the hand , or any other part of the body while carrying out any other activities of daily living . there was no history of any motor or sensory deficit or incoordination of the upper limbs . his history was insignificant and there was no history of a similar illness in his siblings or other family members . on examination , his higher mental functions were preserved and cranial nerves were intact . on motor examination , there was no nystagmus , but a slight slowing of the horizontal and vertical saccades was observed . there was no incoordination in the upper limbs , but the heel - shin test was slightly impaired . a task - specific dystonia in the form of a writer 's cramp with lifting of the right index finger off the pen and curling of the thumb and the last two digits was documented while writing . there was no evidence of dystonia at rest or extrapyramidal manifestations such as bradykinesia , rigidity , or tremor in any other part of the body . in view of the history of a very mild , slowly progressive , cerebellar ataxia of long duration , slow saccades , hyperreflexia and a prominent writer 's cramp , a possibility of inherited sca with task - specific dystonia was kept . his routine investigations including hemogram , blood sugar , liver , renal , thyroid function tests , and serum vitamin b12 levels were all normal . he tested negative for antibodies to hiv , hepatitis c virus , and hepatitis b surface antigen . magnetic resonance imaging brain revealed significant cerebellar hemispheric and vermian atrophy [ figure 1 ] . his writer 's cramp responded well to botulinum toxin injection with a significant improvement in his handwriting [ figure 2 ] . t1 axial and t2 coronal magnetic resonance imaging brain images showing cerebellar hemispheric and vermian atrophy handwriting specimen before ( a ) and after ( b ) botulinum toxin injection dominantly inherited scas are a heterogeneous group ( 40 and counting ) of neurodegenerative disorders identified by specific mutations . they predominantly affect the cerebellum and its connections and are characterized by prominent limb ataxia with impaired balance , gait , speech , and eye movements . movement disorders in the form of myoclonus , chorea , parkinsonism , tremors , and dystonia have also been described . dystonia is commonly seen only in sca types 17 , 3 , and 2 . however , there are case reports of both focal and generalized dystonia in sca types 1 , 6 , 7 , 8 , 12 , 14 , 20 , and 36 as well . sca1 is associated with abnormal cytosine - adenine - guanine repeats on the ataxin 1 gene on chromosome 6 and can be accompanied by cognitive dysfunction , parkinsonism , and dystonia in addition to ataxia . however , extrapyramidal features are more common in sca types 2 and 3 than sca1 . studied extrapyramidal manifestations in 85 patients of sca types 1 , 2 , and 3 . overall dystonia was encountered in 13 of the 85 ( 15.2% ) cases only and was more common in sca2 ( 17.9% , n = 5/28 ) and sca3 ( 17.6% , n = 3/17 ) than sca1 ( 12.5% , n = 5/40 ) . in a study by lee et al . , extrapyramidal symptoms were found in 53% sca3 ( n = 29 ) and 12% sca2 patients ( n = 17 ) . none of the 6 cases with sca1 had any evidence of extrapyramidal dysfunction in their cohort . found dystonia in only 9.1% ( n = 33 ) of cases of sca1 as compared to 44.4% ( n = 9 ) cases of sca2 . in contrast to the usual pattern of focal or generalized dystonia reported in sca1 , our case presented with a prominent task - specific dystonia in the form of a writer 's cramp and added to the clinical spectrum of this subtype . a review of the literature reveals that even in the sca subtypes manifesting with dystonia , task - specific dystonia such as writer 's cramp is extremely rare . there are only stray case reports of writer 's cramp in sca types 6 , 7 , and 14 from all over the world [ table 1 ] and no reported case of this association in sca1 , as observed in our case . summary of reported cases of task specific dystonia in spinocerebellar ataxias pathways implicated in the manifestation of dystonia include the cerebello - thalamocortical and basal ganglia - thalamocortical circuits . in a pathoanatomical study of patients with sca1 , rb et al . found neuronal loss and degeneration in these pathways besides the primary motor cortex and cerebellum . it was also seen that the extent of the involvement of these structures correlated well with predominant clinical symptoms that were seen in an individual case . drr et al . have observed that the basal ganglia , especially the internal pallidum and the subthalamic nucleus show severe degeneration in sca3 but are relatively spared in sca1 which may account for the more frequent dystonia in sca3 . in sca patients with writer 's cramp , the dystonia can precede , accompany or follow the onset of cerebellar ataxia . in our patient , writer 's cramp manifested almost 1 years after the onset of the gait ataxia but subsequently became the most prominent and disabling complaint . to the best of our knowledge , this is probably the first case report of sca1 with a task - specific dystonia or writer 's cramp . our case highlights the ever - expanding phenotypic heterogeneity of the sca 's in general and sca1 in particular .

dystonia can be encountered in a small subset of patients with spinocerebellar ataxia ( sca ) , but task specific dystonia is extremely rare . we report a case of a 48-year - old male with confirmed sca type 1 ( sca1 ) with mild progressive cerebellar ataxia and a prominent and disabling writer 's cramp . this case highlights the ever - expanding phenotypic heterogeneity of the sca 's in general and sca1 in particular .

in the previous issue of critical care , chou and colleagues report that early initiation of renal replacement therapy ( rrt ) , as defined by rifle criteria , was not associated with reduction in hospital mortality in patients with acute kidney injury ( aki ) and sepsis . the timing of initiation of rrt has been a controversial issue for many years in both patients suffering from chronic kidney disease and those suffering from acute kidney disease . a recently published systematic review and meta - analysis on this topic concluded that earlier initiation of rrt in critically ill aki patients may have a beneficial impact on survival but that , in the absence of new evidence from suitably designed randomized trials , a definitive treatment recommendation can not be made . the idea that ' earlier ' initiation of treatment should be beneficial stems from our belief that sometimes complex interventions may change the course of a disease . in the case of aki and sepsis , it has been hypothesized that early dialysis in sepsis can reduce circulating levels of inflammatory substances and may therefore beneficially impact on the pathophysiological mechanisms of the disease . unfortunately , the validity of this premise has never been proven , and existing evidence points rather in the opposite direction . both literature data and the currently published study once more confirm that patients with sepsis die with aki rather than of aki : no single indicator of renal function or kidney injury was predictive of mortality in the multivariate model as it was applied by chou and colleagues . in the patient group with septic aki , dialysis should not be seen as a curative intervention , but rather as supportive therapy , preventing the patient from dieing from hyperkalaemia or fluid overload during the period the kidneys are temporarily failing . as a consequence , early dialysis , in the semantic meaning of ' intervention before support is needed ' , exposes patients to side effects of the treatment without really helping them . problematic for correct interpretation of the data in nearly all studies , including the one by chou and colleagues , is the fact that virtually all are retrospective . this complicates the interpretation at various levels . as strict clinical rules on the timing of initiation of dialysis were present in these icus during the collection of these data , all patients in fact started at the same ' time point ' in their disease , that is , when they fulfilled one of the preset criteria . as such , the categorization ' early versus late ' loses the semantic significance of a ' time - related event ' , and in fact no conclusions on timing can be made . conclusions on the impact of ' early versus late initiation of dialysis ' would only be justified when , at the moment patients fulfilled the criteria for dialysis , one group started rrt immediately while for the other group dialysis was delayed for one or more days . in the only available randomized controlled trials in this field , early versus late was defined by the urinary output versus biochemical values or by magnitude of urinary output . one important aspect of the current paper by chou and colleagues is that the rifle criteria have no value in determining the need for dialysis , as equal numbers of patients within the different rifle categories were apparently started on dialysis . indeed , if rifle were a good predictor of need for dialysis , one would expect that there was a preponderance of rifle class ' i ' or ' f ' in the cohort of dialyzed patients , quod non . unfortunately , the study does not provide data on the distribution of the different rifle categories in the non - dialyzed patients , but we would not be surprised that these rifle categories were exactly comparable to those in the dialyzed cohort . rifle is a scoring system developed to grade prognosis of acute renal injury , and although it performs quite well at the cohort level , its applicability to individual patient outcome is rather poor . this is even more true in the current study , as the most powerful component of the rifle criteria , urine output , was not taken into account . as a consequence , the correct conclusion of the current data should thus be that serum creatinine is not a good parameter to use for determining when to start dialysis in aki patients , just as has been found in patients with chronic kidney disease . the omission of urine output as a parameter is critical , especially as fluid overload ( central venous pressure > 12 mmhg ) was a criterion to start dialysis . the combination of evaluation of fluid status and urine output , and especially the evolution over time of these two parameters , is probably one of the major decisive criteria to start dialysis at present . when the patient is euvolemic , or even volume overloaded , and still remains oliguric despite maximal support , one should not await further organ damage , and dialysis should be initiated . in addition , as only patients who did actually start dialysis were included , it is difficult to guess what happened to patients with classes rifle 0 or i who did not start dialysis . it is likely that these patients had a better survival than those who did start dialysis , and then , the conclusion of the study would be that early initiation of dialysis based on rifle criteria is detrimental . such a conclusion is different from the statement that early initiation of dialysis based on rifle criteria in patients with sepsis does not reduce mortality . in conclusion , maybe we can not answer this question now because it may be the wrong question : as dialysis is supportive and not curative , patients either need dialysis , in which case it should not be delayed , or they do not need dialysis , in which case it can only harm . once we have agreed on those , the timely initiation of dialysis in those who are predicted to inevitably progress to one of those conditions seems the most appropriate strategy . in contrast , with current practice , it seems unlikely that serum creatinine or another serum / urine - based biomarker will be one of those criteria .

earlier initiation of dialysis may have a beneficial impact on survival of critically ill patients with acute kidney injury ( aki ) . a retrospective analysis in the previous issue of critical care showed that early initiation of renal replacement therapy ( rrt ) , as defined by rifle criteria , was not associated with a reduction in hospital mortality . the retrospective character of many studies describing the results of early rrt initiation and the validity of rifle criteria to determine the need for dialysis can be questioned , in particular when urinary output is not considered . initiating dialysis in aki should be based on clinical criteria and not on serum creatinine or another serum / urine - based biomarker .

in central europe patients with muscle - invasive bladder cancer ( mibc ) have relatively poor prognosis . this includes also patients initially undergoing radical surgical treatment . in order to find underlying reasons , we have previously estimated the timing of radical cystectomy in a multicentre study involving 575 polish patients . however , one of the most important limitations of the abovementioned study was the inclusion criterion of being cystectomised instead of being diagnosed with mibc . consequently , the data on final treatment in the whole population of mibc patients are still limited . within this short communication the aim of this study was to describe patterns of care in polish patients with newly diagnosed mibc . this is a multicentre retrospective cohort study involving 296 consecutive patients with primary mibc diagnosed in the years 20122013 in 13 polish urological centres . in all patients the diagnosis was made based on histological examination of surgical specimens from transurethral resection of the bladder tumour . the mean age of the cohort was 72.1 years and male - to - female ratio was 3.2 : 1 ( 225 vs. 71 ) . differences between cystectomised and non - cystectomised patients were evaluated with u mann - whitney test and test for quantitative and qualitative variables , respectively . comparison of patients depending on qualification for radical cystectomy table presents absolute , median , or percentage values . haemoglobin ; rc radical cystectomy data on nicotine use was available only in 220 patients ( 77.2% of cohort ) among 121/285 ( 42.5% ) patients disqualified from rc , 32/121 ( 26.4% ) patients were qualified for a second step of transurethral resection of the bladder tumour ( turbt ) intentionally followed by systemic chemotherapy , four ( 3.3% ) patients after complete turbt were qualified for adjuvant intravesical chemotherapy only , while the remaining 85 ( 70.2% ) patients were qualified for palliative treatment in the form of chemotherapy and/or radiotherapy and/or best supportive care . despite poor outcomes of treatment of patients with mibc in central europe , data on patterns of surgical and medical management , as well as its quality , are still unavailable . we performed a retrospective study aimed at the description of further treatment in patients diagnosed with primary mibc . the most important finding of our analysis is the high percentage of patients qualified for radical treatment . a clinically important fact is that that these numbers would probably be higher if we excluded from the analysis patients with metastatic disease , who are not candidates for rc by definition . available data on patterns of care in mibc patients published in the last 10 years present significantly lower rates of curative treatment , covering 2152.5% of patients [ 47 ] . however , the studies cited above included more patients and/or were based on cancer or national registries . some portion of patients included in these analyses was treated with radiotherapy alone , which nowadays is not regarded as a radical approach . we have also found that patients disqualified from curative treatment were older , had lower bmi values , lower haemoglobin concentration , and declared lower rate of nicotine abuse and shorter time interval between first symptom to diagnosis . while age alone should not influence clinical decisions , it is suggested that older mibc patients are less frequently qualified for radical surgery , and it is well established that the morbidity related to rc is increased within this group [ 912 ] . low haemoglobin concentration , as well as malnutrition is associated with shorter survival after rc [ 9 , 13 , 14 ] . moreover , abnormal bmi value increases the risk of surgical complications [ 12 , 15 , 16 ] . our findings on nicotine use and time from first symptom to diagnosis are both surprising and unexplainable . considering the pathogenesis and clinical course of the disease , one can suspect that these results are fortuitous . finally , they are of no practical significance . the study 's strengths are its multi - institutional character , involvement of both academic and non - academic urological departments , and enrolment of a representative cohort of patients . the most important limitation of the study is the lack of data on lymph node and distant metastases . as available clinical staging is limited to regional status , among patients disqualified from surgery there are both patients unfit for surgery and patients with initially metastatic disease . their differentiation in the present study was not performed . because the majority of polish patients with primary mibc receive curative treatment , the stage of the disease alone seems not to be the leading cause of poor survival . however , the appropriateness of qualification for rc and treatment quality needs to be assessed for a final conclusion on the factors influencing outcomes of treatment in poland . the results presented within this paper come from post hoc analysis of data collected during a multicentre study aimed at oncological characterisation of a large cohort of polish patients with primary urothelial carcinoma of the bladder . the study was produced under the auspices of the residents section of the polish urological association . all the investigators , but two ( m.s . and p.r . ) , were urologists in training .

a potential reason for poor survival among patients with muscle - invasive bladder cancer ( mibc ) in poland is initial disqualification from curative treatment due to advanced stage of the disease or low performance status . the aim of this study was to describe patterns of care in patients with newly diagnosed mibc.this is a multicentre retrospective cohort study involving 296 consecutive patients with primary histologically diagnosed mibc . therapeutic decisions and potentially underlying clinical factors were analysed.full clinical data was available for 285 patients . one hundred and sixty - four ( 57.5% ) patients were qualified for radical cystectomy ( rc ) , 32 ( 11.2% ) patients for a second step of transurethral resection of the bladder tumour ( turbt ) intentionally followed by systemic chemotherapy , four ( 1.4% ) patients after complete turbt were qualified for adjuvant intravesical chemotherapy only , while the remaining 85 ( 29.8% ) patients were qualified for palliative treatment in the form of chemotherapy and/or radiotherapy and/or best supportive care . patients disqualified from curative treatment were older ( 78 vs. 69 years , p < 0.02 ) , had lower bmi values ( 24.5 vs. 25.7 kg / m2 , p < 0.02 ) , lower haemoglobin concentration ( 11.6 vs. 12.9 mg / l , p < 0.02 ) , declared lower rate of nicotine abuse ( 50.5% vs. 72.1% , p < 0.02 ) , and had a shorter time interval between first symptom and diagnosis ( 30 vs. 60 days , p = 0.02).as the majority of polish patients with primary mibc receive curative treatment , the stage of the disease alone seems not to be the leading cause of poor survival . however , appropriateness of qualification for rc and treatment quality needs to be assessed for final conclusion on the factors influencing outcomes of treatment in poland .

myocardial bridging is defined as an epicardial coronary artery that goes intramurally through the myocardium beneath the muscle bridge . while generally benign , myocardial bridges can cause ischemia , ventricular tachyarrhythmias , atrioventricular block , and sudden cardiac death.1)2 ) for symptomatic patients , various therapeutic approaches have been attempted , but the optimal treatment of myocardial bridging still remains controversial.3)4 ) coronary stenting has been another therapeutic option with medical and surgical treatment , but the high risk of perforation and high rate of in - stent restenosis have limited its use.5 - 10 ) recently , we experienced a patient with a perforated coronary artery after implantation of a drug - eluting stent ( des ) that was successfully rescued by deployment of covered stent in symptomatic myocardial bridging . a 46 year - old woman who had no coronary risk factors , presented with exertional chest pain for several weeks . the chest pain was typical for angina pectoris and depressed st segments were noted at the exercise test . echocardiography revealed normal left ventricular ( lv ) systolic function { ejection fraction ( ef)=72% } without any regional wall motion abnormality . we performed coronary angiography which showed significant stenosis ( up to 80% ) aggravated by severe myocardial bridging at the mid - portion of the left anterior descending ( lad ) artery ( fig . so we decided to do a percutaneous coronary intervention ( pci ) at the lad lesion . through a 7 fr judkins guiding catheter , predilatation was performed with a maverick balloon catheter ( 2.515 mm , boston scientific , natick , ma , usa ) at 10 atmospheres for 20 seconds . we deployed a taxus stent ( 3.516 mm , boston scientific , natick , ma , usa ) according to the size of the predilated balloon catheter . but the middle segment of the lesion was not compliant , so the stent was not fully expanded with nominal pressure . , the coronary artery was perforated and some extravasation of contrast media was observed in the pericardium ( fig . but , the patient 's vital signs were stable ( blood pressure 115/70 mmhg ) with only a mild increase in heart rate . however , because the perforated site was entrapped intramurally through the myocardium in the interventricular groove , there was no evidence of accumulated blood at the dependent position of the pericardium on fluoroscopy or echocardiography . so we decided to observe the patient with close monitoring of symptoms and vital signs . but echocardiography showed no evidence of pericardial effusion . on an intravascular ultrasound ( ivus ) study , a large perforated site and a perivascular hematoma were observed in the mid - portion of the deployed stent ( fig . 1c ) . based on the ivus findings , a jo covered stent ( 319 mm , jomed international ab , helsingborg , sweden ) was deployed at the perforation site . four months after the procedure , ct angiography showed no evidence of residual hematoma or pseudoaneurysm ( fig . 2a and b ) . follow - up coronary angiography performed at 8 months after the procedure showed good distal flow with minimal stenosis at the proximal edge of the stent ( fig . 2c ) . although coronary stenting is an effective interventional approach to improve symptoms in selected patients with myocardial bridging , it is associated with a high risk of coronary perforation.5 - 7 ) the reason for this phenomenon is not clear . autopsy findings showed that tunneled segments in myocardial bridging tend to be deficient in vascular smooth muscle density , which may be more prone to vascular disruption during high inflation pressures during pci.11)12 ) another study revealed that the vessel area in the myocardial bridge segment was significantly smaller than that in the adjacent reference segments proximal and distal to the myocardial bridge throughout the cardiac cycle.13 ) this finding might explain the higher rate of coronary perforation associated with coronary stent implantation for myocardial bridges . the histological and anatomical differences in tunneled coronary arteries may require an adjustment in stent diameter and inflation pressures to help reduce the risk of coronary perforation . in our case , we selected an oversized stent , resulting in coronary perforation . pre - interventional ivus may be helpful in selection of an appropriate size of stent and , in particular , in cases that require high inflation pressures for optimal stent implantation . although coronary perforation is an uncommon complication following pci , it usually causes a catastrophic result including cardiac tamponade , emergency coronary artery bypass surgery , or pseudoaneurysm formation , with the potential for late coronary rupture and death.14)15 ) but , in myocardial bridging as observed in our patient , because the perforation site and extravasated blood are mainly confined in the interventricular groove , perforation itself usually does not cause hemodynamic instability . we were able to evaluate the lesion using ivus to decide on implantation of a covered stent after perforation . coronary stenting in myocardial bridging has been associated with a high restenosis rate.9 ) possible factors for this include shear stress from persistent external compression from myocardial bridges , causing neointimal proliferation , and long stent and recoil phenomena when inadequate pressures are used for stent deployment . a des was chosen as a feasible alternative to surgery because it had the ability to reduce restenosis . in our case , we performed pci with a des , which also showed good distal flow with minimal restenosis at the proximal edge of the stent at follow - up angiography . our case showed that , even though coronary perforation had occurred in myocardial bridging , the perforation site was confined in the interventricular groove . therefore , it could be managed more easily than conventional coronary perforation .

we successfully rescued a patient whose coronary artery perforated following implantation of a drug - eluting stent ( des ) , by deploying a stent - graft in symptomatic myocardial bridging . our case demonstrated that coronary perforation could be handled without difficulty when perforated myocardial bridging is confined to the interventricular groove

the accelerometer is a useful measurement tool for monitoring physical activity , especially the upper extremity activities of patients with stroke1 . unlike previous assessment tools for upper - limb function after stroke , accelerometers can also assess level of performance of activities of daily living , as well as capacity ( the ability to perform some action)2 . thus , accelerometers allow objective measurement of upper - limb activity during daily living as well as in clinical practice3 . a previous study found that accelerometry of daily arm activity was significantly related to the mal - aou scale , an upper - limb assessment tool which takes the form of a semistructured interview4 . an accelerometer was also used in a study assessing the reliability of log-14 for upper limb measurement5 . however , subjects in these studies wore the accelerometer for more than one day , making it difficult to evaluate upper - limb movement of specific activities . the action research arm test ( arat ) is a tool that assesses the abilities of grasp , grip , pinch and performance of gross movements after stroke6 . it is a highly reliable and validated measurement tool for the evaluation of upper limb motor impairment7 . the arat is used to measure specific upper limb movements including fine and gross movements8 , and a comparison between the accelerometer and arat would determine whether the accelerometer is a useful tool for monitoring specific upper limb activities . accordingly , the purpose of this study was to investigate the sensitivity of an accelerometer in measuring upper extremity activities . fifteen subjects were recruited from rehabilitation hospitals in won - ju , korea for this study . the inclusion criteria were : a diagnosis of stroke with hemiparesis , no severe deficits in cognitive function ( mini mental state examination ( mmse ) score > 22 ) and the absence of orthopedic upper limb limitations . we obtained written informed consent using the form approved by the yonsei university wonju institutional review board from all research subjects before beginning the study . the accelerometer used in this study was a fitmeter developed by fit dot life corporation of korea in 2010 . the fitmeter is small ( 35 mm 35 mm 13 mm ) and light ( 13.7 g ) ; thus , it is easy and convenient to attach to a specific body part . the frequency of measurement and sensitivity of the fitmeter are from 1/32 to 30 seconds and 2 to 8 g , respectively9 . for this study , the fitmeter was set at 1/32 seconds and 2 g to measure slower activities and fine motor movements . the arat assesses motor function of the upper limbs after stroke , as well as the amount of movement possible in stroke recovery stages . the arat uses a wooden box and different sizes of blocks and other objects . the arat consists of 19 test items including those that involve grasp ( 6 ) , grip ( 4 ) , pinch ( 6 ) and gross movement ( 3 ) actions . the highest possible arat score is 57 , based on a four - point scale that rates the quality of movement over 60 seconds as follows : 0 = no movement , 1 = partial movement , 2 = movement performed slower than normal ( 560 seconds ) , 3 = movement performed normally ( in less than 5 seconds ) . the test takes 515 minutes to complete , depending on the subject s symptoms10 . the inter - rater and test - retest reliabilities for stroke patients were 0.99 and 0.98 , respectively indicating high reliabilities11 . subjects wore accelerometers embedded within wrist bands on both wrists and performed the arat items . subjects were required to place both arms on the desk before and after performing the arat in order to clarify the start and end point of the test . the mann - whitney test was used to compare median differences in arat and acceleration scores between the affected and non - affected sides . spearman s rank correlation coefficient was calculated to test the significance of relationships between acceleration and arat variables . table 1table 1.subject characteristics at baseline ( n=15)characteristicgender , m / f ( total)9/6 , 15age ( yrs)67.3 9.9affected side , l / r , ( total)7/8 , 15time since stroke onset ( y)3.1 2.3values expressed as mean sd or n. shows the demographic characteristics of the study subjects . the study included nine males and six females with a mean age of 67.3 9.9 years . there was a significant difference in the median score between the affected and non - affected sides both the arat and accelerometer results . the arat scores were higher on the non - affected side ( p<0.05 ) , while upper limb activity was higher on the affected side ( p<0.05 ) ( table 2table 2.median differences in arat and acceleration scores between the affected and non - affected sidesaffected sidenon - affected sidearat42.8 ( 11.1)57 ( 0)accelerometer74488.3 ( 51778.1)35963.1 ( 11240.5)values are mean ( sd ) , p<0.05 ) . the correlation coefficient between arat and accelerometer results indicates that the results were not significantly correlated ( table 3table 3.correlation between arat and the accelerometerarat ( affected side)accelerometer ( affected side)0.24accelerometer ( non - affected side)0.91 ) . values expressed as mean sd or n. values are mean ( sd ) , p<0.05 previous accelerometer studies primarily measured physical activity over several days12 . in contrast , this study was performed in a clinical setting and measured and compared upper extremity movements of specific activities performed using the paretic and non - paretic hands . the accelerometer results indicated there is a significant difference between the affected and unaffected upper extremities , and this was confirmed by the arat . this outcome was not consistent with previous studies comparing accelerometer recordings and the results of the motor activity log and the actual amount of use test1 . first , because we did not consider the timing of arat activities , there are data errors associated with the amount of upper limb movement as measured by the accelerometer . in addition , differences in quantitative and qualitative data of the accelerometer and arat affected the correlation coefficients . however , the results of this study indicate that the accelerometer is a sensitive tool for measuring upper limb activity . future studies should use an accelerometer to measure specific activities in a clinical setting .

[ purpose ] this study investigated the sensitivity of an accelerometer in a comparison with the action research arm test ( arat ) . [ subjects ] fifteen stroke patients participated in this study . [ methods ] subjects wore accelerometers on both wrists and performed the arat items . we then compared the data measured by the accelerometer with that of the arat . [ results ] arat scores were higher on the non - affected side than the affected side , while the amount of upper extremity movement was higher on the affected side . the correlation coefficients for the two tools were not significantly different . [ conclusion ] our findings indicate that an accelerometer is a useful and sensitive instrument for clinically measuring the upper extremity activity of patients with stroke .

the deleterious impact of hydrosalpinx on fertility is best exemplified by studies showing a 50% reduction in ivf pregnancy rates in their presence ( 1 ) . chlamydia infection and pelvic inflammatory disease are associated with an increased risk of both tubal infertility and ectopic pregnancy , with the association proportionate to the number of infections(2 ) . various theories have been proposed to explain the observation of lower ivf pregnancy rates in the setting of hydrosalpinx , to include mechanical effects , embryo and gametotoxicity , decreased expression of key implantation molecules , and/or a direct effect on the endometrium leading to intrauterine fluid formation(3 ) . drainage of hydrosalpingeal fluid into the endometrial cavity may exert a mechanical effect by washing the transferred embryo from the cavity . cultured epithelial cells isolated from hydrosalpinx affected tubes produce a fluid which is hostile to both spermatozoa and early mouse embryo development(4 ) . multiple studies have demonstrated deficiency in endometrial markers of embryo receptivity in the presence of hydrosalpinx . il-2 , t lymphocytes , cd3 + , cd8 + , cd4 + , alpha v beta 3 integrin , lif , mmps and hoxa10 have all been shown to be decreased in the endometrium from women with versus without hydrosalpinx(5 - 8 ) though molecular inflammatory changes in the setting of hydrosalpinx are documented , a hysteroscopic endometrial phenotype in the presence of hydrosalpinx has not been well characterized . eighteen months prior to presentation , she was diagnosed with a chlamydial infection by cervical culture and treated with a single dose azithromycin . a negative chlamydia culture was confirmed four months prior to presentation . as part of her initial infertility evaluation at our center , a hysterosalpingogram ( hsg ) was performed in a window of doxycycline prophylaxis which demonstrated bilateral hydrosalpinges , with the right tubal diameter measuring greater than the left and bilateral tubal occlusion ( figure 1 ) . hysterosalpingogram demonstrating bilateral distal tubal occlusion with hydrosalpinges staged combined endoscopy was performed in the operating room . hysteroscopy revealed endometrial inflammation as evidenced by diffuse hyperemia and mucosal oedema ( figure 2a ) . laparoscopic findings were significant for a right sided hydrosalpinx measuring over 2 centimeters in diameter and dense adhesions of the bowel to the left hydrosalpinx and ovary . she underwent right salpingectomy and left tubal occlusion at the visible isthmic region via clip placement . she experienced an unremarkable postoperative course and was scheduled for in vitro fertilization ( ivf ) treatment . hysteroscopic appearance of endometrial cavity immediately pre - salpingectomy ( a ) and six months after interruption of communication with hydrosalpinges ( b ) . for each series , images from left to right depict right cornual , fundal and left cornual regions , respectively . six months later , the patient underwent hysteroscopic endometrial cavity evaluation in preparation for ivf . at hysteroscopy , the patient subsequently underwent ivf with transfer of a single blastocyst culminating in the delivery of a healthy infant at term . given the importance of tubal patency in non - ivf treatment and the detrimental impact of tubal pathology such as hydrosalpinx , oviductal evaluation represents an important part of the initial female infertility work up . given the trend among women to present for infertility care later in the reproductive lifespan , patients undergoing ivf may not have had prior tubal evaluation via either of the standard modalities . transvaginal ultrasound in the detection of hydrosalpinx has been reported , albeit with user - dependent sensitivity . the present case suggests that an inflamed endometrial appearance may be a hysteroscopic sign of a communicating hydrosalpinx requiring dedicated tubal study prior to initiating fertility treatment as depicted in figure 2 , the endometrial surface appears erythematous with friable and tortuous vascularity in the setting of a hydrosalpinx , and normal appearing after surgical interruption of hydrosalpingeal drainage . subsequent ivf treatment and transfer of a single blastocyst resulted in an intrauterine pregnancy , with subsequent term delivery , indicative of functional embryo receptivity on a molecular level , although no molecular evidence is available for confirmation . in view of these findings , we propose an inflamed appearance at hysteroscopy for endometrial cavity screening should prompt a dedicated study to rule out hydrosalpinx prior to proceeding with ivf treatment . to date , such a hysteroscopic endometrial phenotype in the presence of hydrosalpinx has not been described .

we report the hysteroscopic findings in a 22 year old nulligravid patient with bilateral communicating hydrosalpinges . the inflamed hyperemic endometrial cavity encountered preoperatively normalized at second look hysteroscopy six months after bilateral tubal interruption . the patient underwent successful ivf with culmination in a singleton , live birth . we propose that an inflamed appearance at hysteroscopy , done for endometrial cavity screening , should prompt a dedicated study to rule out hydrosalpinx prior to proceeding with ivf treatment . to date , such a hysteroscopic endometrial phenotype in the presence of hydrosalpinx has not been well characterized .

we compared the lumbar repositioning error ( re ) according to different lumbar angles in a flexion pattern ( fp ) subgroup of patients with non - specific chronic low back pain ( nsclbp ) . approximately 85% of this population was classified as having nsclbp , with no radiological change between the 12th rib and the inferior gluteal fold1 . the management approach has been the use of subgroups classified on the basis of pain - provoking postures and movements in nsclbp2 . repositioning error ( re ) is defined as proprioceptive impairment , known to result in poor spinal stability3 . impaired proprioception in positions such as sitting and standing can be related to lbp . in particular , it is associated with flexion direction in the subgroup with pain provoked by lumbar flexion4 . however , few studies have examined re in subjects with flexion - related lbp during the performance of different lumbar flexion angles , as in extension on sitting and standing upright postures . thus , the purpose of this study was to compare changes in lumbar re at different angles while standing and re - standing and sitting and re - sitting in a subgroup of fp subjects with nsclbp . s system5 , 6 and was defined as subjects with pain provoked by postures and movement - related flexion of the lumbar spine . in the fp subgroup , symptoms were relieved by movement associated with extension , lordosis of lumbar segments , and the loss of a neutral spine posture due to a flexed spine5 , 6 . the subjects were aged 44.78.2 years ( mean sd ) with a height of 165.45.1 cm , a body weight of 62.37.2 kg , a korean oswestry disability index ( odi ) of 30.04.0% , and a visual analog scale ( vas ) score of 5.51.2 . ethical approval was obtained from the inje university faculty of health science human ethics committee . we used a dual inclinometer ( acumar , lafayette instrument co. , lafayette , usa ) to measure the lumbar re at the main and companion parts ( l1 and sacrum ) . intra - test reliability of the dual inclinometer was 0.90 , and the inter - test reliability was 0.85 . the subjects were required to stand upright for 5 s and to hold lumbar flexion angles of 30 and 15 and a lumbar extension angle of 15 for 5 s , followed by return to the starting position . the re of the lumbar spine was defined as the difference in the mean sagittal angles between neutral sitting and re - sitting phases and between the standing and re - standing phases of the task . the spss software ( ver . 12.0 ; spss , chicago , il , usa ) was used for all analyses , and the level of statistical significance was set at 0.05 . one - way repeated - measures analysis of variance and the least significant difference ( lsd ) test as a post hoc pair - wise comparison were used to determine significant re differences among the six tasks . the lumbar re increased significantly with flexion at 30 during sitting . upon sitting upright , the lumbar re between sitting and re - sitting was significantly greater with a flexion of 30 ( 5.13.7 ) than 15 ( 2.03.7 ) or with an extension of 15 ( 1.51.9 ) ( p < 0.05 ) . in addition , re while sitting with a flexion of 30 was significantly greater than that between the standing and re - standing positions during flexions of 30 ( 1.91.5 ) and 15 ( 0.70.6 ) and an extension of 15 ( 0.80.6 ) ( p < 0.05 ) . upon standing upright , the lumbar re between the standing and re - standing positions during a flexion of 30 was significantly greater than during a flexion of 15 or extension of 15 ( p < 0.05 ) . in our study , lumbar spine re increased in a specific direction while in the sitting position in the fp subgroup . in accordance with previous research5 , 6 , the fp subgroup in this study had a deficit in flexed - toward status while sitting , but this was not apparent while standing . however , while the previous study used lumbar full flexion in the standing position , our study used a flexion angle of 30. the neutral re from 30 of flexion was greater than those from 15 of flexion or 15 of extension while standing , and less than that from 30 of flexion while sitting . the standing position would have added proprioceptive input from other distant receptors , while the adoption of a seated position would minimize other proprioceptive inputs by immobilizing the lower legs and the pelvis7 , 8 . furthermore , this may be due to characteristics of the fp group , where pain is provoked during sitting , whereas the pain tended to be relieved upon standing2 . the results of this study revealed the highest displacement changes between sitting and re - sitting on flexion of 30 of the lumbar spine from the upright sitting posture . when spinal structures , such as passive ligaments or active muscles are stretched or flexed during sitting , reflexive muscle activity may be reduced , passive structures may be lengthened , and their tension may be reduced due to unsuitable stretching stimulation from the central nervous system9 . dolan and green10 reported significantly increased lumbar re following 5 min in a slouched posture , but not following a 3-s duration . our study showed increased re immediately while sitting at a flexion angle of 30. we suggest that a higher lumbar flexion angle may have a negative impact on lumbar positioning sense while in the sitting posture than in the standing posture in fp subjects with nslbp . we also suggest that the measurement method for lumbar res would be more effective while sitting using a flexion angle of 30 in fp subjects .

[ purpose ] this study determined the change in lumbar position sense according to lumbar angles in a flexion pattern ( fp ) subgroup of patients with non - specific chronic low back pain ( ncslbp ) . [ subjects ] thirteen subjects with fp low back pain participated . [ methods ] the lumbar repositioning error ( re ) of subjects was measured between a neutral starting position and re - position phases at three angles , in sitting and standing upright positions . [ results ] lumbar re was significantly greater during lumbar flexion at a 30 angle in the sitting position than in the other tasks . [ conclusion ] in the flexion - related subgroup , the lumbar re measurement may be a more sensitive evaluation method using a lumbar flexion angle of 30 while in the sitting position , compared with other angles in sitting or standing positions .

role of n - acetylcystein ( nac ) in adults with non - acetaminophen induced liver failure was described in few studies in literature . studies were particularly relevant to countries where , liver transplantation facilities are limited or unavailable . found a significantly improved transplantation free survival at 3 weeks and at 1 year with the use of nac in non - acetaminophen related liver failure , the benefit being confined to those with early hepatic encephalopathy . dengue infection is prevalent in southeast asia , and according to the epidemiological unit , ministry of health , sri lanka , during the last 4 months of the year 2012 , 11148 suspected dengue cases and 46 deaths have been reported . the elevation of transaminases is usually less than five - fold greater than upper limit of normal . however , levels more than five - fold were reported 36.8% and 74.4% of patients with classical dengue and dengue hemorrhagic fever ( dhf ) respectively . fulminant hepatitis tends to occur more often in dhf or dengue shock syndrome compared to classic dengue infection and case fatality rate of 50% being reported . although , nac has shown benefit in non - acetaminophen related liver failure , it was not well studied in dengue associated severe hepatitis . a previously healthy 54-year - old mother of three admitted with 3 day history of fever , headache and body ache . physical examination on investigation on the day of admission revealed platelets 84,000/cumm and haematocrit ( hct ) 37% . on the 2 day , she was transferred to intensive care unit ( icu ) as her platelets dropped to 41,000 per cu mm and hct increased to 47% . liver transaminases showed mild to moderate rise with ( ( ast ) aspartate transferase ) 302 u / l and ( ( alt)alanine transferase ) 262 u / l and patient was given total dose of 10 g of acetaminophen over 3 days at the time . she had stable hemodynamics apart from heart rate of 121 beats / min , but she developed right side moderate pleural effusion , icterus , mild ascites and right hypochondrial pain . in the icu , she deteriorated further , with a decline of glasgow coma scale ( gcs ) to 11 , but no focal neurological signs . urgent computed tomography brain was done and it neither showed intracranial hemorrhage nor evidence of increased intracranial pressure . her liver functions continued to deteriorate and liver enzymes reached peak value of ast 16261 u / l and alt 4545 u / l , ( pt / inr ) prothrombin time / international normalized ratio 1.7 and total bilirubin 5.9 mg / dl on 4 day of admission ( 7 day of illness ) . intravenous nac was started at 100 mg / kg / day as an infusion and continued for 5 days with liver failure regime . marked improvement in liver enzyme was noted and sgot and sgpt levels dropped by more than half by 48 h of treatment . on the 9 day of admission , liver function revealed ast 300 u / l , alt 223 u / l and pt / inr 1.2 , and her conscious level improved to gcs of 15 . during the course of illness , she had mild gum bleeding and few ecchymotic patches with lowest platelet count of 18,000 per cu mm . her serology was positive for dengue antibodies but negative for hepatitis a and b. hepatitis e serology was not done due to unavailability . co - infection of malaria was not excluded as she was from neither endemic area nor her symptomatology typical of malaria including fever pattern . possibility of leptospirosis can not be excluded in this case as serology was not done . however , she did not have any exposure and her renal functions were never abnormal . during her follow - up visit at 2 weeks after discharge , she had normal liver profile and did not have any evidence of chronic liver disease . both virus itself and dysregulated immune response to virus are being described as possible mechanisms of liver damage in literature . although , severe hepatitis associated with dengue fever is a rare occurrence , it carries significant mortality and morbidity . nac , mostly used in acetaminophen poisoning , acts through its antidote effect of repletion of hepatocellular glutathione stores . nac scavenges free radicals , improves antioxidant defense and acts as a vasodilator to improve oxygen delivery and consumption . these properties of nac have been postulated to improve outcome in patient with dengue associated acute liver dysfunction . concluded that benefit is seen when nac is used early stage of liver failure rather than late stage . a retrospective analysis on nac in dengue associated liver failure by kumarasena et al . showed that 5 patients who survived out of 8 were in early ( coma grade 1 , 11 ) liver failure stage at the time when nac was started . this case report also supports the view that intravenous administration of nac is safe and benefits patients , if started in early stage of liver failure . this patient was treated with intravenous nac 100 mg / kg / day infusion for 5 days compared to 150 mg / kg bolus over 15 min followed by 12.5 mg / kg / h for 4 h and then 6.25 mg / kg / h for 72 h was given by kumarasena et al . in their retrospective analysis . reported a pediatric patient with dengue associated liver failure successfully treated with nac and they have given intravenous nac 100 mg / kg / day for 6 days . large randomized trials should be carried out to establish its efficacy along with appropriate dosage , timing , and duration of treatment .

although , n - acetylcystein ( nac ) has shown benefit in non - acetaminophen related liver failure , it was not well studies in dengue associated severe hepatitis . we report a case of dengue hemorrhagic fever associated severe hepatitis ( encephalopathy grade 2-drowsy and intermittent disorientation ) treated with nac resulted in good outcome without hepatic transplantation .

conventional inguinal hernia repair in children involves ligation of the hernial sac at the internal inguinal ring . laparoscopic surgery has been applied in children , and the repair is based on the same principle . this study aimed to document the authors ' experience with laparoscopic inguinal hernia repair in children . patients were admitted in the evening 1 day before surgery and discharged within 24 hours following surgery . two lateral ports of 3-mm were made through the right and left pararectal region to maintain a triangular orientation . in cases with small defects , laparoscopic ring closure ( lrc ) was done with 4 0 absorbable purse - string suture ( figures 1 , 2 , and 3 ) . in early cases and when difficulty was noted , saline was injected to separate the peritoneum from cord structures . the procedure was modified in 24% of children with a dilated internal ring ( figure 4 ) . ligature of the hernial sac at the internal ring is inadequate in such cases . here the sac was identified and dissected from the cord structures and then divided ( figure 5 ) . in patients with a large hernial sac , the landmarks identified included the iliopubic tract ( ipt ) , arching of fascia transversalis , cord structures , and the peritoneal reflection ( figure 6 ) . the ipt was identified as a shiny white band running under the cord structures at the inferior border of the internal ring . the tranversus arch was identified as the arching of the transversalis fascia immediately above and lateral to the internal ring . the ipt was approximated to the transversus arch by using nonabsorbable 2 0 interrupted suture to narrow the internal ring ( figure 7 ) . all patients were evaluated after 5 days , 4 weeks , 6 months , 1 year , and then annually , when possible . laparoscopic ring closure : continuation of the purse string on the inferior aspect of the internal ring . laparoscopic ring closure : completion of the purse string suture and occlusion of the internal ring . laparoscopic iliopubic tract repair : completion of the dissection and landmarks identified : ( a ) dilated internal ring , ( b ) iliopubic tract , ( c ) cord structures , ( d ) transversalis fascial arch . ninety - three indirect inguinal hernial sacs were closed ( 28 right , 7 left , 58 bilateral ) in 64 children ( 56 boys and 8 girls ) , ranging in age from 3 years to 13 years ( median , 5.1 years ) . the mean operating time for lrc was 25 minutes ( range , unilateral 21 to 35 ; bilateral 28 to 50 ) . the contralateral processus vaginalis was patent in 20% of children . in 24% of children , of the 29 bilateral hernias , 20 underwent lrc , 7 underwent liptr , and 2 underwent lrc on one side and liptr on the other side . the median follow - up was 30 months ( range , 2 to 84 ) . the first recurrence was in a 3-year - old female child after 8 months , following a right lrc . the second one was in a 4-year - old boy after 5 months , following a left lrc . the essential step in the conventional method for inguinal hernia repair in children is simple ligation of the hernial sac without narrowing the open ring . from our point of view first and the most obvious is the confirmation of the hernia and looking for the contralateral side . first championed by ger , ring closure is essentially a high ligation of the indirect hernia sac , which is the preferred technique in pediatric patients . this technique re - establishes the usual anatomic relations of the medial aspect of the internal ring and cord structures , giving them a flush union with the transversus abdominis muscle and eliminating the lead point that allows the intestine to enter the inguinal canal . ligature of the hernial sac at the internal ring alone is inadequate in patients with dilated internal ring ( 24% of children ) , as it does not take care of the component of the dilated ring . reports exist in the literature of methods to tighten this dilated ring . in hernias exceeding 4 mm to 5 mm in diameter , the external hemicircumference of the neck was opened to bring the conjoined tendon closer to the crural arch with a nonresorbable suture without the use of a prosthesis . ipt repair has been described as ringplasty , wherein the deep structures of the lateral iliopubic tract were approximated to the proximal arching musculotendinous fibers of the transversus abdominis muscle gazayerli described a laparoscopic preperitoneal ipt repair by approximating the transversus abdominis to the ipt , similar to the anterior preperitoneal approach described by nyhus and associates . we believe that in children with a dilated internal ring , a routine lrc would be inadequate . this is also observed in our 2 recurrences with one of them occurring within 5 months of lrc . the recurrence rate of inguinal hernias is slightly higher with laparoscopic herniorrhaphy than with the conventional technique . following liptr , there have been no recurrences . in children with recurrences after lrc , the surgeon has an undisturbed anatomy for the groin incision ; the risk of an injury to the vas deferens , subsequent testicular atrophy , and the risk of superior displacement of the testicle seem less likely . the problems we faced during liptr were trivial and related to the little scar tissue at the internal ring . this could be explained by some peritoneal fluid passing in between the knots placed during ring closure . our series shows that both techniques are safe , reproducible , and technically easy for experienced laparoscopic surgeons . the long - term follow - up of liptr would help assess any technical modifications . laparoscopic inguinal hernia repair in children can be offered , as it is safe , reproducible , and technically easy for experienced laparoscopic surgeons .

background : this study aimed to document the authors ' experience with laparoscopic inguinal hernia repair in children.methods:ninety-three hernia repairs were performed in 64 children . the neck was closed with a purse string suture by using 4 - 0 absorbable suture.results:ninety-three indirect inguinal hernial sacs were closed in 64 children . nine percent of children had an ectopic testis . the mean operating time for laparoscopic ring closure was 25 minutes ( range , unilateral 21 to 35 ; bilateral , 28 to 50 ) . the contralateral processus vaginalis was patent in 20% of children . in 24% of children , the final procedure was modified based on the findings of a dilated internal ring . a laparoscopic ilio - pubic tract repair was done in these cases . laparoscopic mobilization , orchiopexy followed by ilio - pubic tract repair was done in 9% of children . scrotal swelling occurred in one child . hydrocoele occurred in one patient . recurrence rate was 3.1%.conclusion : laparoscopic inguinal hernia repair in children can be offered , as it is safe , reproducible , and technically easy for experienced laparoscopic surgeons . iliopubic tract repair may be added in cases with dilated internal ring . recurrence following laparoscopic ring closure can be managed with laparoscopic ilio - pubic tract repair . the long - term follow - up of laparoscopic ilio - pubic tract repair is awaited .

nontyphoidal salmonella species are important food borne pathogens and acute gastroenteritis is the most common clinical manifestation accounting for about 70% of cases . splenic abscess is a very rare complication of nontyphoidal salmonella infections since the presence of antibiotics . here a 63-year - old woman from eastern part of turkey was admitted with the complaint of back pain , vomiting and nausea since 20 days . laboratory findings were as follows : white blood cell ( wbc ) count was 14670/mm ( 88.9% polymorphonuclear cells ) , haemoglobin 9.5 g / dl , platelet count 183000/mm , erythrocyte sedimentation rate 76 mm / h , and c - reactive protein ( crp ) 58 mg / dl ( normal value < 5 mg / l ) . an abdominal ultrasound revealed a hypoechogenic cystic structure with a diameter of 6172 mm in the upper part of the spleen with calcifications ( figure 1 ) . salmonella enteritidis was yielded from culture of the samples of the abscess obtained during the operation . it was sensitive to ampicillin , cotrimoxazole , cephalosporins of third generation , and ciprofloxacin . after isolation of the s. enteritidis from abscess , the patient was questioned in detail , it was learned that the patient had diarrhoea and fever existed 2 days and recovered without antibiotic treatment one month ago . the usual clinical presentation of nontyphoidal salmonella infection is self - limited gastroenteritis , however bacteraemia and focal extraintestinal infections may occur . risk of bacteraemia and focal extraintestinal infections are high in individuals with comorbidities such as malignancy , human immunodeficiency virus ( hiv ) , diabetes mellitus , and patients receiving immunosuppressive therapy . invasive nontyphoidal salmonellae disease is a major cause of mortality in african children and hiv - infected african adults . any tissue or organ may be seeded hematogenously by nontyphoidal salmonella and may form a local infection , become obvious months or even years after the initial bacteraemia producing characteristic clinical syndromes . some serovars of salmonella show a higher tendency for causing bacteraemia and these serovars differ in different countries . non - typhoidal salmonella serovars s. typhimurium and s. enteritidis are cause of invasive disease in industrialized countries , also they are predominant in african region . s. enteritidis had the highest blood invasiveness among non - typhoidal salmonella species in malaysia . our patient had diabetes mellitus and probably s. enteritidis bacteraemia occurred during the course of diarrhoea one month ago which lead to bacterial seeding in the spleen . antibiotic treatment for patients with mild to moderate gastroenteritis due to non - typhoidal salmonella is not indicated in healthy adults . however antimicrobial therapy should be initiated for patients who are severely ill and for patients with risk factors for extraintestinal spread of infection . the symptoms of splenic abscess are usually nonspecific the most frequent symptoms and signs are fever , abdominal pain and tenderness over left upper quadrant , splenomegaly , leucocytosis , and left lower chest abnormalities . our patient had back pain , vomiting , nausea and leucocytosis which were not specific for splenic abscess . computed tomography remains the gold standard and the most sensitive tool for the definitive diagnosis of splenic abscess . ultrasonography has 76% , ct has 96% sensitivity for the detection of abdominal masses . in our patient , diagnosis was confirmed with ct . at present , splenectomy is the gold standard for treatment while ultrasound- or ct - assisted percutaneous drainage and antimicrobial therapy can be considered as therapeutic alternatives especially in the presence of an isolated abscess . we preferred splenectomy for treatment as the causative microorganism was not known initially . in conclusion , although non - typhoidal salmonella gastroenteritis is rarely resulted with splenic abscess , patients with comorbities are at increased risk .

splenic abscess is a very rare complication of non - typhoidal salmonella infections . we report a case of splenic abscess caused by salmonella enteritidis . the patient is a 63-year - old woman with diabetes mellitus and underwent splenectomy . this case suggests that the patients with comorbities are at increased risk for invasive infections in non - typhoidal salmonella infections .

a 60-year - old patient presented in the outpatient department with chief complaints of hematuria with off and on episode of urine retention . there was history of passage of small stones in urine occasionally for last few days . on ultrasonography he was found to have 35 g prostate with slightly thickened bladder wall with a large stone in the urinary bladder . x - ray kub showed a large radio - opaque shadow in the pelvic region [ figure 1 ] . patient underwent open cystolithotomy and large bladder stone [ figure 2 ] with multiple small stones were retrieved . patient was started on alpha blockers in the immediate postoperative period and foley 's catheter was removed on the eighth postoperative day . at follow - up of three weeks after the surgery the patient was voiding in good stream with minimal post - void residual urine . as the name implies this variety of stone has a characteristic shape resembling a child 's toy [ figure 3 ] . these types of stone are commonly described in the veterinary literature with common occurrence in cattle , cats and dogs . dogs are mostly commonly affected and canine jackstones are usually composed of silica . depicting child toy called jackstone calcium oxalate is the most common component of urinary calculi . calcium oxalate monohydrate calculi are usually smooth and black , whereas stones comprising calcium oxalate dihydrate tend to be irregular and yellow . prostatic diseases , previous lower urinary tract surgery , metabolic abnormalities , upper urinary tract calculi , intravesicular foreign bodies , spinal cord injuries , transplant surgery etc . the presentation of vesical calculi varies from completely asymptomatic to symptoms of suprapubic pain , dysuria , intermittency , frequency , hesitancy , nocturia , and urinary retention . other common signs include terminal gross hematuria and sudden termination of voiding with some degree of associated pain referred to the tip of the penis , scrotum , perineum , back , or hip . the discomfort may be dull or sharp and is often aggravated by sudden movements and exercise . assuming a supine , prone , or lateral head - down position may alleviate the pain initiated by the stone impacting the bladder neck by causing it to roll back into the bladder . in our case the prostate is the likely cause of this stone . enlarged prostate probably restricts the calculus into its eccentric location and contributes to the growth of stone by causing stasis of urine . it is important to recognize the characteristic shape of the jackstones as they are susceptible to lithotripsy . we did not offer lithotripsy as this modality is known to be less efficacious in case of vesical calculus .

jackstone calculi are urinary tract stones that have a specific appearance resembling toy jacks . they are almost always composed of calcium oxalate dihydrate consist of a dense central core and radiating spicules . they are usually light brown with dark patches and are usually described to occur in the urinary bladder and rarely in the upper urinary tract . their appearance on plain radiographs and computed tomography in human patients has been described .

we quantified the results of entry screening for influenza a(h1n1)pdm09 at auckland international airport . using the information generated during screening , we retrospectively estimated the number of infected travelers who actually passed through the airport . to estimate the sensitivity of screening , we then compared screening findings with the expected number of infected travelers who passed through the airport . ethical approval was received from the northern x regional ethics committee of the new zealand ministry of health . the numbers of crew members on inbound international aircraft were estimated by using averages for flights into auckland . the number of travelers detected at each step and referred to the next step of the screening process was obtained from auckland regional public health service records . a confirmed case was one that met the current case definition ( as published on the ministry of health website , www.health.govt.nz ) and one for which rt - pcr result was positive . we estimated the number of infected travelers screened as the total number of confirmed cases in new zealand during this period , multiplied by the proportion of overseas - acquired cases , and the proportion of international travelers arriving at the airport . on april 30 , 2009 , nonseasonal influenza a ( h1n1 ) was made notifiable , and these data were collated on the national surveillance database ( episurv ) ( www.surv.esr.cri.nz/episurv ) . the proportion of infected travelers who acquired the infection overseas was extrapolated from ministry of health records of the first 100 cases of pandemic ( h1n1 ) 2009 because this information was not collected for all travelers with confirmed infection . the proportion of travelers who passed through the airport was determined from statistics new zealand ( www.stats.govt.nz ) arrivals records . confidence intervals were calculated by using the online calculator for screening on open epi ( 4 ) . during the screening period , 456,518 international travelers were screened ; 406 ( 0.09% ) of these were referred for medical assessment . of those , rt - pcr results were located for 89 ( 82% ) , among which 4 were positive . the expected number of infected travelers estimated to have passed through the border during the screening program was therefore 69 , giving an estimated sensitivity of 5.8% ( 95% ci 2.3%14.0% ) . this form of border screening is therefore unlikely to have substantially delayed spread of the pandemic into new zealand in 2009 . limitations of influenza screening include the high proportion of asymptomatic infected travelers ( 5 ) , incubation of infections acquired before or during a flight ( 3 ) , reliance on self - identification , limitations of case definitions , and limitations of thermal scanning ( 6 ) . modeling data have shown that the ability of border screening to delay global pandemic influenza is closely linked to the effectiveness of the screening process or travel restriction used . to delay influenza spread by 1.5 weeks , border restrictions need to reduce imported infections by 90% ( 7 ) . the potential effectiveness of screening arriving travelers to prevent or delay influenza epidemics has been debated . mathematical models and literature reviews have argued for ( 7,8 ) and against ( 911 ) this approach . some authors have found that entry screening for respiratory conditions or influenza a(h1n1)pdm09 is insensitive and not cost - effective ( 12 ) . this study has several limitations , particularly with regard to estimating the number of infected travelers who would have passed through the airport during the screening period . most cases of illness acquired overseas would probably not have been notified , particularly those in patients with mild illness who did not see a doctor or who saw a doctor but did not receive a diagnosis . the estimated proportion of overseas - acquired cases was based on data from the first 100 cases and would have decreased as the pandemic progressed . the net effect of these factors is unknown , but they would probably have increased the estimated number of undetected infected travelers passing through screening , thereby further reducing the estimated sensitivity of screening . it might provide public assurance and confidence that something is being done ( 14 ) . the communication of health information and advice on how to seek treatment is consistently recommended as a pandemic prevention strategy ( 12,15 ) and is usually delivered as part of border screening programs . these benefits need to be balanced against the considerable resources used , opportunity cost ( resources used for this activity and thereby unavailable for other activities ) , uncertain effectiveness , and inconvenience of border screening . to delay or prevent influenza entry at borders , influenza screening needs to be considerably more effective than the mostly passive program described here . we hope that during this interepidemic period , a major international review of the role of international air travel in the dissemination of emerging infectious diseases will be conducted to identify effective interventions . such a review should consider systemwide approaches , including exit screening , standardized health declarations , active screening of individual passengers ( including use of rapid laboratory tests and thermal scanning ) , passenger tracking , policies and practices that support sick travelers wishing to defer travel , and circumstances where airline travel should be suspended entirely .

entry screening for influenza a(h1n1)pdm09 at auckland international airport , new zealand , detected 4 cases , which were later confirmed , among 456,518 passengers arriving april 27june 22 , 2009 . on the basis of national influenza surveillance data , which suggest that 69 infected travelers passed through the airport , sensitivity for screening was only 5.8% .

neurofibromatosis type 1 ( nf-1 ) is an autosomal dominant disorder affecting 1 in 25003500 individuals . the fact that it is a progressive disorder and that almost 50% of cases are sporadic mutations multiplies its clinical magnitude several folds . discuss a report in which they described and classified seven different types of nf . of these , nf-1 and nf-2 it is a distinct relative of nf-1 but is 10%20% rarer , accounting for approximately one case in 36,00080,000 individuals . we present a case ; there was nf-1 with an extra calvarial plexiform neurofibroma ( pnf ) in occipito - cervical region with intracranial extension . a large portion of the visible swelling consisted of a neurofibroma and multiple caf au lait spots on the left upper and lower trunk [ figure 1 ] . swelling was excised completely . ( a ) clinical photographs showing the occipito - cervical swelling . ( b ) clinical photographs showing caf au lait spots on the left side of upper and lower trunk this 5-year - old boy presented with history of a gradually progressive painless swelling in the occipito - cervical region since birth . on examination , he had a large ( 10 cm 10 cm 5 cm ) occipito - cervical swelling , that was soft and nonfluctuant , and transillumination was negative . the boy had no neurological deficits and no signs or family history of nf-1 . in neuroimaging studies , the lesion was seen as an isointense on t1-weighted and heterogeneously hyperintense mass on the t2-weighted image and heterogeneous enhancement mass on postcontrast study [ figure 2 ] . there were multiple small underlying bone defect in the right occipital region [ figure 3 ] . the excision was total , and cerebrospinal fluid ( csf ) leak was seen on bone defect site , so periosteum was buried on bone defect site for preventing the csf leak . histopathological findings in hematoxylin and eosin staining were showing focal myxoid generation and spindle cells , with serpentine nuclei and wispy cytoplasmic border suggestive of pnf . t1-weighted with postcontrast sagittal magnetic resonance image shows heterogeneous enhancement lesion in the scalp of the right occipital region and nape of neck ( a and b ) both photographs showing three - dimensional computerized tomography skull showing thing of occipital bone right - sided with multiple bone defects after that pnf of scalp reported by ohaegbulam . in general , pnfs are found in association with nf ( in 26.7% of patients ) . pnf is usually found along the course of a major nerve trunk , the ophthalmic division in the face . it is an unencapsulated lesion and infiltrates the surrounding soft tissue to produce a fusiform appearance . it usually involves one large segment of the body and may involve both sides of the body either symmetrically or asymmetrically . the patient in this report had a pnf with nf and with telangiectatic discoloration of the overlying swelling , with multiple caf au lait spots on the left upper and lower trunk . only nine reports were found in the literature , of these nine , one case had nonoperative management , and two had only a biopsy . in the initial description by helmholtz and cushing , as detailed by scott , the location of the pnf was at the forehead and temporal region . it was suggested then that there was a propensity for the forehead and the temporal region , especially the area of the scalp innervated by the trigeminal nerve . although the forehead and the distribution along the course of the trigeminal nerve are the most common location , there have been a few reports of the pnf occurring at the occiput detail given in table 1 ; pnf occurs with slight male preponderance ( male / female 7:5 ) . most reported cases have been congenital , but presented later in life due to apprehensions of a social or psychosocial nature , mostly previous reported cases , there was only thinning of the occipital bone with no true dysplasia . review of literature of occipital neurofibroma nf is generally considered to be a neurocutaneous disorder of neural crest origin with very little emphasis on osseous abnormalities , although osseous dysplasia is one of the seven criteria for diagnosing nf-1 . most of the osseous lesions are thought to be secondary to the altered functioning of the nf-1 gene . besides true dysplasias , the secondary involvement of the bones may be due to compression of the malignant tumors seen in association with this disorder , such as malignant peripheral nerve sheath tumors and rhabdomyosarcomas . osseous manifestations in nf-1 are relatively common and occur in up to 50% of patients with nf-1 . there are only a few cases reported , and in most , it is difficult to ascertain whether the osteolysis was a result of the erosion secondary to the giant tumor or was merely associated as part of the disease syndrome . surgery for pnf has been described as being difficult because these tumors are usually very vascular , and torrential hemorrhage has been reported . there have been earlier reports of tumor firmly adhering to a dominant transverse sinus precluding complete excision . even in patients in whom there was no involvement of the sinuses , some authors have avoided reconstruction due to the theoretical risks of csf fistulas , the risk of malignancies in nf-1 is approximately 2.7% more than in the general population . there is approximately a 10% chance of malignant transformation reported in an existing case of nf . early surgical management should be the treatment of choice , not only just for cosmetic considerations but also for the theoretical risk of malignant transformation . however , the occurrence of osteolysis may also be prevented if it is indeed secondary to the pressure effects of the lesion , which can not be disproven conclusively .

plexiform neurofibroma ( pnf ) of the scalp is an extremely rare lesion reported in association with neurofibromatosis ( nf ) . occipital location of pnf is even more infrequent ; we reported one pediatric case of pnf in occipito - cervical region with multiple small occipital bone defects and associated with nf-1 .

a large increase in fusion rates for spine surgery has been observed in the last 20 years9 ) , and the frequency of revision spine surgery continues to increase . the most common reasons for additional surgery7 ) following fusion surgery are implant complications and pseudoarthrosis . . they may be in the shape of a male or female rectangle or hexagonal , octagonal , or star - shaped . therefore , it is necessary to have the instrument set used during the first operation or to have an appropriate screwdriver available . this is not always possible and in some circumstances the implant may no longer be in production . when revision surgery studies were examined5,12 ) , methods to remove broken screws are reported but very little information related to removing normal screws has been provided . no previous report has described a single - piece screw remover to remove normal screws . a small part of the rod is cut and placed in the screw when a removal set is not available . after tightening the screw head , mobile screw 's head this screw is a single piece that can be removed by hand or with pincers10 ) . the polyaxial remover presented here requires only a cap - screw screwdriver to remove the polyaxial screws . due to the structure of the remover and the method , existing screws can be removed with minimal dissection of surrounding soft tissue . debridement is important to remove scars , envision the bone elements , and prepare a new fusion bed during revision surgery . additional procedures applied to remove screws cause the surgeon to expend extra effort , which may lead to loss of time . our tool simplifies and accelerates the screw removal process ; thus , shortening operating time . the aim of our polyaxial screw remover design is to facilitate screw removal and thereby reduce blood loss , infection risk , cost , and effort expended by the surgeon . the patient demographic data and reasons for primary and revision surgery are given in table 1 . the aim when removing polyaxial screws is to stabilize the polyaxial head so the screw becomes monoaxial and can be removed by turning . the method described by kose et al.6 ) was of benefit when we designed our instrument , which has not been described previously in the literature . in the method of kose et al . , the screw becomes monoaxial with a rod and cap screw , whereas we used a u - shaped end section in our instrument without the need for a rod . the polyaxial remover consists of a single - piece shaft , sleeve , and handle ( fig . the shaft section is hollow , and the screw cap is tightened with a screwdriver . thus , a spine screw enters easily , and the surgeon avoids damaging surrounding tissue . the head section of a polyaxial screw was removed , and the screw was placed in the u - section ( fig . the cap screw was tightened on the screw , and the screw was fixed in the end section of the apparatus . the polyaxial screw that became monoaxial can be removed by turning the handles on the shaft . this method also removes the need to clean the fibrous tissue covering the internal screws and to see the screw shape . a total of 42 polyaxial screws were removed from five patients with the new single - piece screw remover ( fig . the polyaxial cap screws were 4.5 hexagonal screws in four patients and star - shaped in one patient . after removing the cap screws and rods , all of the polyaxial screws were made monoaxial with the instrument described above and were removed rapidly in a minimally invasive way with no complications . we have described a screw removal instrument designed to overcome the problems of screw removal often encountered during revision surgery . our clinical application shows that these screws can be removed without the need for additional materials or soft tissue debridement within or below the screw head . the correct screwdriver for the materials used can not always be obtained to remove existing implants during revision surgery because of the different designs . this explains why revision surgery can be a longer and more fatiguing operation with increased blood loss . of the several known spine surgery problems , the most accepted and most widely used method for repair is pedicle screw fixation1,2,3,4,5,6,7,8,9,10,11 ) . after fixation , the most common reasons for revision surgery7 ) are implant problems and pseudoarthrosis . methods and instruments to remove broken screws have been described in the literature5,12 ) . however , no additional methods6,10 ) , other than the rod techniques for removing normal screws , have been described . in the u - rod technique described by kse et al.6 ) , the cap screws are removed with an allen key , and the screw is placed in the u - shaped rod after removing the rods . after tightening the screw head , the screw with a mobile head becomes a monoblock screw . this single - structure screw can be removed by hand or with pincers . in addition , large metal cutters are required to cut the rods , a rod bender is needed to shape , and pincers are needed to turn the rod placed on the screw head . our screw remover design is a single piece and will not damage surrounding tissue or the pedicle ( fig . there is no need for metal cutters or a rod bender ; thus , reducing the time and effort required . the tool also removes the need to clean the fibrous tissue covering the internal screws to determine screw shape . second , a polyaxial remover was used after removing the cap screws , so it was necessary to have a screwdriver suitable for the cap screws . however , cap screws are generally hexagonal or star - shaped and there are few varieties . these screws are generally found in sets , or the screwdriver in the revision set can be used . another limitation was that the polyaxial remover could not be used if the cap screw is damaged or broken . the screw removal technique using the polyaxial remover is practical and easy and requires a minimal amount of time . the instrument described here can be used to remove all types of polyaxial and monoaxial screws used in spine surgery . here is no requirement for additional screw removal tools or materials and unnecessary debridement is avoided .

objectiveto present the clinical benefits of an instrument designed to facilitate removal of polyaxial screws during revision surgery.methodsall polyaxial screws can be removed without additional materials or a large amount of debridement using our newly designed instrument . forty - two screws were removed from five patients without any complications using this instrument.resultswe removed the cap screws and rods from the 42 polyaxial screws in five patients and made them monoaxial using the new screw removal apparatus . the screws and rods were removed quickly in a minimally invasive way with no complications . no damage to the pedicle or surrounding soft tissue occurred during screw removal . no neurogenic changes developed during revision surgery after changing the screws.conclusionthis newly designed screw removal instrument was used safely and effectively to remove all polyaxial and monoaxial pedicle screws .