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a 42 year old gentleman presented with rest tremors , bradykinesia and rigidity for 10 years . he responded very well to the treatment [ updrs scale ( off 40 , on 24 ) ] . after three years of onset of the disease , family members noticed that while walking he was having lateral flexion of trunk and was leaning on left side ( see video ) . along with this he also developed striatal hand ( figure 1 ) . during supine position x - rays whole spine , mri brain and paraspinal electromyography ( emg ) were normal . this patient of pd was diagnosed to have pisa syndrome due to lateral flexion and axial dystonia on standing which used to completely improve in supine position . pisa syndrome is a combination of lateral deviation of spine and corresponding tendency to lean on one side.3 exact pathogenesis of ps is still not known . it is believed that cholinergic excess in patients of ad receiving cholinesterase inhibitor cause ps . in patients of pd it may be possible that striatal dopamine deficiency or imbalance in dopaminergic - cholinergic level is responsible for ps.4 this imbalance seems to be asymmetrical and is responsible for the lateral flexion on one side . our patient also had significant improvement in lateral flexion on standing during on period , however striatal hand symptoms showed no response to levodopa / carbidopa . it has been described that pd subjects in advanced stage of disease ( hy iii & iv ) have more postural abnormality and striatal deformities while our patient manifested in early part of disease ( hy iib).3 dopa agonists and amantidine has been tried with variable response . neurosurgical interventions like pallidotomy and deep brain stimulation has also been tried in 13 subjects with good results.5

pisa syndrome is ( ps ) usually seen in patients receiving antipsychotic drugs and characterised by lateral flexion of trunk and axial dystonia . it is believed that antipsychotic drugs lead to dopamine blockage causing ps . we describe a parkinson s disease patient who was doing well with levodopa / carbidopa for 3 years and developed lateral flexion of trunk . his abnormal posture used to completely improve upon lying down position . he also had striatal hand deformity suggestive of focal dystonia .

a 12-year - old male , whose father was a ldl receptor ( ldl - r ) mutation carrier , developed corneal arcus and multiple skin lesions with a 10-year history of xanthomas . both his parents had elevated levels of total serum cholesterol and ldl cholesterol . his elder brother died of myocardial infarction secondary to fh at the age of 7 years . physical examination showed the presence of subcutaneous yellow nodules at the knuckles of his fingers [ fig . some other yellow nodules of varying sizes under the skin erupted over the buttocks [ fig . the ocular exam revealed a partial circumferential white - grey deposit corresponding to corneal arcus [ fig . b - scan revealed extensive plaques and enhanced intima - media thickness of common carotid arterial wall . laboratory studies disclosed the following values : total serum cholesterol , 752.1 mg / dl ( normal range , 110 - 220 mg / dl ) ; triglyceride , 96.6 mg / dl ( normal range , 50 - 150 mg / dl ) ; ldl cholesterol , 661.3 mg / dl ( normal range , 80 - 140 mg / dl ) . findings were consistent with type iia hyperlipoproteinemia . as is shown in the figure [ fig . 3 ] , there are cytosine ( c)>thymine ( t ) heterozygous double peaks at 97 in the second exon of ldl - r gene , which is in genbank as a known mutation of nm_001195798.1:c . this mutation resulted in the change from c to t 33 codon in e2 of ldl - r gene and , thus , glutamine became the stop codon in the corresponding amino acid ( np_001182732.1:p.gln33x ) . findings at presentation ( a ) xanthomas over the fingers ( b ) xanthomas over both elbows(c ) xanthomas over both knees tending to coalesce ( d ) xanthomas over the achilles tendons ( e ) xanthomas of varying sizes under the skin erupted over the buttocks findings at presentation . the ocular exam showed a partial circumferential ( from 2 oclock to 4 oclock ) white - grey deposit corresponding to corneal arcus . ( a ) right eye the ocular exam showed a partial circumferential ( from 2 oclock to 4 oclock ) white - grey deposit corresponding to corneal arcus . there are c > t heterozygous double peaks at 97 in the second exon of ldl - r gene homozygous fh is clinically characterized by cutaneous xanthomas , enlarged achilles tendons , atherosclerosis , and corneal arcus , usually developing from early childhood . although no significant correlations were obtained between corneal arcus and patterns of hyperlipoproteinaemia in previous observations , a recent study of homozygous familial hypercholesterolemia series indicated that patients with corneal arcus had higher cholesterol - year score and was correlated with calcific atherosclerosis . one explanation is the proximity to limbal vasculature that may increase endothelial permeability to lipids via active scavenging mechanisms . another is the temperature gradient that can alter lipid deposition as the infiltrating particles pass from limbal blood vessel into the cornea . early combination therapy with ldl apheresis , statins , and cholesterol absorption inhibitors are advised in children with homozygous fh at the highest risk .

we report the case of a 12-year - old male who developed corneal arcus and multiple skin lesions with a 10-year history of xanthomas . the lesions appeared over his fingers , hands , elbows , knees , buttocks and feet . laboratory studies showed a total serum cholesterol level of 752.1 mg / dl ; a triglyceride level of 96.6 mg / dl ; a low - density lipoprotein cholesterol level of 661.3 mg / dl . findings were consistent with homozygous familial hypercholesterolemia . to our knowledge , this is the first such case to be reported from china .

patients with eos usually present in the fourth and fifth decades of life unlike skeletal osteosarcoma patients who present in the fifth and sixth decades of life . there are also reports of eos involving the face , breast , abdominal wall , soft tissues of the back and retroperitoneum , and site of a vaccination scar . found that 10% of the patients had previous irradiation and 13% of the patients had history of trauma related to the site of eos . the genetic change most commonly associated with osteosarcoma is the loss of the p53 tumor suppressor gene on chromosome 17 and loss of retinoblastoma gene rb1 on chromosome 13 . a genetic association between paget 's disease and osteosarcoma has also been identified on chromosome 18q . other genomic changes , such as loss on 13q and 15q and gain on 1q and 8q , have been detected in patients with eos . the contribution of the major susceptibility genes on chromosome 10q ( ret proto - oncogene ) is well established in hd . in addition , potential modifying associations exist with chromosomes 2 , 9 , 13 , 20 , 21 , and 22 . variations of main ret proto - oncogene account for as much as 50% of familial and 2030% of sporadic cases . the etiology of the majority of sporadic cases is not clear , appearing to arise from combined cumulative effects of susceptibility loci on other chromosomes controlling the mechanism of cell proliferation , differentiation , and maturation . presence of genitourinary abnormalities in the form of horse - shaped kidney and azoospermia , and sporadic hd pointed toward a genetic abnormality in our patient . hence , we performed genetic screening at the common sites for both hd and osteosarcoma on chromosome 10 ( ret proto - oncogene ) and chromosome 17 ( p53 tumor suppressor gene ) . however , we found no abnormalities at both these loci , suggesting a role of combined cumulative effects of susceptibility genes on other chromosomes . inactivation or alteration of a gene located at this susceptibility loci could have been an early event in the development of eos in our patient . the cornerstone of treatment consists of radical surgery and polychemotherapy . due to the rare nature of this disease , no data from randomized studies concerning the type of chemotherapeutic combinations are available . in a small retrospective study , goldstein - jackson et al . reported favorable results when eoss were treated as conventional osteosarcomas . active chemotherapeutic agents for skeletal osteosarcoma consist of a combination of doxorubicin and cisplatin , with or without the addition of other drugs such as methothrexate , ifosfamide , and etoposide . the use of multi - agent chemotherapy has extensively improved the outcome for skeletal osteosarcoma . however , these treatments are very intensive , and therefore acute and delayed toxicities can be expected . doxorubicin can cause bone marrow toxicity and cardiotoxicity , whereas cisplatin can cause nephro - and ototoxicity . adjuvant radiotherapy does not increase the survival when patients are treated with effective surgery and chemotherapy , and increase the risk for secondary tumors . however , radiation should be considered in the setting of an unresectable or incompletely resected primary tumor . in this case , the tumor was completely resected , but the resection margins were close due to the orbital localization of the tumor , making wide resection impossible . the first case was an 11-year - old boy who developed an eos of the orbit following radiotherapy for retinoblastoma in infancy . the second patient was a 22-year - old otherwise healthy male with no predisposing factor . our case is the oldest patient ever detected with an eos in the orbit with no history of previous radiotherapy or trauma . one could suggest a possible association between eos and patients known hd , but this has never been reported in the literature before .

primary extraskeletal osteosarcoma ( eos ) is an extremely rare malignancy . in this report , the clinical course of a 32-year - old man presenting with proptoses is described . medical history included hirschsprung disease ( hd ) , horseshoe kidney , azoospermia , and vertebral anomalies . imaging of the orbit showed an oval , well - defined heterogeneous mass adjacent to the lateral wall of the orbit . the patient underwent a lateral orbitotomy and complete excision of the mass . the mass was not attached to the bone . histopathologic and immunohistochemical examination confirmed the diagnosis of an eos . the patient received chemotherapy and radiotherapy and is free of the disease 3 years after the diagnosis . genetic screening showed no mutations for both the ret proto - oncogene for hd and the p53 tumor suppressor gene for osteosarcoma .

a 20-year - old female with extensive terminal ileal and colonic crohn 's disease was initially successfully managed on a drug regimen of prednisolone and mesalazine which helped to induce remission . this maintained her disease remission but was associated with neutropenic sepsis , necessitating the withdrawal of the thiopurine therapy with subsequent clinical and endoscopic disease flare . this required an infusion dose and frequency escalation to maintain clinical response . after 58 weeks of infliximab therapy , the patient was requiring 10 mg / kg 6 times weekly and hence was transferred to the fully human anti - tnf- injection adalimumab for loss of response to infliximab . initially this strategy regained disease control , but due to relapse at 40 mg every other week , the dose was increased step - wise to 80 mg weekly which fortunately maintained remission . during her treatment with adalimumab the presence of purple - coloured plaque - like lesions primarily on her abdomen figure 1 demonstrates the presence of psoriasis which is presumed to be a result of treatment with adalimumab . tnf- has been identified as a key cytokine in the pro - inflammatory pathways that drive diseases such as inflammatory bowel disease and rheumatoid arthritis . anti - tnf- agents such as infliximab and adalimumab are widely used in the treatment of these conditions and have been shown to have an impact on pro - inflammatory and apoptotic pathways . they are also effective in the treatment of the dermatological and rheumatological manifestations of psoriasis . however , there have been a number of cases reported where such agents may be responsible for new onset or worsening of psoriasis . a study of 126 patients who underwent treatment with anti - tnf- inhibitors demonstrated evidence of psoriasis in 76 individuals , palmoplantar pustular psoriasis in 37 cases and psoriasis of the nails in 6 cases . there is mounting evidence that a key innate immune pathway for triggering common human autoimmune diseases , including psoriasis , involves plasmacytoid dendritic cell precursors and type 1 interferon ( ifn ) production . a recent study involving immunohistochemical staining of skin biopsy specimens for myxovirus - resistance protein a ( mxa , a surrogate marker for lesional type 1 ifn activity ) demonstrated increased staining in tnf- inhibitor - induced psoriasis compared with psoriasis vulgaris . research suggests that a psoriasiform eruption during anti - tnf- treatment seems to be a class effect without any confirmed predisposing factors and does not specifically require treatment discontinuation . further studies are however needed to determine the exact mechanism of and appropriate treatment for anti - tnf--induced psoriasis .

anti - tnf- agents are currently utilised for the treatment of a vast array of autoimmune conditions including inflammatory bowel disease , rheumatoid arthritis , ankylosing spondylitis and psoriasis . it is however noted that such therapeutic strategies have been linked to the specific induction of cutaneous - based reactions such as dermatitis , erythema multiforme and psoriasis . here we present the case of a young female patient with crohn 's disease who developed psoriasis following treatment with the anti - tnf- drug adalimumab and highlight the possible pathogenetic mechanisms involved in such an occurrence .

cutaneous paraneoplastic syndromes include acanthosis nigricans , the sign of leser - trlat , eruptive acrochordons , acquired ichthyosis , erythroderma , palmoplantar hyperkeratosis , acquired hypertrichosis lanuginosa , erythema gyratum repens , necrolytic migratory erythema , bazex 's syndrome and dermatomyositis . the presence of either syndrome in patients warrants evaluation for a possible underlying malignancy . here we report the case of a 79-year - old man , who developed numerous several centimeter - large seborrheic keratoses on his chest and back within the time period of one year . workup led to the diagnosis of colonic adenocarcinoma , underscoring the paraneoplastic etiology of the seborrheic keratoses in this patient . a 79-year - old man with a previous diagnosis of chronic lymphocytic leukemia was seen for preoperative evaluation prior to elective right peripatellar bursectomy after having failed antibiotic treatment . on physical examination numerous seborrheic keratoses in a christmas tree pattern were noted on his chest and back ( fig . the patient reported that the lesions had appeared and advanced within the past twelve months on his trunk . he was seen by a dermatologist , who suggested performing endoscopies of the upper and lower gastrointestinal tract . the patient had not noted clinical symptoms otherwise suggestive of colon cancer , such as unintentional weight loss or recent change of bowel habits . the clinical presentation of the seborrheic keratoses in this patient was found to be consistent with the sign of leser - trlat . leser - trlat is a rare dermatologic finding named after two surgeons , the german edmund leser ( 18281916 ) and the french ulysee trlat ( 18271890 ) . however , the first descriptions of patients with characteristic signs of leser - trlat were published in 1900 and 1901 by the german dermatologist eugen hollnder . the leser - trlat sign is characterized by the sudden onset of multiple seborrheic keratoses or an unusually rapid increase in size and number of preexisting seborrheic keratoses , occasionally on an inflammatory base . it is commonly associated with malignancies , primarily gastrointestinal adenocarcinomas [ 5 , 6 ] , but also squamous cell carcinomas , leukemias and lymphomas . although patients with nonmalignant conditions , such as pregnancy , hiv infection or after having received heart transplantation , can also develop seborrheic keratoses of florid eruption , the term leser - trlat usually implies paraneoplasia . the significance of this sign has been debated in the medical literature , as leser - trlat is observed primarily in elderly individuals , who are statistically at higher risk for developing both seborrheic keratoses as well as various malignancies . therefore , accurate diagnosis is linked to the rapidity of onset of seborrheic keratoses , and to the growth behavior of previously noted seborrheic keratoses . only rarely has leser - trlat been reported in younger patients . the seborrheic keratoses seen in leser - trlat differ neither morphologically nor histologically from commonly seen seborrheic keratoses . they are found predominantly on back and chest of patients , and there is no predilection for patients gender or ethnic / ancestral background . the most prevalent clinical symptom is pruritus , seen in 43% of malignant cases [ 12 , 13 ] . in approximately 20% of cases acanthosis nigricans is also present , either at the time of cancer diagnosis or it develops subsequently . while the pathogenesis of this condition is unclear and remains to be elucidated , due to the proliferative nature of cutaneous paraneoplasias , a possible pathogenic role of epidermal growth factor and alpha - transforming growth factor in the development of the seborrheic keratoses associated with malignancies has been hypothesized and reported . on rare occasions , skin manifestations similar to the leser - trlat sign can be mimicked by malignant melanoma , thus justifying excision biopsies and histologic examinations of suspicious lesions in distinct cases . florid eruption or abnormally fast growth of seborrheic keratoses especially in elderly patients should always raise the suspicion for an underlying malignant disease , in particular for gastrointestinal adenocarcinomas . performing an endoscopic evaluation of the gastrointestinal tract of affected patients

we report the case of a 79-year - old caucasian man , who developed numerous pruritic seborrheic keratoses on his chest and back within one year . an underlying malignant disease was suspected . upper and lower endoscopies were performed and the patient was diagnosed with adenocarcinoma of the ascending colon . we discuss the clinical significance of his cutaneous symptoms / signs , and review the medical literature on the paraneoplastic sign of leser - trlat .

a 12-year - old female visited the hospital with the chief complaint of an abnormal finding on a simple chest x - ray . a discontinuity of the diaphragm was observed in the retrosternal portion by chest ct and mesenteric fat was herniated through this discontinuity ( fig . the lack of an intestinal hernia was confirmed by barium esophagography , and there was no finding of incarceration . although the patient was 12 years old , her height was above average for her age at 164 cm . thus , it was determined to perform a laparoscopic procedure considering that the surgical space for the laparoscopic procedure would be large enough , given her height . a 10-mm 30 degree laparoscope was inserted through an upper umbilical incision . to insert additional surgical instruments , a 10-mm troca was inserted through the left flank , the right lower rib , and the right flank incision . the diaphragm injury was observed on the left side of the falciform ligament in the peritoneal cavity ( fig . the great omentum and fat from the falciform ligament were put in the thoracic cavity . the great omentum , which was pushed into the thoracic cavity by the diaphragm injury , was recovered . the falciform ligament was separated from the margin of the thoracic wall to secure space for the operation . after resection of adipose tissue from the falciform ligament , the hernia sac was closed by tying up the mediastinal pleura and the right diaphragm as is done for plication . to avoid creating a dead space in the pleura cavity , an incision of about 1 cm was made in the mediastinal pleura before tie - up to make the dead space disappear by inflation of the lung . the right margin of the diaphragm defect was stretched in the lateral direction to adjust the edge of the diaphragm . a chest tube was inserted to treat a pneumothorax which occurred right after the operation . the patient was discharged on the third day after the surgery without special complications , and her progress was observed in the outpatient department ( fig . morgagni hernias , which are the rarest type among the congenital diaphragmatic hernias , comprise 3~5% of all diaphragmatic hernias . however , other cases are found in adulthood and pregnancy , the latter of which makes abdominal pressure increase because it is usually asymptomatic . according to the research of comer et al . acute symptoms form in only 14% of cases ; however , surgical treatment should be considered because there is a risk of incarceration by an intestinal hernia . in addition , this approach has an advantage in case of adhesion between the pericardia and lungs . on the other hand , disadvantages of this approach are that it is difficult to repair bilateral hernias and to diagnose the obstruction of intestin . herniated organs can be returned to their original places easily with an approach through the abdomen . particularly , this approach should be used in case of bilateral and central hernias , and combined abnormalities in the abdomen . therefore , to prevent excessive pressure on the intestine , the diaphragm injury lesion should be anchored retrosternally . however , the argument for the minimally invasive surgical technique still stands because this procedure is difficult . to compliment this procedure , the method in which the diaphragm defect is sutured on the abdominal wall and the suture is fixed to the subcutaneous layer of the abdominal wall has been developed . in the present case , a horizontal procedure with laparoscopy to treat a morgagni hernia is not a difficult method . however , follow up for cases of pregnancy and trauma , which cause an increase in abdominal pressure , is essential .

a 12-year - old female presented with the abnormal findings on the chest pa . the chest ct revealed a retrosternal defect of the diaphragm and a fatty opacity in the pleural cavity , resulting in a diagnosis of morgagni hernia . it was decided to undergo a laparoscopic surgery . the retrosternal defect of the diaphragm measuring 3.5 cm in diameter was found , through which a portion of the greater omentum and the fatty tissue connected with the falciform ligament were herniated into the pleural cavity . the greater omentum was pushed back into the peritoneal cavity and the fatty tissue connected with falciform ligament was excised . the mediastinal pleura was plicated and the defect of the diaphragm was repaired primarily . immediately after the operation , the patient developed a right pneumothorax for which a chest tube was inserted . she was discharged at the post - operative third day without any further complications .

paradoxical enlargement of lymph nodes in tuberculosis is well established in the early phase of treatment.[13 ] this is more common in hiv co - infection . mediastinal lymphadenopathy resulting in recurrent laryngeal nerve palsy , pulmonary artery occlusion and superior vena caval obstruction has been described . we report a case of bronchial obstruction in a patient with fully sensitive organism in the late phase of the treatment . a 26-year - old somalian woman was diagnosed with mycobacterium tuberculosis infection in january 2008 . she presented with cervical and mediastinal lymph node enlargement without any obvious lung parenchymal involvement . acid fast bacilli ( afb ) were seen on fine needle aspiration of the cervical lymph node . after initial problems with compliance , full standard quadruple therapy was effectively started in april 2008 . maintenance treatment was commenced in june 2008 . the chest x - ray ( cxr ) on first presentation did show a narrowed left main bronchus in the absence of any clinical signs or symptoms . the only relevant past medical history was of childhood asthma , for which she had no hospitalizations and did not use any regular inhaled therapy . she presented in august 2008 to the accident and emergency department with a one - week history of worsening shortness of breath . inhaled steroids and bronchodilators were started for a possible asthma exacerbation and she was discharged home . her symptoms deteriorated and when seen in the tb clinic four weeks later , her exercise tolerance was limited to about 100 yards . on examination , a repeat cxr showed marked narrowing of the left main bronchus , confirmed on ct scan which showed almost complete occlusion of the left main stem bronchus . spirometry showed fev1 - 1.09 l / min ( 36.3% of predicted ) , fvc 1.44 ( 41.7% of predicted ) [ table 1a ] . the flow - volume loop had a characteristic shape of large airway obstruction [ figure 1 ] . spirometry at presentation flow - volume loop of the patient which is characteristic of large airway obstruction she was commenced on oral prednisolone 30 mg once daily . there was no concern with compliance at this time . a bronchoscopy also showed complete occlusion of left main bronchus with white necrotic material [ figure 2 ] . white necrotic matter seen occluding the left main bronchus at bronchoscopy her treatment was continued with both anti - tuberculous therapy and steroids . three days prior to the clinic visit she said she had coughed up a large lump of white material and her breathing had returned to near normal since . 2.81(81.6% ) [ table 1b ] with normalization of the flow volume loop [ figure 3a ] . bronchoscopy was repeated and this showed a patent left main bronchus with some necrotic material on the lateral wall [ figure 3b ] . spirometry after treatment ( a ) flow - volume loop improved with less evidence of airway obstruction , ( b ) patent left main bronchus with only a small amount of necrotic matter remaining at repeat bronchoscopy she continued with anti - tuberculous treatment for a full one year and steroids were slowly tapered off . her cxr showed marked improvement in the narrowing of the left main bronchus towards the end of treatment . endobronchial tuberculosis is less common since the advent of effective anti - tuberculous chemotherapy , but mediastinal lymphadenopathy due to mycobacterial infection is still common in developing countries . paradoxical reactions on the whole are more common with hiv co - infection ( 28% ) than without ( 10% ) . lymph node enlargement is the most common presentation and bronchial obstruction due to mediastinal lymphadenopathy is more common in the pediatric age group . in adults , patients with tuberculous mediastinal lymphadenopathy presenting with recurrent laryngeal nerve palsy , pulmonary artery occlusion and superior vena caval obstruction have been described . these presentations may be due to either external compression or erosion into the surrounding structure . in this particular case , the bronchoscopy showed caseating material eroding into the left main bronchus . nakvi et al . , described this in children , in whom the obstruction was relieved by either aspiration or surgical excision . response to steroids has not been consistent in many series described but generally paradoxical reactions , unlike endobronchial tb , are thought to be more steroids responsive . our patient responded well to steroids . even after coughing up necrotic debris and relieving endobronchial obstruction late paradoxical reactions should be considered in any patient on anti - tuberculous chemotherapy presenting with stridor .

we present here a case of bronchial obstruction secondary to late paradoxical reactive enlargement and erosion by mediastinal lymph nodes into the left main bronchus in a 26-year - old woman with tuberculosis lymphadenitis . bronchial obstruction due to paradoxical reactions , especially in the late phase of treatment , has not been described in adults before .

in this issue of critical care , zwiers and colleagues describe the incidence of acute kidney injury ( aki ) in neonates on extracorporeal membrane oxygenation ( ecmo ) based on a 12-year cohort study from sophia children s hospital pediatric intensive care unit in rotterdam , the netherlands . of major interest , this study is looking at patients during an era when continuous renal replacement therapy ( crrt ) was not systematically used , giving a clearer perspective on the natural kinetics of renal function , estimated by serum creatinine ( scr ) , during ecmo course . interestingly , the worst renal function occurred early during the ecmo run , within the first 2 days , and with a 2- to 4-day delay until renal function improvement could be observed . this suggests that aki is an early event in newborns requiring ecmo support . establishing the natural history of renal function in patients with multiple organ dysfunction syndrome ( mods ) gives us the expected time period during which one may , it would be valuable to know to what extent improving renal function during ecmo course really is associated with better outcome . better characterization of mods timing in the course of heterogeneous life - threatening diseases is central . international integrated database for the evaluation of severe sepsis and drotrecogin alfa ( activated ) therapy database analysis , regrouping 4,459 patients with sepsis , showed that worsening organ failures particularly , cardiovascular , respiratory , renal , and hematologic failure was the cause of death in patients with sepsis . interestingly , it showed that aggravating renal sequential organ failure assessment score was a late event close to death . one can hypothesize that bi - modal impairment in renal function should result in a more severe outcome . in children and neonates , similar data were published . aggravating pediatric logistic organ dysfunction score within the first 4 days was associated with a 50% mortality . however , in newborns , renal dysfunction does not seem to have a major impact on mortality . in the present study , the authors showed that severity of renal failure was strongly associated with mortality ; the odds ratio of mortality for acute kidney failure ( identified as an scr of greater than 300% of normal value ) was 12.7 . this suggests that , although knowing aki natural course during ecmo may give arguments for delaying crrt , there is clearly a cutoff value at which aki severity directly impacts mortality . it is not the purpose of this commentary to discuss the ideal timing of crrt during ecmo , because many questions remain to be debated , but this study confirms that aki is a major outcome determinant in the course of ecmo . if not an unresolved dilemma , aki in the course of mods remains a circular reference : aki in patients requiring ecmo therapy is triggered by initial systemic insults , already present at the time of ecmo initiation , and aggravated by reperfusion injury and ecmo circuit - induced inflammation . the close interplay between the cause and severity of the disease , supportive therapies , and host response is influencing the development of aki and mods . identifying as well as determining the relative importance of extrinsic factors a ggravating mods / aki during ecmo therapy is central . besides technologic advances in ecmo circuit biocompatibility improvement reperfusion as well as function , identifying deleterious therapeutic interventions , and improving standard therapies , such as antimicrobial chemotherapy . recently , it was shown in an autopsy series of patients dying of septic shock that renal dysfunction was due mostly to focal renal tubular injury through apoptosis and not autophagy . that study clearly suggested that renal dysfunction found in patients dying of sepsis - induced mods is reversible . ecmo is certainly among the greatest recent advances , making a surgical theater technique widely available in the critical care setting for clinical conditions far away from the original cardiovascular indications . however , a lot remains to be done in order to identify proper indications , optimize all organ support , reduce complications , and better characterize pathophysiologic and cellular mechanisms of organ dysfunctions in children under ecmo . aki : acute kidney injury ; crrt : continuous renal replacement therapy ; ecmo : extracorporeal membrane oxygenation ; mods : multiple organ dysfunction syndrome ; scr : serum creatinine .

whether the egg or the chicken came first remains debated , although the debate is not critical for those who love eating omelets . at least , it is well accepted that one needs to break eggs to fry an omelet . in some ways , there are similarities with acute kidney injury . a lot of debate arises from whether acute kidney injury is a complication of various insults and therapies or an expected continuum of the organ failure process . indeed , it is well accepted that acute renal failure is associated with increased mortality in severely ill children and infants .

the meeting was introduced by edwin l. cooper , a very communicative and scientifically participative professor , who illustrated the main aims of the journal ecam , evidence - based complementary and alternative medicine , and roused everyone to contribute publishing scientific - based evidences on homeopathy and other complementary / alternative medical approaches . he described how the journal born in los angeles ( ucla ) sustained by important collaboration in kanazawa and japan , with assistance from oxford university press . professor cooper illustrated the electronic submission of all contribution types and hastened peer reviewing that results in free access to electronic publication before the appearance of a hard copy issue . professor paolo bellavite opened the conference by discussing the historical background of homeopathy , and then developed into a session of open - ended questions . what can be concluded through meta - analysis ? is it possible to imagine the future of the homeopathic approach by either exceeding and/or incorporating the different doctrines ? 1 ) ; they shape a triangle whose vertices are ( from the top ) as follows : complexity and individuality , similarity and dilutions . daily medical practice and clinical studies lie on the sides of the triangle and within its area , sustained and supported by other scientific trials : from phase i clinical trials to phases ii iv trials , from basic research on the similia principle to electromagnetic properties of water passing through the paradox of very high dilution . taken together , this could increase the area of the triangle , which determines the acceptance of homeopathic medicine . similarity : healing is achieved by taking a drug that proved by healthy individuals have yielded symptoms and signs very similar to those of the patients . similarity : healing is achieved by taking a drug that proved by healthy individuals have yielded symptoms and signs very similar to those of the patients . the published homeopathic clinical trial and meta - analysis review was very detailed ( as many as 80 studies were reviewed ) . results varied , and the following observations were extensively discussed during the course of the conference : ( i ) homeopathic research requires more rigorous trials ; ( ii ) clinical studies on asthma , allergies and other respiratory pathologies yielded the best results ; and ( iii ) classic homeopathy requires more specific trail methodology . special attention was given to a recent famed meta - analysis from the lancet ( 1 ) which addressed the prejudice toward homeopathy , primarily due to the choice of trials that were analyzed . homeopathic trails must employ more rigorous methodologies : they frequently lack in randomization criteria , placebo use and laboratory markers . the speaker stressed efficacy in homeopathy , which could be evidenced both referring to complex therapeutic method ( the use of individualized therapy ) and to specific drug effects on specific pathology . provings , i.e. homeopathic pathogenetic trials ( htps ) , contributed by professor giuseppina pitari . milestones of homeopathic medicine , htps suffer about large methodological variability : different aims , different described outcomes , placebo usage , supervising , poor remedy description , multivariegated symptoms collections , lack of data analysis , etc . a possible methodology was described and some indications were discussed on study design ( double - blind placebo - controlled trial ) ( 2 ) , doses and potency of the drug , description of the potentized substance detailing its toxicological effects , time of observation . carlo m. rezzani concluded the session by describing a research project ( ciflicol ) on clinical report cases : an electronic case sheet can be drawn up and sent to a worldwide database , continuously updated ( www.hmssrl.com ) . the intriguing discussion about placebo solution preparation was flowed into the description of the world of high dilutions. the most characteristic and controversial principle of homeopathy is that the potency of a remedy can be enhanced by dilution , in a procedure known as dynamization or potentization. paolo bellavite discussed about basic researches showing limits , successes and possible hypotheses . life has evolved around water , into water , because of water : special water properties permit hydrophobic interaction , very few water molecules go with biological compounds keeping its imprint , water clusters activated during homeopathic dynamization can reach a cellular receptor and trigger specific responses . papers on animal or in vitro models showed the effects of very diluted and potentized on human basophils , chicken embryos , rat duodenum , mouse blood , etc . hormetic effect were reviewed : stimulus or molecule different doses trigger opposite effects on the same receiver system . as paolo bellavite clearly showed as a system 's ( cells , organs and organisms ) starting conditions can be crucial to treatment results and as some drug effects can be paradoxical , thus supporting the possible use of similia principle as curative efficient approach ( 3 ) . at the end of the conference the speaker expounded coherence of homeopatic medicine towards the dynamic complexity of diseases . homeodynamic conditions make possible the organized complexity of life and a pharmacological complex information mimicking disease via high homeopathic dilutions are reported to be effective both in humans and animal / in vitro models . methodological problems in clinical trials can be overcome drawing up a specific approach to clinical homeopathic research with a large approved consensus .

verona 's school of homeopathic medicine ( www.omeopatia.org ) organized a day of full immersion in the field of homeopathy , focusing on the validity of this much - debated discipline . there is widespread consensus in the medical community that evidence - based medicine is the best standard for assessing efficacy and safety of healthcare practices , and systematic reviews with strict protocols are essential to establish proof for various therapies . students , homeopathic practitioners , academic and business representatives , who are interested in or curious about homeopathic practices attended the conference .

we have introduced and reported a potential novel clinical indicator for keratoconus ( kcn ) cornea instability assessment based on consistent , increased overall corneal epithelial thickness . as in our previous report , most epithelial measurements have been performed in vivo , by high - frequency scanning ultrasound biomicroscopy ( hf - ubm ) . we report herein similar data using anterior segment optical coherence tomography ( as - oct ) . we present the case of a 25-year - old male subject , diagnosed with topographic and topometric asymmetrically manifested kcn among his two eyes . 170. corrected distance visual acuity with this prescription was 20/20 + . the left eye ( os ) , the more symptomatic one , had an uncorrected distance visual acuity of 20/80 and a manifest refraction 7.00 3.50 the kcn diagnosis was further confirmed by corneal tomography imaging , obtained by wavelight oculyzer ii ( alcon surgical , ft . , usa ) which is a pentacam hr ( high - resolution ) scheimpflug imaging camera [ 2[ga1 ] ] . four consecutive corneal scans in each eye were obtained by as - oct rtvue-100 ( optovue inc . , examples of od and os epithelial pachymetry maps ( 6 mm diameter ) are provided in fig . repeatability , which can be assessed by the standard deviation of the central epithelium of the 4 scans , was 0.60 and 1.50 m in od and os , respectively . 2a , od had a central epithelial thickness ( epi ct ) of 52.23 0.60 m . overall epithelial thickness ( epi average ) was computed by taking the average of the 17 segment thickness points shown in fig . 1c , d. the values displayed in each of the 17 segments are the average of the epithelial thickness measurements within each segment . od had an epi average of 51.97 0.70 m . os had an epi ct of 56.75 1.50 m and an epi average of 55.65 1.22 m . the epithelial topographic thickness variability ( epi variability , fig . our results show that on average , the epi variability was 1.53 0.21 m for the less affected od , while it was 9.80 0.41 m for the more affected os . hf - ubm has known intrinsic examiner operative challenges ( both for capture and interpretation ) , and although available for over a decade , it has seen very little clinical use and mainstream practice acceptance . clinical availability of corneal epithelial mapping by as - oct is currently in practice , and introduces a simple and effective clinical tool for corneal epithelial mapping . the data are very easily captured and epithelial thickness parameters are automatically calculated by a novel software algorithm and presented in topographic imaging [ 3 , 4 ] . this case illustrates a vivid agreement with previous findings by kanellopoulos et al . that an overall thicker epithelium with large variations can be observed in the topographically evident kcn eye , and to a much lesser degree in the less affected eye . qualitatively , the defining feature of the epithelium in kcn is the pattern , such that there is a thin epithelium over the cone surrounded by a thicker epithelium as can be seen in fig . 1d . quantitatively , epithelial thickness variations can be expressed as the topographic thickness variability and are computed by the standard deviation of the 17 segment thickness points . specifically , in our case , it was 9.80 m in the affected kcn eye and 1.53 m in the less affected eye . in fig . 2b , the graph compares the corresponding values of epithelial thickness from the hf - ubm study and the case described herein . this case not only confirms our findings regarding the increased overall epithelial thickness in ectatic eyes , but also that the new as - oct feature of epithelial imaging may illustrate these differences in a more pronounced way , as indicated by the data shown in fig . the ease of use of as - oct epithelial imaging in comparison to hf - ubm as well as the increased predictability that may be offered by as - oct epithelial imaging may hold promise for wider clinical application , such as the screening of young adults for early kcn and , in a much wider perspective , potential candidates for laser cornea refractive surgery .

purposeto evaluate safety , efficacy and ease of measurement of epithelial thickness in a keratoconic patient based on anterior segment optical coherence tomography ( as - oct).methodsa 25-year - old male patient , previously diagnosed with keratoconus , with highly asymmetric manifestation among the two eyes , was subjected to as - oct corneal epithelial imaging . we investigated epithelial thickness and epithelial topographic thickness distribution.resultsmean epithelial thickness was 51.97 0.70 for the less affected right eye ( od ) , and 55.65 1.22 for the more affected left eye ( os ) . topographic epithelial thickness variability for the od was 1.53 0.21 m , while for the os it was 9.80 0.41 m.conclusionsthis case further supports our previous findings with high - frequency ultrasound measurements of the increase in overall epithelial thickness in keratoconic eyes in comparison with normal eyes . as - oct further offers ease of use and possibly higher predictability of measurement . this case report , based on as - oct imaging , verifies increased overall epithelial thickness in keratoconic eyes , as introduced by a previous study [ kanellopoulos et al . : clin ophthalmol 2012;6:789800 ] , based on high - frequency scanning ultrasound biomicroscopy imaging .

granulomas are focal accumulations of epithelioid cells , typically with a rim of lymphocytes and fibroblasts , representing a delayed - type hypersensitivity reaction in response to antigenic stimulation . this stimulation is usually triggered by exposure to antigen that can not be degraded or in cases of immune dysfunction . liver granulomas have been reported in 215% of unselected liver biopsies , with the etiology varying according to geography . in the us , 75% of cases are result in sarcoidosis , mycobacterial infection , primary biliary cirrhosis and drug - induced liver injury . in other countries , such as iran , mycobacterium tuberculosis and visceral leishmaniosis account for 60% . studies have shown that 615% of granuloma findings are idiopathic [ 1 , 2 ] . few histological features associated with certain diseases include caseous necrosis in acid fast bacilli containing granulomas , or ova in schistosoma mansoni . a 38-year - old male sudanese immigrant pronounced brain dead after a motor vehicle accident became an organ donor for liver and kidneys . his serology came back positive for hepatitis b core antibody and surface antibody , hbsag negative , hepatitis c and hiv negative . gross appearance of donor liver gave suspicion of granulomas based on surface heterogeneity ( fig . his liver biopsy at the donor hospital was consistent with < 5% macro and micro steatosis , no fibrosis but significant for the presence of having multiple granulomas ( fig . the initial slide review by the pathologist found multiple granuloma , and requested re - biopsy from the donor liver . the re - biopsy result found parasitic disease in the hepatocyte that later found to be schistosomiasis ( fig . lack of data regarding the behavior of this disease in the recipient , the biopsy result caused us to decline the liver due to the parasitic liver disease . schistosomiasis is a chronic enteral parasitic disease caused by trematode worms of the genus schistosoma . it is estimated that over 258 million people are affected yearly , with most of its disease burden in africa , south america , the caribbean , the middle east and asia . at the present time , due to increased global mobility , schistosoma may be encountered in non - endemic areas . over 90% of the cases of schistosomiasis are currently found in sub - saharan africa , where more than 200 000 deaths occur yearly due to the disease . schistosomiasis is the most prevalent parasitic disease in sudan due to the wide river basin areas and large irrigated agriculture along the nile rivers . once infection occurs , the adult worms reside in veins releasing eggs that are either shed into the environment through feces or urine , or are retained in host tissues where they induce inflammation and then die . when schistosoma eggs are trapped in tissues , they activate an immunologic reaction leading to the development of granuloma and fibrosis . hepatic schistosomiasis leading to severe fibrosis is a well - recognized cause of chronic liver disease and portal hypertension . it results from the host s granulomatous cell - mediated immune response to the ova antigen and progressing to irreversible fibrosis . multiple factors influence the development and pathology of schistosomiasis including the type of immune response developed by the host , host genetic background , intensity and number of infections . most schistosomiasis is caused by schistosoma hematobium , s. mansoni and s. japonicum ; however , s. mansoni and s. japonicum tend to cause hepatobiliary disease , while s. hematobium mainly affects the urinary tract . due to the life cycle of schistosoma , the acute phase of infection is usually asymptomatic , the only chronic sequelae identified via tissue biopsy is necrotic eggs with granuloma formation . strategies such as living donation , split liver transplantation , domino liver transplantation and the extensive use of marginal donor grafts have been implemented in many countries . as a result of this , there are an increasing number of unusual diseases from asymptomatic donors , including schistosomiasis . reports have shown schistosomiasis transmission through liver grafts from deceased donors with no consequences for graft or recipient survival . a retrospective study in brazil highlights cases of living donor liver transplantation ( ldlt ) with schistosomiasis diagnosed on post - reperfusion liver biopsy showing granulomatous reaction with s. mansoni eggs in transplant recipients . also describe the first intentional transplant with schistosomiasis , where known schistosoma eggs in feces were found in a donor . after treatment with praziquantel , ldlt with a graft was performed with no donor or recipient complications after 2 years post - operatively . chronic changes caused by schistosomiasis , leading to granuloma formation to a parasite s egg , might be only a sign of a distant infection . donor organs from endemic areas who have negative fecal examinations and normal liver function tests may be considered for partial liver donation . while organ shortages remains a major limitation to liver transplantation , expansion of the donor pool criteria to include patients presented in this case report might allow expansion and allocation of donor livers .

abstracta shortage of donor organs is a major limitation to liver transplantation . expansion of donor pool criteria to include patients with schistosomiasis diagnosed on liver biopsy might allow the allocation of more transplant livers . schistosomiasis is a chronic parasitic disease affecting millions in endemic areas including sub - sahara africa that might lead to the development of granulomas as a response to the parasite s ova and might cause chronic liver disease and portal hypertension . due to increased mobility globally , schistosomiasis may be encountered in non - endemic areas . currently , the usage of donor livers with known schistosomiasis is not universally defined .

however , not every orthopaedic surgeon has had the opportunity to observe a case of simultaneous shoulder dislocation with ipsilateral humeral shaft fracture . the literature related to this rare injury is limited to case reports often with inadequate follow ups . herein we present a case of a 15-year - old boy with an anterior shoulder dislocation and ipsilateral fracture of humerus , and discuss his non - invasive treatment . the patient was a 15-year - old , boy who had been working at a bakery , and his right upper extremity had been caught in an electrical mixer used to mix wheat dough . he had sustained a combined anterior dislocation of shoulder with ipsilateral fracture of humeral shaft and greater tuberosity ( figure 1 ) . the patient underwent an urgent attempt for closed reduction of both shoulder and humeral shaft under general anesthesia . counter traction method by grasping the proximal fragment and traction against the counter traction through a rolled sheet in the axilla of the patient ( figure 2 ) . the successful closed reduction , achieved at first attempt , was followed by coaptation plaster splint for four weeks followed by sarmiento splint for an additional four weeks ( figure 3 ) . anteroposterior radiograph showing an anterior dislocation of the right glenohumeral joint with ipsilateral fracture of humerus . the neurologic exam for axillary and radial nerve was normal before and after the procedure . despite the somehow common occurrence of the shoulder dislocation and humeral shaft fracture , simultaneous occurrence of them is rare such that all the reports in the literature are restricted to case reports by various authors . in 1977 chen et al . reported two cases of this injury , and their attempt at reviewing the literature only revealed only 14 case reports . they first fixed the humeral fracture with a plate , and then closed the reduction of shoulder dislocation in both cases . . also reported one case treated by humeral shaft internal fixation with plating and closing the reduction of shoulder dislocation . review of various cases presented in the literature shows that the main problem for surgeons has been the lack of adequate lever arm to do the closed reduction of the joint . this forces surgeons to first fix the shaft by a plate or external fixator , and then to attempt closed reduction of the shoulder joint . there is , however , a report , similar to our experience , of successful closed treatment of both problems . the other problem with this rare combination is the possibility that shoulder dislocation is missed , especially if it is posterior , and the x ray is of poor quality and does not clearly show the shoulder joint position . the present case indicates that closed reduction of both injuries under general anesthesia was accopanied by good clinical results 15 years later .

simultaneous dislocation of shoulder and humeral shaft fracture is a rare injury , and there is no clear protocol for its treatment . herein we present a case of a 15-year - old boy , who suffered from a job - related accident and sustained fracture of humeral shaft associated with ipsilateral anterior shoulder dislocation and fracture of greater tuberosity 15 years ago . he received closed reduction of both injuries and coaptation plaster splint for four weeks , followed by sarmiento splint at that time . fifteen years after the injury , he has no problem related to the previous injury , and does not experience any episode of shoulder instability .

a 9-year - old boy presented to the outpatient department with bilateral webbing from the lateral chest wall extending up to his elbows , which looked like wings [ figure 1 ] . this deformity had been present since birth and involved anterior axillary fold as well as medial elbow [ figure 2 ] . this was successfully treated on the left side with multiple z - plasty operation ( parents consented for only one side initially).during the procedure , the fibrous bands were excised down to deep fascia and z - plasty flaps were transposed to lengthen the axillary and elbow contractures [ figures 3 and 4 ] . bilateral upper limb webbing ( pterygia ) webbing involving only anterior axillary fold early post op . it can be a part of multiple pterygium syndrome , also referred to as escobar syndrome . it is a rare autosomal recessive disorder , which is characterized by a web across every flexion crease in the extremities , most notably the popliteal space . in addition , this syndrome is associated with other structural anomalies : a vertical talus , vertebral segmentation anomalies and congenital lordoscoliosis . other associated anomalies are multiple joint webs , unusual finger contractures , syndactyly , rocker bottom feet , ptosis , antimongoloid slant of palpebral fissures , epicanthal folds , cleft lip and or palate , highly arched palate , scoliosis and short stature . autosomal recessive inheritance of multiple pterygium syndrome was suggested by findings in affected siblings with normal parents . it is differentiated from other types of inheritance like autosomal dominant type of multiple pterygium syndrome , autosomal dominant type of popliteal pterygium syndrome and x - linked dominant inheritance variety . pterygia seen in this variety of multiple pterygium syndrome of neck , antecubital and axillary areas are not present in the popliteal pterygium syndrome . cleft lip ( with or without cleft palate ) has been seen in nearly all cases of popliteal pterygium syndrome , whereas cleft palate is present in only 41% of multiple pterygium syndrome . intellect is usually normal in these cases , although there is a danger of mental retardation being assumed due to the multiple congenital defects , misdiagnosis , and delayed motor milestones . manual dexterity is often better than predicted from the appearance of the hands as seen in our case . correction of restricting webbing by simple z - plasty adds to the mobility of the affected limb . a joint approach with a team of paediatrician , genetics consultant and orthopaedic and plastic surgeon

reporting images in a case of a 9-year - old boy who presented with bilateral congenital webbing ( pterygium ) of axillae and elbows . this deformity was restricting his axilla and elbow movements . this was successfully treated on one side with multiple z - plasty . an outline of multiple pterygium syndrome is given herewith .

it is composed of several mullerian type lesions like endometriosis , endocervicosis and endosalpingiosis occurring together . a 32 year old lady presented with complaints of dysuria and burning micturition during menstrual cycles . abdominal examination and per speculum examination was normal . cystoscopy showed a 4 cm raised papilliferous area in the right lateral wall of bladder with intact overlying mucosa . histopathological examination of the biopsy specimen revealed endometrial glands and stroma along with endocervical type of glands and glands lined by ciliated cells in the bladder wall [ figures 1 and 2 ] . these findings indicate that the bladder lesion was mullerian in nature with endometriosis , endocervicosis and endosalpingiosis . the patient was found to be asymptomatic on follow up of 2 years but on cystoscopy a mass lesion was persistent which was smaller in size ( 1.5 cm ) in comparison to the earlier lesion . ( a ) endometrial type of glands and stroma extending deep within the bladder wall ( b ) glands lined by tubal type epithelium ( hematoxylin & eosin ( h&e ) ; 200 ) endocervical glands in the bladder wall lined by mucinous tall columnar epithelium with basal nuclei along with an endometrial gland ( arrow ) h & e ; 200 . urinary bladder is the most common site of genitourinary endometriosis . in addition to classical endometriosis , glandular lesions made up of endocervical type glands ( endocervicosis ) can also occur in the bladder . this lesion is usually seen in women of child bearing age and presents as a mass in the posterior wall or dome of the bladder with suprapubic pain . other mullerian tissues such as tubal epithelium have been reported in the bladder under the name of endosalpingiosis . the term mullerianosis is used when there is a complex combination of these mullerian - type lesions . to our knowledge , only five cases of mullerianosis of urinary bladder have been reported . endometriosis of the urinary bladder classically affects women in the second to fifth decade of life but may be seen in postmenopausal women receiving exogenous oestrogens . endometriosis may also be rarely seen in men with prostatic carcinoma who have received exogenous oestrogen therapy . endometriosis usually presents as a mass lesion on cystoscopy , most commonly in the trigone or posterior wall . endocervicosis is characterised by extensive involvement of bladder wall by benign / mildly atypical columnar mucin secreting endocervical glands . mullerianosis is associated with presence of tubal - type epithelium in addition to endometriosis and endocervicosis . the pathogenesis of these lesions remain debatable and both implantative and metaplastic origins have been suggested . implantative origin is supported by a frequent past history of pelvic surgery in endometriosis and endocervicosis . in the present case the differential diagnosis of mullerianosis would include a variety of lesions both neoplastic and non - neoplastic . possibility of a minimal deviation adenocarcinoma of cervix infiltrating the bladder has to be ruled out . in the present case , cervix was normal on examination and the clinical course was indolent . minimal deviation adenocarcinoma will show at least focal atypia , which was lacking in the present case . benign lesions like cystitis cystica and cystitis glandularis can mimic mullerianosis but both these entities do not show deeply situated glands as in mullerianosis . urachal remnants in the bladder can also show tubular structures lined by mucinous epithelium however they are usually incidental findings observed near the dome of bladder and is usually surrounded by a loose peritubular fibromuscular tissue . mullerianosis of the bladder is a rare lesion , which may present as a mass lesion mimicking a tumor .

mullerianosis of the urinary bladder is a rare and morphologically complex tumor - like lesion , composed of several types of mullerian lesions like endometriosis , endocervicosis , and endosalpingiosis . this disease occurs in women of reproductive age group . implantative and metaplastic origins have been suggested in the pathogenesis .

currently for super - obese individuals , bariatric surgery is the only evidence - based approach that results in clinically significant and sustainable weight loss . with the increasing prevalence of obesity , there is an increased demand for bariatric surgery . subsequently , a large number of patients shall be presenting with the complications of bariatric surgery . a 42-year - old caucasian female underwent laparoscopic roux - en - y gastric bypass in february 2007 . she subsequently developed gallstones , a known complication of bariatric surgery due to rapid weight loss . she presented to our unit in february 2012 with a week long history of right upper quadrant pain associated with vomiting . blood tests revealed deranged liver function tests : alkaline phosphatase of 169 u / l and alanine aminotransaminase of 188 u / l . a computed tomographic scan was organized confirming the appearances of a dilated common bile duct and the potential of a calcified stone in the common bile duct . she was managed conservatively with analgesia and ursodeoxycholic acid to attempt to dissolve the stones . her liver function tests and pain improved , and she was discharged with an outpatient magnetic resonance cholangiopancreatography ( mrcp ) . in march 2012 , her mrcp showed a dilated common bile duct of 10 mm and two small filling defects suggestive of gallstones . again , this confirmed the dilated common bile duct , but also showed free drainage of bile into the duodenum and no gallstones ; hence , therapeutic interventions were not undertaken . she was referred to the gastroenterologists for further advice , but as of june 2012 she was still symptomatic . ultimately , an endoscopic retrograde cholangiopancreatography ( ercp ) and sphincterotomy was deemed to be necessary to alleviate her symptoms . owing to the altered anatomy post roux - en - y gastric bypass , this was to be a technical challenge . the bypass of the distal stomach and proximal small bowel is achieved by anastomosis of the gastric pouch and the jejunum known as the roux alimentary limb . the biliary pancreatic limb is then anastomosed to a small bowel . as a result it is technically challenging to perform an ercp in the conventional manner , two subsequent port sites were used : a 5-mm port site in the right upper quadrant and a 10-mm port in the left hypochondrium ( fig . 1 ) . stay sutures were placed in a diamond - shaped configuration around the gastrotomy site these can allow both traction and counter - traction . a sterile ercp scope inserted manually through a left 10-mm port site all under laparoscopic visualization . there were no obvious gallstones in our patient ( fig . 2 ) . the gastrotomy site was closed with a two layer closure : the first layer as a running stitch and then as a horizontal mattress with 2.0 vicryl . she was admitted for an overnight stay and discharged the following day . upon review of the patient in a routine 6-week follow - up clinic , she was asymptomatic and was discharged back to the care of her general practitioner . with the increased demand for bariatric surgery worldwide , there will be an increased role of endoscopy to diagnose and manage complications . with altered anatomy , the transgastric approach can be used as an access point into the gastrointestinal tract . we have demonstrated that the technique when performed correctly can be a safe and viable option in most district general hospitals , which would certainly have a laparoscopic surgeon and a gastroenterologist on site .

the prevalence of bariatric surgery is increasing worldwide and as a direct consequence , there shall be an increasing number of patients presenting with the complications of bariatric surgery , often to non - specialist units . the authors report a case of a 42-year - old caucasian female who had previous laparoscopic roux - en - y gastric bypass , open cholecystectomy and abdominoplasty presenting with right upper quadrant pain in keeping with retained common bile duct stones . after the failure of conservative management , a laparoscopic - assisted transgastric endoscopic retrograde changiopancreatography and sphincterotomy was performed . we shall be describing our technique .

disability evaluation typically requires an assessment of the activities of daily life ( adl ) and instrumental adl ( i - adl ) . in japan , a physical disability certificate is based on the patient 's adl and i - adl grades . the independent adl scale , which is widely considered within the field of rehabilitation medicine to be a particularly important component in the evaluation ( 1 , 2 ) , considers both individual and universal factors ( fig . assessment is performed by analyzing the results of test activities relating the patient 's personal life . for example , adult independence refers to a person who does not require external care in daily life , whereas child independence includes care within the family and school life . such personal factors have been nurtured through development in the community and belong to the community 's values and customs . the universal factors , on the other hand , are measured against the common rule of humanbeings . furthermore , disability evaluation should also include assessment for the supporting system in the community - based rehabilitation ( cbr ) and other problems . these other problems , which relate to objective signs , the timing of evaluation and multiple impairments , are still under fierce debate . , japan officially recognizes three forms of the identification booklet for the disabled , one for physical disability , one for mental disability , and another for psychological disability . during the course of this investigation , several reports issued by the ministry of health , labor and welfare of japan , including the outline of the systems and basic statistics in annual reports on health and welfare 1998 - 1999 social security and national life from white paper and reports were reviewed ( 3 ) . data concerning the annual change in the number of people with physical disabilities by type and age per 1,000 people were used to analyze total disability evaluation , while the annual change in the number of children with physical disabilities by type and age was used to analyze child disability evaluation . the number of people with physical and mental disabilities was used to determine the relationship between welfare facilities and the home in the community . finally , several data related to the elderly were used to anticipate bigger problems in future . the results of this study , as provided in detail in the appendices to this report , are summarized as follows . recent trends in the last 10 yr suggest a sharp rise in physical disabilities and internal disorders among the elderly ( table 1 , 2 ) . data on children by type ( table 3 ) and age ( table 4 ) shows an increase in the rate of internal disorders and young children with physical disabilities . data on the overall number of people with disabilities ( table 5 ) reveals that 28.5% of patients in health care facilities have mental retardation , whereas only 5% have physical disabilities . data suggesting bigger future problems for the elderly are also presented in fig . 2 and table 6 and 7 . in japan , disability is categorized under three identification booklets : 1 ) physical disability , which is classified by four subgroups , 2 ) mental disability , and 3 ) psychological disability . the classification of the physically disabled subgroups ( table 1 - 4 ) was very useful for determining the problems for government policy on welfare and public health . the increasing rate of internal disorders within the physically disabled has become a major theme for following global welfare services in the future . a multi - system for evaluating disabilities has resulted in many welfare services , depending on the various kinds of disabilities . however , the achievements of the welfare system , based on different types of disabilities , have led to a complex or confused service system . the system has also been affected yearly by the changing social community around persons with disabilities . accordingly , the increasing ratio of the elderly population has prompted the development of a new disability evaluation related to practical adl . to cite another example , mental disability has been evaluated from infancy because of a lack of social response . as such , most people with mental disability usually tend to receive the identification booklet up to the age of 18 yr . however , table 5 shows that , in japan , adults with mental disability tend to be less adaptive to the community than those with physical disability . therefore , the ministry of health , labor and welfare of japanese government has started to rearrange many welfare services to be more suitable to a small community around persons with disabilities . moreover , the concept of preventing disabilities is emphasized not only in rehabilitation medicine , but also in cbr , indicating that disability evaluation needs another factor of support system based on their community . thus , it is important to think about the disability evaluation according to the characteristics of age , impairment and assistive environment . in the field of cbr , disability evaluation has recently become important for measuring the effects of rehabilitation treatment in medical insurance and decision of the grade of welfare service volume in japan . while the first and second groups have been historically well - established without any disagreement on the classifications of disability type , the third group concerning psychological disability has recently been questioned with respect to the visibly less apparent disabilities relating to cognition , memory , attention , emotion , and social behavior after traumatic head injury . although the new system to evaluate and support such a disability group is under development throughout the country , problems with classification continue to be debated .

to examine the current state and social ramifications of disability evaluation in japan , public data from annual reports on health and welfare 1998 - 1999 were investigated . all data were analyzed based on the classification of disabilities and the effects of age - appropriate welfare services , which have been developed through a half - century of legislative efforts to support disability evaluation . these data suggest that disability evaluation , while essentially affected by age and impairment factors at a minimum , was impacted more by the assistive environment for disabilities . the assistive environment was found to be closely linked with the welfare support system related to a global assessment in the field of community - based rehabilitation .

pseudomonas aeruginosa is a gram - negative , aerobic bacterium found in water and soil . it is a normal flora of the skin and gastrointestinal tract of human beings ( 1 , 2 ) . this bacterium as an opportunistic pathogen is one of the most important microorganisms in nosocomial infections in immune - compromised patients including patients with malignancies , cystic fibrosis and burns with multiple pathogenic factors and high rate of resistance to most of the antibiotics . the presence of glycocalyx in cellular membrane is responsible for easy binding of the bacterium to the host cell , biofilm formation and protection of bacteria against penetration of antimicrobial agents and phagocytic system ( 3 ) . in a previous study , p. aeruginosa has been isolated from nosocomial pneumonia ( 16% ) , nosocomial urinary tract infections ( 12% ) , post - operative wound infections ( 85% ) and hospital blood infections ( 10% ) which included 23% of total isolated bacteria from admitted patients ( 4 ) . selection of the appropriate treatment approach against p. aeruginosa is limited due to its resistance to common recommended antibiotics ( 5 , 6 ) . bacterial resistance to antibiotics is increasing which makes humans to apply effective antimicrobial agents with fewer side effects such as medicinal plants instead of medicines with less efficacy and greater side effects ( 7 ) . plants have different chemical compounds like secondary metabolites ( 8 , 9 ) with many biochemical and bioactivity properties showing applications in various industries such as pharmaceutical , chemical , cosmetic and food industry ( 1013 ) . the aim of this study was to introduce the most important native medicinal plants of iran being effective on p. aeruginosa . all required information was obtained by literature review using keywords including p. aeruginosa , medicinal plant extracts or essential oils of published articles in authentic scientific databases such as science direct , wiley - blackwell , springer , google scholar , scientific information database ( sid ) and magiran . according to literature review , our results showed 12 different native medicinal plants were effective against p. aeruginosa in iran including eucalyptus camadulensis , marticaria chamomilla , ferula gummosa boiss , lawsonia inermis , ocimumgra tissimum , allium sativum , satureja hortensis l , satureja bachtiarica bunge , satureja khuzestanica ( jamzad ) , thymus daenensis celak , thymus carmanicus jalals and camellia sinensis . medicinal plants are good alternatives for synthetic preservatives in food and drug industry due to their antimicrobial compounds . e. camadulensis ( eucalyptus ) leaves are rich in polyphenols and terpenoids and eucalyptol or cineole ( 26 ) . phytochemicals results showed that alfa and beta - pinenes are the main ingredients of ferula gummosa boiss ( galbanum ) ( 27 ) and lawsoniainermis ( henna ) containsmanitol , tanic acid , mucilage and galic acid but its most important ingredient is 2-hydroxy-1,4-naphoquinone ( lawson ) recognized as bioactive agent . ocimumgra tissimum ( basil ) contains essential oils , tannins , glycosides , saponin , anthocyanin , eugenol , linalool , methyl cinnamate , camphor and thymol . different species of satureja ( savory ) contain monoterpenes , phenolic compounds especially carvacrol , thymol and eugenol as well as some of the sesquiterpenes . the main phytochemical essential oils of aromatic plants are hydrocarbons , aldehydes , ketones , alcohols , phenols , ethers and esters with phenolic and terpenic sources . the presence of linalool , alpha - pinene , beta - pinene , borneol , carvone , limonene , carvacrol , p - cymene and terpinenein essential oils of understudy plants showed antimicrobial properties particularly ( 28 ) . these compounds with high hydrophobic properties can separate lipids from bacterial cell wall and thereby increase the permeability of the membrane which leads to ion expelling and electron imbalance and eventually cell death . the researchers demonstrated that plant species used in popular medicine are promising resources for antimicrobial treatments ( 13 , 14 , 29 , 30 ) . phytochemical analysis has shown that bioactive compounds of medicinal plants with their antioxidant and antimicrobial properties are good substitutions for synthetic drugs in food and drug industry .

background and objectives : pseudomonas aeruginosa is a gram - negative , aerobic bacterium found in water and soil . it is a normal flora in skin and gastrointestinal tract of human beings . p. aeruginosa as an opportunistic pathogen involved in nosocomial infections having multiple pathogenic factors and shows high rate of resistance to different antibiotics . the aim of this study was to identify the most important native medicinal plants of iran effective on p. aeruginosa.materials and methods : all required information was obtained by searching keywords such as p. aeruginosa , medicinal plant extracts or essential oils in published articles in authentic scientific databases such as science direct , wiley - blackwell , springer , google scholar , scientific information database ( sid ) and magiran.results:according to the literature review , our results showed 12 different native medicinal plants were effective against p. aeruginosa in iran including eucalyptus camadulensis , marticaria chamomilla , ferula gummosa boiss , lawsonia inermis , ocimumgra tissimum , allium sativum , satureja hortensis l , satureja bachtiarica bunge , satureja khuzestanica ( jamzad ) , thymus daenensis celak , thymus carmanicus jalals and camellia sinensis.conclusion:phytochemical analysis has shown that bioactive compounds of medicinal plants with their antioxidant and antimicrobial properties can be good alternatives for the synthetic medicines in food and drug industry .

a 75-year - old female presented to the chest clinic of our hospital with a history of a cough with mucoid expectoration , episodic dyspnea with wheeze of 1 month duration . she did not have any symptoms of chest pain , hemoptysis , recurrent fever , weight loss , reflux , or symptoms of postnasal drip . she did not give any past history of atopy or asthma , and her family history was also negative for same . general examination revealed that she had pallor . her chest x - ray is shown in figure 1 . what is the abnormality seen ? chest x - ray posteroanterior view showing elevated hemidiaphragm on the right , with a retrocardiac opacity on the left side diaphragmatic eventration is congenital in nature and is due to incomplete muscularization of the diaphragm with a thin membranous sheet replacing normal diaphragmatic muscle . over a period of time , this region stretches and thins out , and on inspiration does not contract normally . radiologically , elevation of the affected portion of the diaphragm is usually seen as a smooth hump , while the remainder of the hemidiaphragm contour is normal . hiatus hernias occur when there is a herniation of abdominal contents through the esophageal hiatus of the diaphragm into the thoracic cavity . usually , both these defects are seen in isolation . however , combination of both an eventration and hiatus hernia occurring together is a very rare entity , and a thorough literature search showed only three previous such cases . the coronal reconstructed 64 slice computerized tomogram showed eventration of right hemidiaphragm with liver occupying the right lower hemithorax . widening of the esophageal diaphragmatic hiatus with herniation of stomach and perigastric fat was also seen [ figure 2 ] . coronal reconstructed 64 slice computed tomography image showing eventration of right hemidiaphragm with liver occupying the right lower hemithorax . widening of the esophageal diaphragmatic hiatus with herniation of stomach and perigastric fat is also seen her spirometry showed a mild reversible airflow obstruction . she was managed conservatively considering her age and her respiratory symptoms resolved significantly after the institution of inhaled corticosteroids .

although diaphragmatic anomalies such as an eventration and hiatus hernia are commonly encountered in incidental chest x - ray imaging , the presence of concomitant multiple anomalies is extremely rare . this is all the more true in adults . herein , we present the case of a 75-year - old female , while undergoing a routine chest x - ray imaging , was found to have eventration of right hemidiaphragm along with a hiatus hernia as well .

a 40-year - old patient was admitted to the hospital on january 29 , 2004 , with fever , generalized weakness , diarrhea , and vomiting . initial blood tests showed hemoglobin level of 8.5 g / dl , leukocyte count of 4.9 x 10/l , and platelet count of 110 x 10/l . erythrocyte sedimentation rate was elevated at 88 mm / h . her serum glucose was 8.5 mmol / l , and urea and electrolytes were normal . she was started on intravenous ciprofloxacin , but her fever persisted , and she became increasingly confused . her old hospital records became available shortly after her death , and we noted that she had been diagnosed with systemic lupus erythematosus ( sle ) in 1994 . when she last attended the outpatient department 3 months before her hospital admission , she was prescribed 50 mg azathioprine and 5 mg prednisolone daily . she was a housewife and lived in cit la cure , a poor suburb of the capital city port - louis . , her home becomes very muddy after heavy rainfall , and her feet were often in mud while performing her household duties . after 5 days of incubation , it produced colonies that appeared dry and rugose on the plates after 48 h and was identified as b. pseudomallei by using api 20ne ( biomrieux , marcy letoile , france ) with the profile 1156577 . antimicrobial susceptibility testing by disc diffusion showed the organism to be resistant to colistin , ampicillin , cephalexin , gentamicin , and ciprofloxacin and susceptible to co - amoxiclav , tetracycline , cefotaxime , ceftriaxone , ceftazidime , piperacillin , and meropenem . a large zone of inhibition was seen around the co - trimoxazole disc , within which a thin film of growth was observed . we are not aware of any study looking for the organism in soil in this country . groodoyal , pers . comm . ) . whether human cases of melioidosis have been missed in the past is not known , and cases may be missed currently . recognizing the disease depends on awareness on the part of clinicians and on the ability of microbiology laboratories to identify the causative organism ( 1,8 ) . before 1998 , oxidase - positive , gram - negative bacilli other than p. aeruginosa were not identified to species level in laboratories in mauritius . since then , at our laboratory , which receives specimens for bacteriologic investigations from all government healthcare institutions , such organisms are routinely identified by api 20ne when isolated in pure culture from blood , but only occasionally when isolated from nonsterile sites such as sputum and pus swabs . some clinicians routinely request blood cultures from patients with high fever before starting antimicrobial drugs , although in practice , the specimen is often collected by nursing staff after the first dose has already been administered . other clinicians only request blood cultures if fever persists after a few days of empiric antimicrobial therapy . in the case reported here , prior administration of cefotaxime may have delayed b. pseudomallei culture from blood until 5 days of incubation , when the median time to obtain a positive blood culture result is typically 48 hours ( 1 ) . an association between rainfall and melioidosis has long been recognized ; most cases in thailand ( 9 ) and northern australia ( 10 ) occur during the wet season . the increased number of cases noted during the rainy season may be caused by the movement of b. pseudomallei from deeper layers toward the surface when dry topsoil is moistened by rainfall ( 2 ) . in mauritius 196 mm rainfall was recorded in port - louis , which is 37% higher than the 19712000 mean rainfall for the region during this month . january 2004 was the sixth wettest january of the past 30 years in port - louis . recent reviews have suggested a predominant role for percutaneous b. pseudomallei infection in the pathogenesis of melioidosis ( 11 ) . studies carried out in regions where melioidosis is endemic have shown that exposure to wet soil and water are associated with increased risk for disease ( 9 ) . the feet of our patient were regularly exposed to wet soil during rainy periods . in melioidosis - endemic areas , although a large percentage of the population has been exposed to b. pseudomallei , as determined by seroprevalence studies , only a few develop melioidosis ( 12 ) . most cases occur in patients with underlying illnesses , such as diabetes mellitus , renal disease , and alcoholism ( 9,10 ) or in those who are immunosuppressed ( 1 ) . this first case of melioidosis in mauritius occurred in an immunosuppressed patient who had a history of prolonged and regular exposure to mud during a year when rainfall was higher than average . this combination of 3 risk factors does not occur regularly , and it is possible that few additional cases will be recorded in mauritius in the future . however , clinicians and laboratory staff must remain aware of this disease , particularly because in a noncommunicable disease survey carried out in 1998 , almost 20% of the mauritian population > 20 years of age were found to have type 2 diabetes mellitus ( 14 ) , the most common predisposing condition for melioidosis ( 1 ) . determining the distribution of b. pseudomallei in soil in mauritius by conducting environmental investigations will also be useful .

we report the first case of human melioidosis from mauritius , where burkholderia pseudomallei has never been isolated . the patient was immunocompromised , had never traveled abroad , and had a history of regular exposure to mud . she became ill at a time when rainfall was higher than the monthly average .

failure to grasp the rationale behind cleaning and shaping concepts can increase the incidence of procedural complications such as blockage , ledge formation , apical transportation and perforations . according to the glossary of endodontic terms of the american association of endodontists , canal transportation is defined as follows : removal of canal wall structure on the outside curve in the apical half of the canal due to the tendency of files to restore themselves to their original linear shape during canal preparation ; may lead to ledge formation and possible perforation . various undesirable apical preparation outcomes such as damage to the apical foramen , elbow formation , zip formation and perforation have been described as possible results of canal transportation . perforation represents a communication between the root canal space and the external root surface , causing irritation of the periradicular tissues . similar terms describing the shape of a zipped apical part of the root canal are an hourglass shape , a teardrop or a foraminal rip . it can result in a poorly cleaned root canal , which fails to provide a resistance form to compact gutta percha , and leads to obturation which is vertically overextended but internally under - filled . apical transportation can be categorized into : type 1 : represents a minor movement of the position of the physiologic foramen , resulting in its iatrogenic relocation . type ii : represents a moderate movement of the physiologic position of the foramen , resulting in a considerable iatrogenic relocation on the external root surface . in this type , a larger communication with the periapical space exists , and attempt to create a more coronal shape may weaken or perforate the root . type iii : represents a severe movement of the physiologic position of the canal , resulting in a significant iatrogenic relocation of the physiologic foramen . canals exhibiting type i transportation can be cleaned and obturated , if sufficient residual dentin is maintained and shape created above the foramen . type ii cases are managed by placing a barrier to control bleeding and provide a backstop to pack against during subsequent obturation procedures . in type iii situations , a barrier technique is usually not feasible ; it requires obturation as best as possible followed by corrective surgery . a healthy 21-year - old young male presented with the intention of seeking relief from discomfort and swelling associated with a painful endodontically retreated maxillary central incisor . five months ago , he had undergone root canal treatment for the resolution of a lesion of endodontic origin associated with 11 and 12 . clinical examination revealed non - carious , discoloured , tender 11 and incisally fractured non - tender 12 . softness to palpation indicated loss of buccal cortical plate over root apices of 11 and 12 . iopa radiograph , obtained at the time of visit , exhibited periradicular radiolucency involving insufficiently obturated 11 and overfilled 12 . periradicular curettage and removal of extruded gutta percha , after raising a mucoperiosteal flap under local anesthesia , exposed the apical canal transportation of 11 . repair of this canal defect by packing a wet sand consistency mix of white mineral trioxide aggregate ( mta ) ( angelus , brazil ) , with distilled water , restored it . cold compaction by a ball burnisher , after removal of overextended gutta - percha , sealed the root apex of 12 . a decrease in the size of radiolucency in iopa radiograph supports the clinical finding [ figures 1a g ] and [ figures 2a and b ] . ( a ) previous occlusal radiograph , ( b ) preoperative iopa radiograph , ( c ) exposed lesion , ( d ) exposed apical transportation , ( e ) defect repaired by mta , ( f ) third month recall , ( g ) recall radiograph ( a ) preoperative radiograph , ( b ) fourth month recall radiograph it is important to understand the objectives for shaping and cleaning the root canal system . the factors associated with an increased risk of canal transportation include insufficiently designed access cavities , use of inflexible instruments , instrumentation technique , insufficient irrigation during mechanical enlargement , degree and radius of a canal curvature , unseen canal curvatures in two dimensional radiography and experience of operator . canal transportation may result in inadequately cleaned root canals , over - reduction of sound dentin and destruction of the integrity of the root . mineral trioxide aggregate is the material of choice for managing various undesirable apical preparation outcomes . the sealing ability of this non - absorbable material is not adversely affected by blood contamination . cementum grows adjacent and onto this radiopaque material , thus allowing for a normal periodontal attachment apparatus . in this case , mta was used to repair apical transportation defect , as it has above - mentioned advantages over other materials . apicoectomy or root - end preparation was avoided as it would have further removed the sound dentin . many endodontic complications , including canal transportation , can be prevented while cleaning and shaping . apical transportation may lead to post - operative flare - up , surgery and extraction . however , depending upon the extent of occurrence , it can be managed . in the present case , the outcome of this case , indicate that the result of these procedures , is predictable and successful .

procedural accidents leading to complications such as canal transportation have been ascribed to inapt cleaning and shaping concepts . canal transportation is an undesirable deviation from the natural canal path . herewith a case of apical transportation of root canal resulting in endodontic retreatment failure and its management is presented . a healthy 21-year - old young male presented discomfort and swelling associated with painful endodontically retreated maxillary incisor . radiograph revealed periradicular radiolucency involving underfilled 11 and overfilled 12 . insufficiently obturated 11 exhibited apical transportation of canal . this type iii transportation was treated by periradicular surgery and repair using white mineral trioxide aggregate ( mta ) . comfortable asymptomatic patient presented uneventful healing at third and fourth month recall visits . a decrease in the size of radiolucency in radiograph supported the clinical finding . in the present case , mta is useful in repairing the transportation defect . the result of these procedures is predictable and successful .

circumscribed palmo - plantar hypokeratosis ( cph ) is a rare dermatosis , first described by perez et al . in 2002 . only about 50 cases have been described in the literature so far , of which two were with a unique plantar localization . the disorder usually affects women ( with a female : male ratio of 4:1 ) , with a mean age of presentation of 61 years . it presents clinically with annular erythematous depressed patch rimmed by a slightly hyperkeratotic border ; the lesion is usually unique and localized over the thenar or hypothenar eminence of the palms , rarely on the soles . cph presents clinical stereotypical features even if sometimes it is misdiagnosed as palmar porokeratosis or bowen 's disease . its main histopathologic feature shows a characteristic epidermal depression with an abrupt decrement in the thickness of the stratum corneum , with a sharp stair a 68-year - old woman presented with an acquired , asymptomatic , slowly enlarging erythematous patch on the thenar eminence of her right hand . her medical history included a mammary adeno - carcinoma treated with surgery and radio - therapy 10 years before . examination revealed over the right eminence thenar an approximately 15 10 mm , well - circumscribed circular erythematous area , depressed , with sharp step between involved and uninvolved skin [ figure 1 ] . histopathology showed a sharply demarcated depressed area with markedly thinned stratum corneum with normal basal layers . slight dilatation of the dermal superficial vessels was noted , with poor lymphocytic infiltration and edema . there was no evidence of cornoid lamella excluding a diagnosis of porokeratosis . corneocytes of the upper layers showed signs of vacuolization and they became eosinophilic at the edge [ figure 2 ] . based on these findings the patient is currently on clinical follow - up and in 7 years there has been no evidence of enlargement or evolution . a circular and well - circumscribed area of depressed and erythematous skin on the right thenar eminence skin biopsy specimen from border of the lesion : sharp step - off from the thick stratum corneum of normal acral skin to the thin stratum corneum of the involved skin ( hematoxylin eosin staining ; original magnification 200 ) cph is a benign dermatosis of unknown origin predominantly seen in adult women ( female : male ratio 4:1 ; mean age 61 years ) . argued that it is a localized acquired epidermal malformation because of the presence of the lesions for many years and the absence of a trauma at the site of the lesions . using transmission electron microscopy , showed a reduction in keratin bundles and keratohyaline granules , and increased lipid in the horny layer , suggesting a primary disorder of keratinization . in contrast to these studies , resnik et al . suggested that circumscribed palmar or plantar hypokeratosis represents a localized defect in the maturation of keratin , which could follow subclinical episodes of trauma . in interestingly , in this case the lesion had been present for an unusual short time of 6 months . cph is chronic dermatosis with a benign evolution ; the lesions slowly progress or remain stable over years as in our case . only in one recently reported case the lesions typically involve the thenar and hypothenar eminences of the palm or the medial side of the sole . clinically they appear as circular and well - circumscribed areas of depressed and erythematous skin with slightly elevated scaly borders . in most cases , histopathologic features are characteristic and they consist of a localized depression of the epidermis , with a sharp stair between normal and involved skin . the stratum corneum is thinner and orthokeratotic , with a stratum granulosum slightly decreased compared to the adjacent non - involved skin . , a biopsy should be performed at the border 's lesion to compare the horny layer of the involved skin with that of the non - involved adjacent skin . in literature , various topical treatments are proposed , including corticosteroids , salicylic acid and retinoids , but they all have been disappointing . urbina et al . reported resolution in one case after 4 years of topical therapy with calcipotriol . boffa and de gaetano reported a case of resolution after treatment with liquid nitrogen cryotherapy . no improvement was evident after treatment with a topical steroid for 3 months . in summary as has been described in literature , we did not find evidence of trauma as an inciting factor in our case . unlike another reported case , we did not identify evidence of hpv within lesions by immunohistochemistry . we consider this disorder as an acquired non - inflammatory defect in keratinization on acral sites with a remote potential of malignant transformation ; for this reason , we consider a prolonged clinical follow - up essential , as in our case .

circumscribed palmar or plantar hypokeratosis is a rare benign epidermal malformation of the skin . clinically it shows asymptomatic , well - circumscribed , and depressed erythema persisting for many years on the palms or soles . its main histopathologic feature shows a characteristic epidermal depression with an abrupt decrement in the thickness of the stratum corneum , with a sharp stair between normal and involved skin . we describe a case of a 68-year - old woman who presented with an erythematous , asymptomatic , well - circumscribed , depressed patch , on the right thenar eminence which had been present for years .

the technique described provides extracorporeal control of one limb of the suture and has the advantage of continuous tension application on both ends of the thread during knot formation . it is easy to learn , fast to perform , and no new equipment is used . the needle end is advanced through the trocar , and the free end is firmly grasped extracorporeally by the assistant . the needle is passed through the tissues to be sutured and held with the right needle holder . the left - hand instrument holds the filament about 5 cm proximal to the needle , thus resulting in a triangle formation , the apex of it being presented by the tissues to be sutured and the base by the segment of the filament between the left instrument and the needle holder ( figure 1 ) . the needle is rotated around the free end of the filament , which is aligned and held in place by external traction ( figures 2 and 3 ) . as many turns as desired are thrown choosing the appropriate direction : for clockwise throws , the needle is grasped proximally and for opposite throws distally . the surgeon prepares to rotate the needle around the limb of the suture for a first time . accurate and safe tissue suturing is essential for advanced laparoscopic surgery . during the last decade , suturing devices such as clip applicators , staplers , and endo - loops have been developed . extracorporeal knots are easy to perform and are usually created by multiple throws that are advanced intracorporeally with a knot pusher . most important , tension can not be maintained while throwing turns , and the tying instruments before the next throw release the ends of the filament . on the other hand , sequential throw formation in opposite directions is technically demanding , resulting in an unsecured sliding sequence instead of a square knot . another problem is tissue exposure to unnecessary manipulation and traction from pulling long lengths of suture through the needle track and pushing the knot into position . intracorporeal knots seem to deal better with the last issue , and they have the advantage of focusing on the operative field during creation . however , a considerable degree of virtuosity is required , and these knots are not available as an option to occasional laparoscopists . on the other hand , the main problem of preserving ligature tension while forming the knot remains to be settled . even though the technique is an intracorporeal one , continuous tension is guaranteed by providing extracorporeal control of one limb of the suture , thus allowing the manipulation of the needle end with both instruments . winding sequential throws in both directions the procedure necessitates the presence of a curved needle and can be used for ligating tissues that must be securely approximated , as in gastric banding and nissen fundoplication . the greek term for intracorporeal knot is easy to learn and is secure and therefore presents an option not only to the laparoscopic surgeons but to other surgeons as well .

accurate placing of securely tied knots in laparoscopic surgery is technically demanding and time consuming . surgeons must face difficulties arising from 2-dimensional vision , spatial limitations , and restricted movement . issues to be taken into account include security , virtuosity , and cost effectiveness . the authors believe that in spite of advances in instrumentation and optics , training should aim at manual skill development and application of the basic principles of general surgery .

congenital pericardial defects ( cpds ) are uncommon anomalies that are usually discovered unexpectedly by a cardiologist , radiologist , or surgeon . occasionally , these defects may cause symptoms such as chest pain , dyspnea , arrhythmias , syncope , and even sudden death . arrhythmias , including atrial fibrillation ( af ) , are uncommonly found in cases of cpd . herein , we report the case of a patient with persistent af associated with a cpd and herniation of an enlarged left atrial ( la ) appendage . the patient was treated successfully with a hybrid procedure , in which total thoracoscopic ablation ( tta ) was followed by an electrophysiological ( ep ) study . his medical history was otherwise unremarkable , except for a history of hypothyroidism diagnosed eight years previously . a hybrid procedure involving tta followed by an ep study preoperative computed tomography ( ct ) and echocardiography showed no evidence of thrombi or vegetation with la enlargement . the size and volume index of the la were 55 mm and 69.4 ml / m , respectively . tta was performed under general anesthesia with double - lumen intubation . on the right side , a larger than normal atrium a large pericardial defect was found , through which an enlarged la appendage was herniated ( fig . 1b ) . although handling the lighted dissector ( atricure lumitip dissector ; atricure inc . , cincinnati , oh , usa ) was difficult due to the large mobile la appendage , pulmonary vein isolation and additional superior and inferior line ablation were successfully completed , and the la appendage was resected with an endoscopic stapler . an episode of af occurred on the fourth postoperative day , and the patient was converted to sinus conversion through cardioversion . a follow - up ep study on the eleventh postoperative day revealed successful pulmonary vein isolation , and cavotricuspid isthmus ablation was performed . left - side defects are most common , accounting for approximately 70% of cases , followed by right - side defects ( 17% ) and complete defects ( 9% ) . although patients are usually asymptomatic and most cpds are discovered incidentally , patients with partial defects may present with acute symptoms such as chest pain , dyspnea , arrhythmia , syncope , and even sudden death due to strangulation of the herniated cardiac chamber . echocardiography and ct are the most common methods used to evaluate cardiac and thoracic abnormalities ; however , pericardial defects may go unrecognized for years , as in this case . magnetic resonance imaging ( mri ) has been reported to be superior to echocardiography , but cardiac mri is still only applicable to a limited set of lesions . furthermore , enlargement of the la and la appendages due to herniation , as in our patient , may contribute to the development of persistent af , and may even be its cause . several methods have been proposed for the treatment of cpd , including la appendectomy , extension of the pericardium to reduce incarceration , primary closure , and patch closure using synthetic materials . however , no consensus currently exists regarding therapeutic options for cpd due to the small number of cases . although a previous case of paroxysmal af was resolved via resection of the la appendage instead of antiarrhythmic interventions , in cases of persistent af with an enlarged chamber , as in our patient , antiarrhythmic management techniques such as tta are probably necessary . however , it involves complex techniques and invasive procedures , requiring median sternotomy and cardiopulmonary bypass [ 1012 ] . accordingly , some researchers have recently suggested that minimally invasive thoracoscopic ablation be used , and hybrid tta has shown excellent results with low morbidity rates [ 1315 ] . in our patient , the hybrid tta procedure was initially planned for his persistent af , and the cpd was an incidental finding . although the visual field was disturbed by the enlarged la appendage , the entire ablation procedure was successfully performed and the la appendage was resected safely . in conclusion , cpd is a possible incidental finding when performing cardiac operations , including tta . enlarged la and la appendages due to herniation through the defect may cause persistent af , and antiarrhythmic treatment may be necessary for such patients . tta can be successfully performed in patients with persistent af associated with a cpd and herniation of the enlarged la and/or an la appendage .

congenital pericardial defects ( cpds ) are infrequent anomalies that are usually asymptomatic and are discovered incidentally during unrelated interventions . here we report the case of a cpd with herniation of an enlarged left atrial appendage identified during total thoracoscopic ablation ( tta ) for persistent atrial fibrillation ( af ) . the persistent af was successfully treated with a hybrid procedure , in which tta was followed by an electrophysiological study .

lung cancer is the leading cause of cancer - related mortality in both men and women throughout the world . it is also the most commonly diagnosed cancer worldwide with most of them at an advanced stage with a poor prognosis . a 78-year - old male smoker patient diagnosed to have non - small cell lung cancer ( nsclc ) , stage iv ( adrenal and liver metastasis ) was subjected to whole body bone scan to detect skeletal metastases , if any . the patient had received local palliative radiotherapy 20 gy/5 # for 1 week and was on chemotherapy with gefitinib since 12 days . increase in tracer uptake was noticed in the lower cervical vertebrae , right first rib , and medial cortical margin of the lower third of shaft of left femur [ figure 1a and b ] . hybrid single photon emission computed tomography / computed tomography ( ct ) localized the cervical uptake to left articular process of c6 and left transverse process of c7 vertebrae with dense sclerotic changes likely degeneration due to altered weight distribution . tc99m - methylene diphosphonate whole body bone scan in ( a ) anterior projection ( b ) posterior projection showing abnormal tracer uptake in the lower third of shaft of left femur ( arrow ) . hybrid single photon emission computed tomography / computed tomography ( ct ) localized the ct images ( c and d ) of the lower third of the left femur showed lysis of the anteromedial cortical margin ( arrow ) ct scan of the lower third of the left femur showed lysis of the anteromedial cortical margin with no soft tissue component [ figure 1c and d ] . on the basis of bone scan and pattern of cortical bone destruction , the diagnosis of a skeletal metastasis from a bronchogenic carcinoma was suggested . incidence of bone metastases in cancers of the breast , prostate , lung and kidney , is very high , approximately 70% of all patients . the reported incidence of bone metastasis from nsclc is around 15%-40% , most of which involves spine , rib , and pelvis and 6% in femur . bronchogenic carcinoma is an established entity in causing cortical bone metastases literature review also shows primaries in lung , breast , kidney , and pancreas as causes of cortical metastasis . of particular interest is the involvement of femur with studies reporting osteolytic cortical lesions preferentially located at the femur as typical of bronchogenic carcinoma . the reason for this special predilection for implantation in the cortical bone , particularly the femur , is still unknown . it is speculated that the vascular network , originating in the overlying periosteum of the long bones , serves as a pathway for metastatic deposits to destruct the cortical bone . literature review not only shows predominance of femoral involvement in bronchogenic carcinoma , but also patients with only pure cortical metastases had exclusive femoral involvement . four distinctive patterns of bone destruction in bronchogenic carcinoma have been described . a small focal intra - cortical lesions ( cookie - bite or cookie - cutter pattern ) ; large osteolytic lesions ; saucerized intra - cortical lesions with well defined periosteal reaction ; and lesions with predominant cortical destruction extending into the soft tissue as well as the medullary cavity . the term cookie bite metastasis was initially coined for a small intra - cortical lesion . although these lesions are not entirely pathognomonic , literature review shows that such lesion within the skeleton as initial diagnosis should alert the radiologist to the possibility of a primary neoplasm of the lung . our case further illustrates how a high degree of suspicion is needed in interpreting isolated uptake in bones scan in the femur in patients with bronchogenic carcinoma . moreover , as described in literature , the characteristic pattern of cortical destruction if found in a patient with carcinoma of unknown primary , then the search for an occult primary neoplasm should be started without delay .

detection of skeletal metastasis in patients with lung cancer is important from management point of view . we report the bone scan finding in a patient with non - small cell lung carcinoma showing isolated abnormal tracer in femur and having a characteristic appearance in computed tomography , highlighting the importance of bone scan in patients with bronchogenic carcinoma .

solid pseudopapillary tumour ( spt ) of the pancreas is one of the rare found primary tumors of the pancreas , making up approximately 0.172.7% of cases . there have been controversies regarding the terminology and according to its macroscopic and microscopic pathological characteristics over the years . it has been given multiple descriptive names such as papillary - cystic tumour , solid cystic tumor , papillary epithelial neoplasm , solid , and papillary neoplasm , papillary tumour of the pancreas , or frantz 's tumor until in 1996 the who pancreatic tumour working group recommended the use of the term solid pseudopapillary neoplasm . one of its characteristics is that this tumour seems to preferentially affect mainly young women usually in the second or third decade of life . extrapancreatic spt cases in the retroperitoneum , liver , omentum , and mesocolon have been published . some of them were considered to arise from an ectopic pancreas [ 46 ] . to the best of our knowledge , an 18-year - old woman complaining of abdominal pain , nausea , and vomiting for one month was admitted to our hospital for further evaluation . she had no history of abdominal trauma or surgery , drug usage or smoking . tumour markers ( alpha - fetoprotein , carcinoembryonic antigen , ca-125 , ca15 - 3 , ca19 - 9 , and ca72 - 4 ) of serum and other biochemical analyses were within normal limits . abdominal ultrasonography ( usg ) showed an encapsulated solid mass of 5 5.5 cm in diameter located on the subhepatic region displacing the second part of the duodenum laterally , and the computer tomography confirmed a well - encapsulated mass of 48 51 mm diameter located in the mesenteric region neighboring the superior mesenteric artery and vein , with no distinct separation from the head of the pancreas . the patient then underwent surgery , and exploration revealed an encapsulated mass of 5 5 cm in diameter that was adherent to the mesentery of the transverse colon . the tumor was not infiltrating the pancreatic tissue . no vascular invasion or lymph node metastasis pathologic examination of biopsy material revealed small cells with uniform spherical nuclei with narrow eosinophilic cytoplasm and tumoral cells forming glandular structures ( figure 1 ) . neuron - specific enolase ( nse ) , chromogranin , cd10 , cd99 , cd68 , lca , calcitonin , cea , ema , hmw ck , lmw ck , ck 7 , ki 67 , and synaptophysin antibody staining were negative ; however , the tumoral cells were stained remarkably with progesterone receptor antibody and vimentin ( figures 2(a ) and 2(b ) ) . in light of these findings , spt is a rare disease with a reported incidence of 0.13% to 2.7% of all pancreatic tumors , and cases often have been misunderstood . the spt has usually occurred in young women during the second to fourth decades of life . abdominal discomfort is the prevailing symptom associated in some cases with a palpable mass , anorexia , and weight loss . the most common extrapancreatic sites are mesocolon , liver , retroperitoneum , or greater omentum [ 47 ] . only four spt , developed from mesocolon , have been described previously ( table 1 ) . the presence of ectopic pancreatic tissue is seen in all of these published cases [ 5 , 6 , 8 , 9 ] . ishikawa et al . described the first spt case arising from an ectopic pancreas in the mesocolon . patient was 13-year - old girl , and 8-cm , well - encapsulated , and partly calcified tumor which protruded from the mesocolon was resected surgically . at the base of this tumor , small pancreatic tissues ( islet , acinar , and ductular cells ) were detected in the mesocolic tissue . these four tumours ( table 1 ) tended to grow to a large size ( 60210 mm ) , usually occurred in young female patients ( only one was in a man ) , and produced similar clinical signs . in all four published cases the tumour was found to be separate from the main pancreas at surgery . a case of spt arising in the omentum without pancreatic tissue mentioned that the occurrence of a few spts in the retroperitoneal space outside the pancreas can be related to the localization of the genital ridge during embryogenesis and speculated that spt might originate from genital ridge - related cells that were incorporated into the pancreas during organogenesis . this theory might suggest one explanation for the occurrence of spt in the mesocolon in the presented case . spt can be readily diagnosed by routine histologic examination , but accuracy of diagnosis may be improved with the help of immunohistochemical staining because such tumours are typically negative for cytokeratin , pancreatic enzyme markers , and endocrine markers but positive for vimentin , cd 10 , cd 56 , and alpha - antitrypsin [ 37 , 9 , 10 ] . we encountered an extremely rare case of spt arising in the mesocolon without ectopic pancreatic tissue .

a solid pseudopapillary tumour ( spt ) is an uncommon pancreatic tumour . very rarely it has also been described outside the pancreas , usually arising from heterotopic pancreatic tissue . in this paper , we described a case arising from the transverse mesocolon without heterotopic pancreatic tissue in an 18-year - old girl .

emphysematous urinary tract infections ( utis ) are infections associated with gas formation that may present as cystitis , pyelitis , or pyelonephritis . uncontrolled diabetes mellitus is a major risk factor for this type of infection , as it provides a favorable microenvironment for the gas forming organisms to grow , . we present a case of emphysematous pyelitis caused by candida tropicalis with good outcome due to early recognition and treatment . a 49 year old man presented to the emergency department on day 0 with a two week history of intermittent right flank pain associated with nausea and vomiting . his medical history included diabetes mellitus , hepatitis c , schizophrenia , and intravenous ( iv ) substance abuse . his home medications included nateglinide , metformin , clonazepam , risperidone , fluoxetine , trazodone , and benztropine . on initial physical examination , he had a fever of 38.7 c ( 101.6 f ) , blood pressure of 150/80 mm hg with a heart rate of 80 beats per minute , and right costovertebral angle tenderness , with otherwise unremarkable findings . the initial laboratory workup showed a hyperglycemia of 496 mg / dl , but no leukocytosis or abnormal renal functions . urine analysis was positive for white and red blood cells with negative nitrite and leukocyte esterase . computed tomography ( ct ) scan showed free air within both collecting systems and urinary bladder with no signs of air inside the renal parenchyma . iv ceftriaxone and insulin therapy were started , foley catheter was placed , and urine cytology was sent to the lab . on further questioning , the patient reported passing friable pinkish material with his urine , like chicken fat he described . on day 1 , the patient 's fever persisted and antibiotic were switched to piperacillin - tazobactam . on day 2 , the hemoglobin a1c was 14.9% and a repeat ct scan with iv contrast was still suggestive of emphysematous pyelitis ( fig . enterovesical fistula was ruled out by an oral - activated charcoal test . on day 4 , the urine culture came back positive for c. tropicalis ( > 100,000 cfu / ml ) identified by vitek ms system . iv fluconazole 400 mg was started followed by 200 mg daily . on day 5 , a final ct scan on day 6 showed resolution of the emphysematous pyelitis with some remaining mild parenchymal edema and he remained afebrile . the patient was discharged on oral fluconazole 200 mg to complete two weeks of therapy . emphysematous infections are usually caused by gas - producing organisms ; mainly bacterial like escherichia coli , klebsiella pneumoniae , proteus mirabilis and pseudomonas aeruginosa . fungal etiology has rarely been reported , but in this case the infection was caused by c. tropicalis . the method of choice for diagnosis and follow up of emphysematous uti is ct scan . class 1 , is when gas is only limited to the collecting system . in class 2 , gas is found in the renal parenchyma without extension to the extrarenal space . in class 3a , gas extends to the perinephric space and in class 3b , it extends to the pararenal space . in class 4 , there is bilateral renal involvement or emphysematous pyelonephritis of a solitary kidney , . the new approach in treating emphysematous utis is the use of systemic antibiotics , together with percutaneous drainage of gas and purulent material . if urinary tract obstruction is present , it must be relieved as well , , . in this case , the patient was class 1 and was successfully managed by systemic antibiotic and antifungal therapy , together with decompression of the urinary system by foley catheter . no invasive intervention was required . he responded well to treatment , mainly due to early diagnosis , sequential radiological assessment , and the fact that it was pyelitis rather than pyelonephritis .

a patient with uncontrolled diabetes mellitus presented with fever and flank pain . a computed tomography scan showed free air within both collecting systems . a diagnosis of emphysematous pyelitis was made after other alternative diagnoses were ruled out . urine culture grew candida tropicalis . the emphysematous pyelitis resolved with conservative management using antifungal therapy .

a 22 weeks pregnant woman came to us for advice regarding the chance of offspring getting retinoblastoma as her first child had bilateral retinoblastoma . the other offspring was 1 year and 9 months old at the time of presentation with a history of leukocoria in the left eye . she was diagnosed to have bilateral retinoblastoma ; group b in the right eye and group d in the left eye ( international classification of retinoblastoma classification ) . systemic chemotherapy ( four cycles ) and external - beam radiation therapy failed to control the tumor in the left eye and hence the left eye underwent enucleation with ball implant , 8 months after presentation . the right eye was salvaged with good tumor control and the preservation of vision [ fig . 1 ] . clinical findings in the sibling showing bilateral retinoblastoma ( a ) ultrasound b scan of the left eye showing large tumor in visit 1 ( b ) fundus drawing of the left eye showing tumor coded by yellow color ( c ) ultrasound b scan of the right eye showing tumor . ( d ) post treatment regressed tumor in the right eye at last follow up ( 7 years follow - up ) a cytogenetic analysis done at that time revealed male karyotype with 13q14 deletion in the father , and the mother was normal . while the mother was found to be normal , the father revealed a retinoma in his right eye . as direct screening by mutational analysis is time - consuming and needs more sample , prenatal cord blood was taken for molecular linkage analysis for retinoblastoma . the fetus was found to have the defective rb1 allele inherited from the father [ fig . 2 ] and gene tracking ( predictive testing ) in the case of familial retinoblastoma the child was born at 34 weeks and the first ophthalmic evaluation done on the 2 day of life was normal . the child underwent periodic monthly examination under anesthesia until 2 years of age . at 28 months of age , she developed a tumor ( group a ) in the inferonasal retinal periphery of the right eye that was treated with cryotherapy [ fig . the child continued to be under bi - monthly clinical follow - up . visual assessment with lea symbol charts showed the visual acuity of 20/20 in both eyes . at a recent follow - up at 7 years of age , the fundus was stable , and visual acuity maintained in both the eyes . fundus photographs ( retcam ) of the child with defective allele detected on prenatal diagnosis ( a ) fundus photograph of the right eye at 2 days of birth . ( c ) fundus photograph of the right eye at 28 months showing tumor at inferonasal periphery ( d ) post treatment cryo , regressed tumor at 3 months follow - up prenatal diagnosis of retinoblastoma with clinical follow - up has been reported earlier , however , to the best of our knowledge , this is the first report from india to show the clinical correlate of genetic findings on a prenatal diagnosis . it is important for an ophthalmologist to determine the etiology of an unilateral case ( whether it is hereditary or not ) as the management of the patient ( examination of fellow eye , frequency of follow - up and sometimes treatment which should be provided along with genetic counseling ) may be different when compared to a bilateral case . in this case , already having a child with retinoblastoma ( with one eye enucleated ) prompted the mother to undergo a prenatal screening for the next offspring . even though primary prevention was not possible in this situation , prenatal diagnosis with serial ultrasound scans of the fetus could help look for a tumor in the fetal eye at an early stage . if detected in utero , a fetus with retinoblastoma can be delivered early to initiate early treatment and possibly reduce the morbidity and preserve the vision . though the child was identified to have the inherited paternal risk allele hence , it is likely that these children may develop tumor at a later stage of life alarming for careful and regular long - term surveillance . thus , prenatal diagnosis offers early detection of the tumors in the course of the disease and periodic examinations aid in eye salvage .

retinoblastoma is the most common malignant intraocular tumor in pediatric age group if undetected leads to ocular mortality . prenatal diagnosis is an emerging technology to detect fatal diseases in utero such that subsequent management is planned to reduce the ocular morbidity . we describe a case demonstrating the importance of prenatal diagnosis in a child with a strong family history of retinoblastoma and importance of a long - term clinical follow - up in these cases .

a 64-year - old healthy man underwent phacoemulsification with intraocular lens implant on the left eye . preoperative fundus examination was normal and his best - corrected visual acuity in the left eye was 6/24 because of nuclear sclerosis . the patient was administered a single uneventful peribulbar injection consisting of 5 ml of lidocaine 2% and epinephrine 1:200,000 using a 25-gauge 25 mm peribulbar needle through the temporal portion of the inferior lid . the patient had no complaints and his visual acuity had improved to 6/6 . however , on examination , a left afferent pupillary defect was noted . dilated ophthalmoscopy of the left eye revealed several large confluent peripapillary patches of retinal whitening mimicking soft exudates ( purtscher flecken ) [ fig . fundus fluorescein angiography revealed multiple hypofluroscent areas in the peripapillary region corresponding to the areas of the soft exudates [ fig . 2 ] . swept - source optical coherence tomography of the left eye revealed edema of the inner retinal layers with hyperreflectivity of the nerve fiber layers corresponding to the areas of retinal whitening [ fig . 3 ] . as the patient was symptomless , no treatment was initiated for the treatment of the retinopathy . the patient underwent a comprehensive medical screening including liver function tests , pancreatic enzyme assay , carotid doppler , and transthoracic echocardiography to rule out other causes of purtscher - like retinopathy . subsequent follow - ups at 2 and 4 weeks revealed gradual disappearance of the cotton - wool spots [ fig . 1b and c ] , resolution of the peripapillary scotoma and decrease in the afferent papillary defect . ( c ) follow - up at 4 weeks fundus fluorescein angiography of the left eye ( postoperative day 4 ) revealed blocked fluorescence in the sites corresponding to the retinal and preretinal hemorrhages , capillary dropout in the areas of purtscher flecken and cotton - wool spots optical coherence tomography of the left eye ( postoperative day 4 ) revealed edema of the inner retinal layers with hyperreflectivity of the nerve fiber layers corresponding to the areas of retinal whitening unilateral purtscher 's retinopathy has been reported following facial trauma , periorbital steroid injection , and retrobulbar anesthesia . there have been two previous case reports of purtscher - like retinopathy after retrobulbar anesthesia . to the best of our knowledge , peribulbar anesthesia is known to be safer , but it has still shown to have the tendency to cause potentially dangerous complications including central retinal artery occlusion through the remote effects of the anesthetic agent , amount injected , speed of injection , and postinjection mechanical compression . the pathogenesis of purtscher 's retinopathy is still a subject of debate with several theories being forwarded since the initial mechanical theory proposed by purtscher . the most currently accepted theory is microembolization , causing arteriolar precapillary occlusion and microvascular infarct of retinal nerve fiber layer , forming cotton - wool spots . although bilateral purtscher - like retinopathy has been linked to the activation of complement and formation of leukoemboli , it is difficult to correlate this systemic mechanism to a local event . in this unilateral case , which was triggered by a local event of peribulbar anesthetic injection , the peripapillary distribution of the cotton - wool spots suggests an infarction of the peripapillary arterioles in an area of no collaterals . it could be hypothesized that the sudden increase in orbital volume might have increased the hydrostatic pressure and reproduced the conditions of a valsalva maneuver . however , this theory is challenged by the fact that considering the significantly larger volume of the extraconal space in comparison to intraconal space , 5 ml of anesthetic agent is unlikely to produce significant hydrostatic pressure to produce vascular occlusion . another possibility is that the infarction might have been caused by the accidental embolization of the central retinal artery or a posterior ciliary artery by either some residual air bubbles in the syringe or an orbital fat embolus mobilized by the needle . most likely , the infarction may have been caused by the vasospastic effects of adrenaline accentuated by the intrinsic vasoconstrictive properties of lidocaine . reported a decrease in retinal blood flow velocity by 1015% , 15 min , respectively , following peribulbar anesthesia without a vasoconstrictive agent like adrenaline . we report this case to inform ophthalmic surgeons and anesthetists that although peribulbar anesthesia avoids direct optic - nerve injury , indirect injury may occur from vasospasm in response to the injection .

purtscher and purtscher - like retinopathy is a distinctive retinal syndrome characterized by ischemic retinal whitening in a peripapillary pattern . we report a case of purtscher - like retinopathy in a healthy 64-year - old man after a routine peribulbar anesthetic injection for cataract surgery . although peribulbar anesthesia is considered to be a safer alternative to retrobulbar anesthesia , it has been associated with unusual but grave complications including central retinal artery occlusion .

dynamic contrast - enhanced magnetic resonance imaging ( dce - mri ) is an important diagnosis tool of breast disease because of its good soft tissue resolution and time - signal intensity curve ; it has a higher sensitivity and often presents malignant breast nodules which are missed by physical examination , mammography and ultrasound ( 1 - 3 ) . we present an interesting case of an asymptomatic 30-year - old female asian patient with a small suspicious breast nodule which was not detected by breast palpation , mammography , and color doppler ultrasound , but was detected by dce - mri . this nodule was surgically removed and pathologically confirmed as hemangioma , a kind of benign vascular tumor which is rarely seen in the breast . we present an asymptomatic 30-year - old female asian patient who gave birth 2 years ago . ten months ago , the patient underwent breast palpation , mammography , and ultrasound ( for the first time ) . the results present a nodule ( 1.6 1.2 0.9 cm ) locating on the right breast at 7 o'clock position with an oval shape , clear boundary , internal hypoechoic , and calcifications . , mr scanning ( ge signa hdxt 3.0 t ) was performed according to the recommendation of a clinician . dce - mri did not detect enhancement of the mass ( breast fibroadenoma with calcification ) . surprisingly , a new small nodule ( 0.7 0.5 0.4 cm ) was found at the upper outer quadrant of the right breast . mri presented an oval mass located on the mammary gland , anterior to the superficial pectoralis fascia with a well - defined margin . 1a ) , while a t2-weighted image showed the mass area of high signal ( fig . the region of interest was obtained from the enhancing area of the lesion to generate a time - signal intensity curve . ultrasound was then carefully performed on the patient ( for the second time ) , showing an oval nodule with a well - defined margin and internal hypoechoic ( fig . two experienced radiologists made a surface marker for the lesion based on the dce - mri and ultrasound presentation . in the operating room , the surgeon confirmed the lesion again by ultrasound ( for the third time ) before the surgery . the lesion was pathologically confirmed as breast capillary hemangioma , which is a benign vascular tumor and rarely seen in the breast ( fig . immunohistochemical staining showed strong positive cd34 expression and negative ck expression of the lesion , which supported our diagnosis ( fig . ultrasound is a very useful tool for checking breast diseases , especially in young women with mammary gland hyperplasia . while it has been reported that hemangiomas always show an oval or lobular shape with well circumscribed or microlobulated margins on sonography ( 4 ) ( which were also seen in our case ) , these characteristics are not sufficiently specific . while hemangiomas often locate in a superficial position ( subdermal or in the subcutaneous tissues ) ( 5 , 6 ) ( which is similar to our case ) , this location makes it difficult to visualize the mass on regular craniocaudal and mediolateral oblique views , tangential views are necessary to prove the superficial nature of the mass . however , benign hemangiomas in the breast have also been described as displaying high vascularity . mri and dce - mri are frequently used to check for breast disease in asian women because of the common occurrence of mammary gland hyperplasia and smaller size of female breasts . it is well known that mri could detect inconspicuous lesions because of its good soft tissue resolution ( 7 ) . breast hemangiomas have been reported to show homogeneous hyperintensity on t2-weighted images as in our case . in a gd - enhanced dynamic study , vorela al et al . reported that hemangiomas present early intensive enhancement followed by a plateau ( 8) , which differs from our case ( washout type curve , indicating malignant breast lesion ) , and the images and curve render it difficult to confirm the diagnosis . the difference of vascular morphology and numbers might be the main reason for the different dce - mri presentation and curve type . angiosarcoma is rare but the most malignant of all breast neoplasm , with a mean survival time of about two years . this tumor presents as an ill - defined , unencapsulated , soft , spongy mass with dilated vascular channels . hemorrhage and necrosis are common , especially in larger tumors . histologically , the endothelial cells of angiosarcoma are usually large , plump , and single layered . hemangiomas are benign vascular tumors of two common types ( capillary or cavernous ) that are based on the size of the vessels involved . in this case , microscope imaging showed that the hyperplastic vascular tumor has a regular morphology and clear boundary , has no interstitial infiltrates , no vascular branching structure , and the papilla structure could be found in the lumens . endothelial cell atypia , papillary tufting , mitotic figures , necrosis , or other features of atypical vascular lesion or angiosarcoma were not identified . it has been reported that breast hemangioma could be found in 11% of postmortem specimens of the female ( 10 ) . if the dce - mri presents a small mass ( usually 2 cm ) with a clear boundary , homogeneous enhancement , superficial location , and even the mass presents a washout type curve , alerting the radiologists that hemangioma could be a possible diagnosis .

breast capillary hemangioma is a type of benign vascular tumor which is rarely seen . little is known about its presentation on dynamic contrast - enhanced magnetic resonance imaging ( dce - mri ) . here , we describe a case of suspicious breast lesion detected by dce - mri and pathologically confirmed as capillary hemangioma . our case indicates that a small mass with a superficial location , clear boundary , and homogeneous enhancement on dce - mri indicates the possible diagnosis of hemangioma , whereby even the lesion presents a washout type curve .

a 55-year - old female patient presented with a four - month history of a continuous burning sensation of the oral mucosa . the patient had witnessed an accident and suffered from major depression 3 years prior to visiting our clinic . the patient had been on the following medication for 3 years : monoprolol 40 mg , benzhexol ( parkin ) 2 mg , sodium valproate ( valprol ) 50 mg , haloperidol ( trancodol ) 5 mg and fluoxetin 100 mg . in the course of managing her depression , the dosage of the drug fluoxetin was briefly increased one year prior to her visit from 100 to 200 mg per day for a month . during this period , the patient experienced a mild burning sensation in her mouth , which returned to normal when the dosage was decreased . the dosage of the same drug was again increased from 100 to 200 mg six months prior to her visit , and the patient had been experiencing symptoms of burning mouth for the last four months . on examination , the patient was apparently healthy , conscious , co - operative , well - oriented , well - built and nourished . intraoral examination revealed that the oral mucosa appeared normal and healthy ; there were no mucosal lesions to explain the pain . the intensity of the burning sensation was rated on a visual analogue scale , with a score of 8 . the patient was referred to her psychiatrist with a request to alter or reduce the dosage of the drug fluoxetin . the drug was discontinued , and her depression was managed instead with the drug colostrinin ( cognate ) . following this change , the patient 's glossodynia disappeared completely within one month . based on her history , clinical features and response after drug cessation , a final diagnosis of selective serotonin reuptake inhibiter ( ssri ) antidepressant - induced bms burning mouth syndrome is defined by the international association for the study of pain as burning pain in the tongue or other oral mucous membrane associated with normal signs and laboratory findings , lasting for at least 4 to 6 months . one of the most commonly affected sites is the tip and anterior two thirds of the tongue , which are the areas of greatest movement in the oral cavity . the prevalence of burning mouth symptoms reported by international studies ranges from 0.6% to 15% . patients are around 60 years of age , and females are more commonly affected than males . lamey and lewis have suggested classifying bms into three patterns : types 1 , 2 , and 3 . type 1 includes symptom - free waking , with sensations developing in the morning and progressively rising to a severe level by evening . type 2 involves continuous symptoms throughout the day ; whereas type 3 features intermittent symptom - free periods throughout the day . nonpsychologic causative factors , such as nutritional deficiencies , have been linked to type 1 , chronic anxiety to type 2 , and food allergies to type 3 . another approach in classifying bms is to divide patients into either primary or secondary groups . while primary bms is idiopathic , secondary bms may be caused by local factors or systemic conditions . local factors associated with bms include mucosal diseases , fungal infections , bacterial invasion , allergies , temporomandibular joint dysfunctions , and salivary gland abnormalities . deficiency diseases , hormonal and immunologic disturbances , and pharmacotherapeutic side effects are included in the systemic conditions . secondary bms requires appropriate diagnosis and treatment of the underlying condition to manage symptoms . in primary bms , set out the guidelines for the management of primary bms based on a systematic review of randomized controlled trials , and recommended the following drugs : ssri , clonazepam , alphalipolic acid , amisulpride and cognitive behavioral therapy . these include the following : efavirenz , clonazepam , hormonal replacement therapies , fluoxetine , sertraline and a broad range of antihypertensive agents such as captopril , enalapril , and lisinopril . thirty - three percent of drug - induced bms have been seen to be dose - dependent phenomena , because the burning sensation appears after elevating the drug dose in search of increased therapeutic efficacy . levenson reported a case of a patient with major depression who developed bms after an increase in the dosage of ssri ( fluoxetine , sertraline ) and whose burning sensation was completely relieved after discontinuation of the medication . the pathogenesis of bms has been described in terms of local factors or conditions that alter the peripheral nerves , resulting in a decreased threshold of burning sensation and peripheral sensitization . if the peripheral sensitization continues for a long period of time , it adversely affects the central nervous system and results in neuroplasticity and central sensitization . the dose of her medication was increased to improve the efficacy of treatment for her depression . however , this resulted in the symptoms of a burning mouth . cessation of the drug is the best way to manage such patients after obtaining physician consultation for the medical management of depression .

burning mouth syndrome ( bms ) is defined as a chronic orofacial pain syndrome , without evidence of mucosal lesions and other clinical signs of disease or laboratory abnormalities . patients with bms complain of burning pain in the mouth , xerostomia and taste disturbances . it is more common among women and the median age of occurrence is about 60 years . bms may be primary or secondary to other diseases . the mainstay in the treatment of bms includes antidepressants , benzodiazepines , and anticonvulsants . a few cases of bms caused due to medication have been reported . the causative drugs include angiotensin - converting enzyme inhibitors , anticoagulants , antipsychotics , antiretrovirals , and benzodiazepines . this is a case report of a patient on antidepressants who developed symptoms of bms thereby causing a dilemma in management .

a 55-year - old man traveled to mainland china from march 5 to march 15 . by march 18 the patient was given a course of antimicrobial drugs , and the fever resolved . by march 23 , at this point , sars was suspected , and the patient was moved into isolation in an intensive care unit the same day and had to be intubated for 5 days . results from the polymerase chain reaction ( pcr ) test indicated the presence of the sars - associated coronavirus . the patient had no previous history of serious respiratory illness or any other serious coexisting conditions , and the source of the exposure could not be determined . the 50-year - old wife of patient 1 was exposed to sars upon her husband 's return on march 15 . on march 23 , a cough developed , and she had shortness of breath and chills . because sars was suspected , the patient was placed on antiviral treatment , and sars was confirmed by pcr testing . patient 2 was placed in isolation on a medical ward on march 25 and remained there until april 11 . the 15-year - old daughter of patients 1 and 2 also became exposed to sars when her father returned on march 15 . , she complained of stomach pain and , upon examination , had a temperature of 37.7c . the 23-year - old daughter of patients 1 and 2 , who lived away the family home , became exposed to sars through contact with both patients 1 and 2 upon a brief visit ( lasting < 1 hour ) on march 19 . by march 27 a pcr test was not completed because her fever resolved , and no other symptoms developed . a 55-year - old man traveled to mainland china from march 5 to march 15 . by march 18 the patient was given a course of antimicrobial drugs , and the fever resolved . by march 23 , at this point , sars was suspected , and the patient was moved into isolation in an intensive care unit the same day and had to be intubated for 5 days . results from the polymerase chain reaction ( pcr ) test indicated the presence of the sars - associated coronavirus . the patient had no previous history of serious respiratory illness or any other serious coexisting conditions , and the source of the exposure could not be determined . the 50-year - old wife of patient 1 was exposed to sars upon her husband 's return on march 15 . on march 23 , a cough developed , and she had shortness of breath and chills . because sars was suspected , the patient was placed on antiviral treatment , and sars was confirmed by pcr testing . patient 2 was placed in isolation on a medical ward on march 25 and remained there until april 11 . the 15-year - old daughter of patients 1 and 2 also became exposed to sars when her father returned on march 15 . , she complained of stomach pain and , upon examination , had a temperature of 37.7c . the 23-year - old daughter of patients 1 and 2 , who lived away the family home , became exposed to sars through contact with both patients 1 and 2 upon a brief visit ( lasting < 1 hour ) on march 19 . by march 27 a pcr test was not completed because her fever resolved , and no other symptoms developed .

with cases of severe acute respiratory syndrome ( sars ) occurring across geographic regions , data collection on the effectiveness of intervention strategies should be standardized to facilitate analysis . we propose a minimum dataset to capture data needed to examine the basic reproduction rate , case status and criteria , symptoms , and outcomes of sars .

the incidence of renal infarction in an autopsy study was 1.4% , whereas clinical diagnosis was made in only 0.014% of the studied patients . the clinical manifestations of renal artery thrombosis vary , depending on whether the occlusion affects both renal arteries or not . delay of the correct diagnosis may prove disastrous for the patient . our case is probably the first one in english literature describing a patient with unilateral renal artery thrombosis after lobectomy for lung cancer . a 53-year - old female patient was admitted to our hospital with the diagnosis of lung cancer . her medical history was clear , except that she was a social drinker and smoker ( 3 - 4 glasses of wine / week , 5 cigarettes / day for 25 years ) . physical examination and blood test analysis were normal , and cardiac function was found to be excellent . the preoperative transbronchial needle aspiration cytology revealed an adenocarcinoma in the left upper lobe . computed tomography ( ct ) of the thorax , abdomen and brain revealed nothing apart from a mass located in the left upper lobe , 2 cm in diameter . the patient received a formal left upper lobectomy in combination with mediastinal lymph node sampling . no complications were observed during the operation , and the immediate postoperative period was straightforward until the third postoperative day , when she began to complain of a flank pain located at the lower side of the left hemithorax and the nearby lumbar area . the pain was not affected by respiratory movements and position changes , and it was continuous without radiation . laboratory data showed a minor elevation of white blood cell count ( 12,500 cells / mm ) and lactate dehydrogenase ( 371 consequently , the patient was treated with painkillers and anti - inflammatory agents . despite the medication given , pain persisted for the next 24 h without any changes . an upper abdomen ultrasound scan was performed combined with a chest x - ray revealing nothing that could explain the condition . an expert nephrologist was called in order to manage the problem . however , precious time was already lost and further therapy with thrombolytics did not offer significant improvement , since the left kidney was considered already nonfunctioning . the levels of blood urea nitrogen and creatinine did not show remarkable elevation compared with the preoperative ones ( urea : 33 41 mg / dl , creatinine : 0.71 0.83 mg / dl ) . the patient was discharged two weeks after the operation and anticoagulation therapy with warfarin was given . six months later , renal function remains satisfying and the patient is free of any symptoms . it is of crucial importance for acute renal artery thrombosis , although it is an uncommon situation , to be diagnosed early . in the vast majority of the cases , however , significant delay is the rule , causing irreversible damage to the kidney . the clinical presentations of renal arterial thrombosis are not specific for the disease and usually include flank pain originating from the ipsilateral lumbar area , accompanied by fever , nausea and vomiting anuria or oliguria combined with proteinuria or hematuria are findings indicating bilateral renal artery occlusion . blood tests are not apt to establish an accurate diagnosis because of lack in sensitivity and specificity . common causes of renal artery occlusion include congestive heart failure , atrial fibrillation , and valvular or ischemic heart disease . moreover , trauma , clotting disorders , cancer and some rare hereditary diseases are also mentioned . plain x - ray and ultrasonography can be significantly helpful , but the final diagnosis is usually determined by contrast - enhanced ct . invasive diagnostic tools such as renal artery angiography are not without complications . in our case , the diagnosis was delayed and as a result the left kidney lost its function . given that the patient was free of cardiac diseases and clotting disorders , lung cancer could have been the cause for renal arterial thrombosis . although the patient was receiving subcutaneous low molecular weight heparin 3,500 iu from the first postoperative day , this very serious complication did happen . diagnosis was reached incidentally with the help of contrast - enhanced ct , which seems to be the gold standard for renal artery occlusion diagnosis . therapy in acute renal thrombosis typically consists of low molecular weight heparin in the acute phase , followed by warfarin . the value of antiplatelets is still under study . because of the high risk for more thromboembolic complications in these patients it is advisable that the anticoagulation treatment should be continued , and a careful follow - up is mandatory in conclusion , renal arterial thrombosis is a rare and serious condition , which should be diagnosed early . nonspecific symptomatology complicates the differential diagnosis and a high level of suspicion is required in order to not lose precious time . if lumbar flank pain appears and all other possible diagnoses are excluded , contrast - enhanced ct should be considered . apart from the usual causes , the clinician should keep in mind this extremely uncommon case , which may be the first one described in english literature .

acute renal arterial thrombosis is a rare but very urgent situation demanding immediate treatment . it is characterized by unspecific symptomatology which often misleads the clinicians . as a result , precious time can be lost until the correct diagnosis is reached . the case of a 53-year - old female who underwent a left upper lobectomy for lung cancer is presented . on the third postoperative day , the patient began to complain of a flank pain located at the lower side of the left hemithorax and the nearby lumbar area . a renal arterial thrombosis was finally diagnosed and subcutaneous low molecular weight heparin was started immediately . the patient was discharged two weeks later and anticoagulation therapy with warfarin was given . six months later , renal function remains satisfying and the patient is free of any symptoms . this is probably the first case in english literature of renal arterial thrombosis following lobectomy for lung cancer .

a 70-year - old japanese man presented at our hospital with an asymptomatic , 20 30 mm irregularly shaped blackish , flat elevated plaque with a gray nodule in the periphery on his left lower leg ( fig . the lesion had been present for 10 years and had recently enlarged , associated with bleeding . dermoscopy revealed leaf - like areas , large blue - gray ovoid nests and multiple blue - gray globules ( fig . basal cell carcinoma and malignant melanoma were listed as suspected diagnoses , and we performed an excisional biopsy of the tumor . histopathological examination of the tumor revealed infundibular structures of the outer hair sheath in the center of the plaque , which consisted of three distinct parts ( fig . 2a ) . the first part showed circumscribed massive aggregation of basophilic basaloid cells containing abundant melanin granules with peripheral palisading and retraction spaces ( fig . this part of the tumor exhibited trichilemmal keratinization with squamous eddies , which were surrounded by a fibrous stroma ( fig . the third part showed reticular aggregation of basaloid cells with small infundibular cystic structures in the papillary dermis ( fig . the stromal part showed no fibrosis and abundant mucin deposition staining positive with alcian blue ( fig . immunohistochemical staining revealed diffuse positivity for anti - bcl-2 in the first part ( fig . 3b ) , positivity only at the periphery of tumor nests in the second part ( fig . microscopic examination of the tumor showed that it consisted of three distinct histological types : massive aggregation of basophilic cells was diagnosed as solid - type basal cell carcinoma , massive aggregation of clear cells with trichilemmal keratinization was diagnosed as proliferating trichilemmal tumor , and reticular small aggregations of basophilic basaloid cells in the papillary dermis were diagnosed as infundibulocystic basal cell carcinoma . basal cell carcinoma with follicular differentiation in 1987 by tozawa and ackerman , and later in 1990 the tumor was proposed as a variant of basal cell carcinoma showing differentiation to infundibular cyst - like structures by walsh and ackerman . clinically , the tumor presents as small papules / nodules , most commonly on the face . characteristic histopathological features of the tumor are small symmetrical , circumscribed aggregations of basaloid cells containing numerous infundibular cyst - like structures . infundibulocystic basal cell carcinoma is different from trichoepithelioma in that there are no follicular bulbs and papillae , no stromal fibrosis around the tumor , reticulated pattern of neoplastic cells , and abundant epithelial mucin . in the present case , anti - bcl-2 immunohistochemical staining was diffusely positive in portions of typical basal cell carcinoma and infundibulocystic basal cell carcinoma . , bcl-2 is present within basal keratinocytes , mesenchymal cells of follicular papilla , clear cells of eccrine glands , melanocytes and lymphocytes . anti - bcl-2 staining is useful for distinguishing between trichoepitheliomas and basal cell carcinoma , the former staining positively in the periphery of tumor nests , whereas the latter shows diffuse and widespread staining in all tumor nests . basal cell carcinoma is a neoplasm proposed to be derived from epithelial germ cells , and infundibulocystic basal cell carcinoma is derived from the infundibulum of the hair follicle , whereas proliferating trichilemmal tumor is derived from the hair follicle isthmus . there have been four reports of basal cell carcinoma arising with trichilemmoma without a pre - existing sebaceous nevus . taken together , the present findings suggest that basal cell carcinoma may be derived from hair germ cells , including the outer hair sheath and hair follicular infundibulum . the authors report no conflicts of interest . there were no funding sources for this work .

a 70-year - old japanese man presented at our hospital with an asymptomatic , blackish , irregularly shaped plaque with a gray nodule in the periphery on his left lower leg . the lesion had been present for 10 years and had recently enlarged , associated with bleeding . histopathologically , the tumor consisted of three distinct parts : the first part showed massive aggregation of basophilic basaloid cells with peripheral palisading and abundant melanin granules , and was diagnosed as solid - type basal cell carcinoma . the second part showed aggregation of clear cells with squamous eddies , and was diagnosed as proliferating trichilemmal tumor . the third part showed reticular aggregation of basaloid cells with infundibular cysts in the papillary dermis , and was diagnosed as infundibulocystic basal cell carcinoma . we diagnosed this tumor as basal cell carcinoma with various forms of hair follicle differentiation , including differentiation into the outer root sheath .

a male patient was referred to a physician in a clinic with the chief complaint of abdominal pain for 2 3 days . the patient 's sister noted that the patient was a psychiatric patient diagnosed with depression , anxiety as well as drug addiction . the patient 's sister also noted that the patient had no verbal communication due to intentional omitting of speaking and denying all drugs for 7 days . considering the patient 's history , the patient also presented agitation , sleeplessness and increased appetite at that period , which matched the description of amphetamine withdrawal . this case was examined by the physician in charge and he found that the patient had voluntary abdominal guarding and poor cooperation with physicians . the patient was firstly suspected to be a possible case of drug withdrawal , and the abdominal pain was treated by oral and intravenous antispasmodic drugs . however , the patient had no better symptom . the patient was referred to the hospital and monitored at the emergency room for one more day . finally by observing the generalized guarding of abdomen and considering the laboratory results ( negative urinalysis , neutrophilia from complete blood count ) , the patient was diagnosed as a possible case of appendicitis . acute appendicitis is a difficult - to - diagnose condition in many situations ( 4 , 5 ) . the diagnosis of acute appendicitis can be delayed in many cases and can cause problems . in this report , this case presents difficulty in diagnosis . the patient 's signs and symptoms of appendicitis are not classical and fully mimicked with the underlying psychiatric signs and symptoms . indeed , the abdominal pain can be an important presentation in the drug withdrawal case ( 6 , 7 ) . it should be noted for the importance of concealing a physical diagnosis by psychiatric history or mental states ( 3 , 8) . it is estimated that about one twentieth to one tenth of the psychiatric cases might have occulted somatic disorders ( 9 , 10 ) . therefore , this case report suggests that general practitioners should be aware of possible undetected physical disorders in psychiatric cases .

physical disorders can be seen in psychiatric patients . in addition , a delayed diagnosis can cause a serious complication of the physical disorder among such patients . in this report , a case of appendicitis in a psychiatric case with drug withdrawal symptoms was reported .

hemangiomas are generally classified into capillary , cavernous , mixed and sclerosing types according to the type of blood cells they are composed . hemangiomas are mostly seen in the head and neck region and thought to originate from endothelial progenitor cells . oral capillary hemangiomas consist small - sized , and thin walled capillary - like vessels , which are lined with a layer of endothelial cells and usually are localized on oral soft tissues . clinically , capillary hemangiomas are soft , small - sized , lobulated , or solitary lesions with a color range between red and blue due to the profundity of the lesion . oral capillary hemangiomas are rarely seen in adults and mainly localized on oral mucosa or gingiva . the intraosseous form of capillary hemangioma is an uncommon entity and reported to be seen in the metacarpal , frontal , and temporal bones . in this report , an unusual case of intraosseous capillary hemangioma of the mandible was presented with radiological and histological features . a 68-year - old female patient referred to our clinic for further examination of the radiolucent cyst - like lesion on her mandible . the clinical examination was normal , and the patient had no complaints regarding the lesion . the lesion was located on left canine - premolar region with sclerotic curly borders that is suggestive of a benign lesion [ figure 1 ] . the sclerotic boundaries of the lesion were prominently revealed on the axial ct section [ figure 2 ] . panoramic radiography revealed radiolucent cyst - like lesion with irregular sclerotic borders localized at the mandibular canine - premolar region axial computed tomography section reveals irregular , nonuniform sclerotic borders of the lesion vertical and sulcular incisions were performed to expose the operation site . a direct approach to the lesion was provided with surgical burs with copious saline irrigation . surgical curettage was performed with unexpected moderate bleeding of the lesion [ figure 3 ] . the surgical specimen has a solid structure with bluish - brown color [ figure 4 ] . a provisional diagnosis of central giant cell granuloma was made due to the intraoperative unexpected bleeding . histological examination revealed numerous small - sized vessels that are consistent with capillary vessels [ figure 5 ] . a single layer of endothelial lining was present in the proliferating capillary vessels [ figure 6 ] . bleeding of the lesion can be clearly seen postoperative appearance of the surgical specimen capillary hemangioma consisting of proliferated , small - sized blood vessels ( he 40 ) proliferated blood vessels lined by a single layer of bland endothelial cells ( he 100 ) intraosseous hemangiomas of the jaws are benign vasoformative neoplasms . they are asymptomatic and mostly discovered incidentally on plain radiographs . intraosseous hemangiomas are commonly localized in calvaria and spine and affects long bones and other skeletal bones less frequently . patients with high- pressure hemangiomas often report a sense of pulsation at the related region . reported two cases of cellular hemangioma , which may correspond to capillary hemangioma on the mandible in their study according to a previously suggested classification system . several cases of cavernous intraosseous hemangiomas of the jaws were reported with clinical and histopathological features . a preoperative arteriogram is recommended for screening of the supplying vascular structures and observation of soft tissue extension . trabecular thickening and sclerotic punctate areas which are called polka - dots can be seen on ct examinations of skeletal intraosseous hemangiomas . plain radiography and ct findings of intraosseous mandibular hemangiomas include unilocular radiolucency with sclerotic rims , resembling a jaw cyst or sunburst or honeycomb appearance radiating from central to the periphery with or without radiopaque areas . capillary intraosseous hemangioma in the current report showed irregular , but well - defined borders in both panoramic radiography , and ct imaging which is consistent with reports of intraosseous mandibular cavernous hemangioma in the literature . if the lesion is asymptomatic or superficial and cause no esthetical problems , they can be left untreated . in general , surgical excision with or without preoperative embolization is suggested in the treatment of intraosseous or soft tissue hemangiomas . preoperative embolization was not considered due to the small size and indolent radiographic appearance of the lesion . however , capillary hemangioma of the mandible has been an uncommon and undefined entity . to our knowledge , radiographic findings of the lesion are similar with a cavernous form of intraosseous mandibular hemangioma . ct imaging of intraosseous hemangiomas enables the clinician to examine the borders and content of the lesion more carefully , and helps the surgeon to decide the treatment modality that is suitable for the current case .

intraosseous hemangioma is a benign vascular neoplasm , which is mostly seen in vertebrae , maxillofacial bones , and long bones . intraosseous hemangioma is rarely seen on jaw bones compared to other skeletal bones and usually occurs in the cavernous form . capillary intraosseous hemangioma of jaws is an uncommon form of intraosseous hemangioma and has not been thoroughly described so far . in this study , a case of capillary intraosseous hemangioma of the mandible was presented with relevant literature review .

lupus vulgaris is the most common morphological presentation of cutaneous tb that can present as papular , nodular , plaque , ulcerative , vegetating , and tumid forms . it often originates from an underlying focus of tb , typically in a bone , joint or lymph node , and spreads by either contiguous extension of the disease from underlying affected tissue , or by hematogenous or lymphatic spread . it is a paucibacillary type of cutaneous tb and occurs in a patient of a moderate immunity . a typical feature of lupus vulgaris is its extremely chronic course , characterized by a slow and steady growth of the lesion over a period of years . unusual variants include the frambesiform , gangrenous , ulcerovegetating , lichen simplex chronicus , myxomatous , and sporotrichoid types . herein we report a rare sporotrichoid presentation of the disease . a 28-year - old female with family history of tb presented with multiple , asymptomatic , raised skin lesions of variable size and shape over her right lower limb . lesions first appeared on the upper surface of her right foot 12 years ago [ figure 1 ] after a thorn prick . thereafter , they extended steadily upward to her inner thigh , increasing both in number and size . there was a history of scarring at one end of the lesions and extension from the other . examination of the right lower limb revealed polysized plaques of 4 - 8 cm diameter with central scarring and peripheral nodularity extending in a linear array from the dorsum of the lateral three toes to the medial aspect of ankle joint , knee joint , and medial aspect of the upper thigh in a sporotrichoid pattern [ figure 2 ] . histopathology revealed hyperkeratosis , acanthosis and elongation of the rete ridges with dense dermal granulomas comprising epithelioid cells , langhan 's type giant cells and chronic inflammatory cells , supporting a diagnosis of lupus vulgaris [ figure 3 ] . plaque from dorsum of the foot in a sporotrichoid pattern plaque in sporotrichoid pattern histopathology showing granulomas comprising epitheloid cells and langhan 's type giant cells ( h and e stain , 40 ) a final diagnosis of sporotrichoid - lupus vulgaris was made and the patient referred to the government tb clinic for category i dots . sporotrichoid form of lupus vulgaris is an unusual variant that mimics sporotrichosis , a subcutaneous fungal disease . bacilli follow the lymphatic channels and during transit , provoke cutaneous granulomatous inflammation resulting in a linear array of papular , nodular and ulcerative lesions over time . it has been shown that sporotrichoid form is more common in children than in adults . the efficient lymphatic drainage in children and high physical activity that makes them prone to trauma may be responsible for this form . as it is a complex process and takes time

lupus vulgaris is the most common presentation of cutaneous tuberculosis in india and can present as papular , nodular , plaque , ulcerative , vegetating , and tumid forms . unusual variants include the frambesiform , gangrenous , ulcerovegetating , lichen simplex chronicus , myxomatous , and sporotrichoid types . we describe a rare sporotrichoid presentation of lupus vulgaris on the leg of a 28-year - old female of 12 years duration .

a 65-year - old female visited korea university anam hospital complaining of chest pain , palpitation , and dyspnea . on the basis of computed tomography , she was diagnosed with takayasu s arteritis including a large aneurysm ( 58 mm ) on the aortic distal arch ( fig . because the patient strongly refused surgery , we performed thoracic endovascular aortic repair ( tevar ) with left subclavian artery ( lsca ) to left common carotid arterial transposition ( fig . after tevar , the patient s symptoms improved , and she was discharged on postoperative day 10 with a normal chest x - ray ( fig . the patient returned to the hospital complaining of moderate dyspnea on postoperative day 19 , and the follow - up chest x - ray showed a severe left pleural effusion ( fig . after the insertion of a chest tube , postoperative chylothorax was confirmed by a pleural fluid analysis . conservative management with a fat - free diet and nil per os was attempted for 2 weeks , but there was no remarkable improvement . therefore , we administered a subcutaneous injection of octreotide ( 0.1 mg ) every 8 hours for 2 weeks ; however , even these injections had no effect . surgical treatment was also considered , but given the possibility of unsuccessful repair due to the uncertainty of the exact site of the thoracic duct leakage and the patient s strong refusal , it could not be our next option . therefore , we performed an intranodal lymphangiogram using lipiodol ( poppy seed oil used as a radio - opaque contrast agent to outline structures in radiological investigations ) on postoperative day 46 . a total volume of 3 to 6 ml of lipiodol was injected into each lymph node at the inguinal area under the guidance of ultrasonography . during the fluoroscopy , the lipiodol had migrated to the retroperitoneal lymphatics but did not advance beyond the l3 level . however , the chest x - ray taken 3 days after the thoracic duct embolization ( tde ) revealed that the lipiodol had migrated up to the subclavian lymphatics ( fig . the chest tube drainage suddenly decreased on the 3rd day after tde , and we were finally able to remove the chest tube on the 13th day after tde ( fig . postoperative chylothorax is a rare but serious complication of the thoracic and esophageal surgical procedure . further , a significant loss of immunoglobulins , t lymphocytes , and proteins into the pleural cavity results in immunosuppression . conservative treatments include drainage of the pleural effusion , a diet including medium - chain triglycerides ( mcts ) , total parenteral nutrition ( tpn ) , and injection of somatostatin analogs like octreotide . an mct diet and tpn reduce the chyle s flow , and somatostatin analogs reduce the intestinal chyle production . in case the high - output chylothorax responds poorly to conservative management , surgical treatment such as thoracic duct ligation , pleuroperitoneal shunt , pleurodesis , or pleurectomy is required . lymphangiography has also been considered a treatment of chylothorax , being less invasive than surgery and having a success rate of about 80% ( table 1 ) [ 36 ] . the mechanism is thought to be caused by lipiodol , which produces an inflammatory process and occludes the chyle leak . complications of lymphangiography are pulmonary oil embolism , hypersensitivity , and lymphatic obstruction , but these are rare and usually minor . in our case , postoperative chyle leakage was not reduced or improved despite the trial of conservative management and octreotide injection . therefore , we performed an intranodal lymphangiogram and tde with lipiodol . through this case , we verified that tde with lipiodol could be considered an alternative for the treatment of postoperative chylothorax , if conservative medical treatment is unsuccessful and surgical treatment is not possible .

chylothorax is a rare postoperative complication of a thoracic surgical procedure . here , we report a case of chylothorax after thoracic endovascular aortic repair with debranching for the distal arch aneurysm of the aorta . first , the patient was treated by a medical method ( nil per os , fat - free diet , and octreotide ) , but this method failed . the patient strongly refused surgical treatment . therefore , we tried to occlude the thoracic duct by lymphangiography lipiodol , and this line of treatment was successful .

since the last description of rebase in the 2003 nar database issue ( 1 ) , there has been considerable growth in the size of the database primarily due to the large number of restriction modification ( rm ) genes that can be found in the sequence databases . more than 200 bacterial and archaeal genomes are available from genbank ( 2 ) and it is now clear that rm systems are much more common than had once seemed likely . mainly , this is because of the difficulty of detecting type i systems or solitary dna methyltransferases by biochemical or genetic assay . putative rm genes identified in these genomes are named systematically according to recently published nomenclature rules ( 3 ) and all have the suffix the rebase website ( http://rebase.neb.com/rebase/rebase.html ) summarizes all information known about every restriction enzyme and their associated proteins . this includes source , commercial availability , sequence data , crystal structure information , cleavage sites , recognition sequences , isoschizomers and methylation sensitivity . within the reference section of rebase , links are maintained to the full text of all papers whenever that is freely available on the web . rebase includes links to genbank and pubmed , and ncbi 's linkout utility uses rebase , pubmed and genbank record numbers to hook directly into rebase 's enzyme , sequence , reference and genome data . links to other major databases such as swissprot ( 4 ) , pdb ( 5 ) and pfam ( 6 ) are also maintained . there are currently 3681 biochemically characterized restriction enzymes in rebase and of the 3612 type ii restriction enzymes , 588 are commercially available , including 221 distinct specificities from a total of 253 total specificities known . as can be seen from figure 1 , the major growth in rebase during the previous two years has been in the number of putative genes for rm system components . more than 620 restriction enzyme genes and 2200 dna methyltransferase genes can be identified in genbank entries . the method used to identify putative rm genes in dna sequences has three fundamental components : the rebase database itself , an expert - derived set of rm system features and a computer program designed to spot these features in anonymous sequences . each sequence analyzed is checked for its overall sequence similarity to rebase gene sequences . for dna methyltransferase sequences , which are the primary indicator of an rm system , the presence , proper order and characteristic spacing of well - conserved motifs suggest the candidates . the more widely divergent genes of the restriction enzymes reside close to the genes for their cognate methyltransferases . such associations point to potential restriction enzyme genes , even when they lack any similarity to genes of known enzymes . publicly available sources of non - eukaryotic sequences are also analyzed frequently by this system . specialized information is available from the rebase lists icon and information about the sensitivity of restriction enzymes to dna methylation can be found by clicking on the rebase methylation sensitivity icon . in the latter case , the data is shown in double - strand format so that the effects of hemi - methylation and double - strand methylation are clearly differentiated . nebcutter analyzes dna sequences for the presence of restriction enzyme recognition sites ( 7 ) . rebsites will generate theoretical digests of an input dna with each of the 253 known specificities . rebpredictor is a tool for predicting restriction enzyme recognition sites that is an updated version of tables ( 8) and a specific blast ( 9 ) option permits a new sequence to be analyzed for rm genes . the rebase genomes icon leads to data for the currently sequenced 193 bacterial and 21 archaeal genomes . schematic representations of the whole genomes and the individual rm system within them are available and , from the pages showing the sequence schematics , there are links to the major database entries for these genes as well as links that will identify the closest neighboring sequences . this can be extremely useful in making predictions about the recognition sequence specificity of newly sequenced systems . this whole section of rebase provides a valuable resource for the annotation of the rm genes in a newly sequenced bacterial genome , particularly given the large numbers of rm systems that are often found . scientists interested in using the sequence information in rebase to annotate microbial genomes are encouraged to contact the rebase staff . special thanks are due to the many individuals who have so kindly contributed their unpublished results for inclusion in this compilation and to the rebase users who continue to steer our efforts with their helpful comments . this database is supported by the national library of medicine ( lm04971 ) and new england biolabs .

rebase is a comprehensive database of information about restriction enzymes , dna methyltransferases and related proteins involved in restriction modification . it contains both published and unpublished work with information about recognition and cleavage sites , isoschizomers , commercial availability , crystal and sequence data . experimentally characterized homing endonucleases are also included . additionally , rebase contains complete and up - to - date information about the methylation sensitivity of restriction endonucleases . an extensive analysis is included of the restriction modification systems that are predicted to be present in the sequenced bacterial and archaeal genomes from genbank . the contents of rebase are available by browsing from the web ( http://rebase.neb.com/rebase/rebase.html ) and through selected compilations by ftp ( ftp.neb.com ) and as monthly updates that can be requested via email .

reactive perforating collagenosis is a rare perforating disorder.it is characterised by collagen bundles perforating the epidermis . reactive perforating collagenosis ( rpc ) is a rare form of transepidermal elimination ( tee ) disorder in which there is extrusion of altered collagen from the epidermis . rpc can exist in two forms ; the commoner acquired form and the rare familial form . in the familial form , the lesions manifest in early childhood is not associated with pruritus and usually spontaneously resolves . we herein report a rare case of rpc in a young child with similar complaints in sibling . a 4 year old boy presented with complaints of multiple , recurrent , dark coloured raised lesions on face , elbows and outer aspect of both legs of 2 years duration . the lesions would start as skin coloured papules which later developed dark central material . some of the lesions would heal on its own in about a month leaving behind residual hyperpigmentation . patient 's mother complained that she had noticed new lesions occurring at sites of trauma . there was history of similar complaints in his elder brother who was staying with his grandparents . dermatological examination revealed multiple , discrete , symmetrical , hyperpigmented , hyperkeratotic papules distributed over face , both elbows and lateral aspect of both legs [ figure 1 ] . hyperkeratotic papules with central plugging over elbows and evidence of koebnerisation similar lesions on lower limbs routine lab investigations were normal . epidermis showed a hyperkeratotic crater filled with keratin , few polymorphonuclear leucocytes and occasional acellular basophilic fibrous strands . the adjacent papillary dermis showed vertically oriented collagen bundles perforating the epidermis [ figure 3 ] . the perforating collagen bundles were better delineated on van gieson stain [ figure 4 ] . based on clinical and histopathological features , the lesions showed some flattening with therapy but the patient continued to get new lesions at sites of minor trauma . histopathology of skin lesions revealing epidermal parakeratosis and perforating bundles of collagen ( h and e , 40 ) perforating collagen bundles evident in van gieson stain ( van gieson stain , 40 ) . there are four different types of perforating disorders and all are characterized by transepidermal elimination of altered dermal substances . they are kyrle 's disease , perforating folliculitis , elastosis perforans serpiginosa and reactive perforating collagenosis . the inherited form of rpc is associated with a positive family history in about two thirds of patients . consanguinity in parents is present in some cases with an autosomal recessive mode of inheritance , though dominant types have also been mentioned . there is a slight male preponderance with siblings being affected as in our case . in the acquired variant , the lesions manifest after the age of eighteen years and are usually associated with itching . it is also associated with hypothyroidism , hyperparathyroidism , liver disorders , hodgkin 's disease , neurodermatitis , hiv and iga nephropathy . the morphology of lesions is similar in both the entities and consists of umbilicated papules and nodules that can be skin coloured , erythematous or hyperpigmented and characteristically have a central keratotic plug . koebnerisation is commonly seen as in our patient . in genetically predisposed individuals , trauma results in altered collagen and scratching induces lesions and common sites of involvement are the accessible and trauma prone sites , as seen in our patient also . research has shown that in most cases no abnormality exists in the ultrastructure of the extruded collagen , but histochemical changes may be present . histologically , early lesions show variable acanthosis and accumulation of collagen , which is basophilic in dermal papillae . mature lesions show characteristic features of rpc where there is plug of parakeratotic material , basophilic debris and degenerated collagen lying within cup - shaped depressions in epidermis . as the condition resolves the important treatment options are topical retinoids , topical corticosteroids , intralesional steroids , psoralen ultra violet - a , narrow band uv - b ( puva , nbuvb ) and allopurinol . the familial form can also be associated with pruritus and may not spontaneously resolve as in our patient , which is not reported before . reactive perforating collagenosis can be rarely familial.the familial form can also be associated with pruritus and may not spontaneously resolve as in our patient . the familial form can also be associated with pruritus and may not spontaneously resolve as in our patient .

a 4 year old boy presented with history of itchy raised lesions on body of 2 years duration . though parental consanguinity was not present , his elder brother had similar complaints . dermatological examination revealed multiple hyperpigmented papules with a central keratotic plug distributed mainly over face and extensors of upper and lower extremities . koebnerisation was present . skin biopsy revealed perforating collagen bundles in the upper dermis and epidermis which was confirmed by van gieson staining . patient was being treated with topical retinoids and intralesional corticosteroids with minimal relief .

bevacizumab is a recombinant humanized monoclonal antibody that selectively blocks the activity of vascular endothelial growth factor ( vegf ) receptor . the addition of bevacizumab to first or second line chemotherapy was associated with longer overall survival ( os ) and progression free survival ( pfs ) in metastatic colorectal patients . the adverse events associated with bevacizumab include hypertension , proteinuria , thromboembolism , impaired wound healing , bleeding , perforation , reversible leukoencephalopathy syndrome , skin rash , and infusion - related hypersensitivity reactions . we present here a case of fatal necrotizing fasciitis in a patient during bevacizumab treatment for colorectal cancer . low anterior resection with ileostomy was performed for rectal mass 5 cm from anus . according to the tumor , node , metastases classification , the pathological stage of the carcinoma was t3n1m0 ( stage iiia ) . the patient was treated with adjuvant 45 gy chemoradiotherapy with 5-fluorouracil 225 mg / m daily and then fluorouracil - leucovorin - oxaliplatin ( folfox4 ) regimen . after two cycles of folfox4 regimen , serum carcinoembryonic antigen levels had increased . in the radiologic evaluation with chest and abdominal computed tomography , new liver metastases in both lobes the hepatic metastases were unresectable , hence the patient shifted to regimen of 5-fluorouracil - leucovorin - irinotecan ( folfiri ) . after 12 courses of the folfiri regimen every 2 weeks , the chemotherapy was stopped due to the stable liver metastases . because of the new metastatic lesions in the liver and pelvic recurrence , bevacizumab added to folfiri regimen . after 10 days of the third cycle of the folfiri - bevacizumab regimen , the patient was admitted with fever , weakness , abdominal pain and erythema of the proximal side of right thigh . the laboratory evaluation revealed a white blood cell count of 22.000/l ( normal value 4,400 - 11,000/l ) with increased c - reactive protein to 160 mg / l ( normal value : 0 - 10 mg / l ) . the magnetic resonance imaging ( mri ) of the pelvis showed widespread significant air - fluid level abscess in the tissues of right gluteus maximus , gluteus minimus and vastus muscles . ultrasonography - guided drainage of the abscess was performed and 10f pigtail catheter was inserted to the abscess location . the microbiological culture of the material was revealed vancomycin - resistant enterococcus ( vre ) , escherichia coli , and bacteroides fragilis . patient responded to treatment for initial few days , but on the 7 day of the antibiotic treatment , acute renal failure and septic shock was developed . necrotizing fasciitis is an uncommon severe soft tissue infection involving the subcutaneous fat and fascia . there are an estimated 3.5 cases of necrotizing fasciitis per 100,000 persons , with a case - fatality rate of 24% despite immediate treatment . severe and acute onset of the pain at the infectious site is the most common clinical presentation . the risk factors of necrotizing fasciitis are diabetes mellitus , malnutrion , trauma , operative interventions , and nonsteroidal anti - inflammatory drugs ( nsaids ) usage . rarely , necrotizing fasciitis can develop due to all - trans - retinoic acid , bisphosphonates , and radiotherapy . in addition , necrotizing fasciitis has also been reported in renal transplant recipient who treated with fk506 . serious adverse events with bevacizumab treatment were hemorrhage , gastrointestinal perforation , and arterial thromboembolic events . one of the pathophysiologic mechanism of necrotizing fasciitis is subcutaneous arteries thrombosis and tissue ischemia , bevacizumab can be the etiologic factor for this . the naranjo probability score revealed that it was probable ( + 5 ) that bevacizumab might be responsible for necrotizing fasciitis . we believe that in our patient , necrotizing fasciitis is due to bevacizumab treatment because there was no additional risk factor and there is temporal relationship between necrotizing fasciitis and bevacizumab treatment . in conclusion , we presented fatal necrotizing fasciitis in a patient during bevacizumab treatment for rectal cancer . although precise mechanism is not clear , necrotizing fasciitis is a fulminant disease and can be observed during bevacizumab treatment .

bevacizumab is a recombinant humanized monoclonal antibody that selectively blocks the activity of vascular endothelial growth factor ( vegf ) receptor and it is used in metastatic colorectal patients . we present here a case of fatal necrotizing fasciitis in a patient during bevacizumab treatment for colorectal cancer . in our review of the literature , necrotizing fasciitis was not reported before or during bevacizumab treatment .

initial laboratory values were : wbc 2900/l , anc 900/l , hemoglobin 12.0 g / dl , mcv 95 fl , and platelet count of 156,000/l . creatinine , ast , alt , bilirubin , tsh , iron , total iron binding capacity , ferritin , and vitamin b12 levels were normal . past medical history was significant for coronary artery disease , ulcerative colitis , and stage ii esophageal adenocarcinoma treated with neoadjuvant concomitant radiation plus carboplatin and paclitaxel followed by ivor - lewis esophagectomy . postoperative esophagocutaneous fistula formation led to long - term reliance on jejunal feeding for all caloric intake . his medications were lorazepam , aspirin , amiodarone , lisinopril , balsalazide , metoprolol , and vitamin b12 . amiodarone was discontinued . a repeat cbc after 1 month showed wbc 2600/l , anc 200/l , hemoglobin 12.7 g / dl , mcv 107 fl , and platelet count of 174,000/l . a second repeat cbc after 2 months following discontinuation of amiodarone showed : wbc 2900/l , anc 300/l , hemoglobin 11.0 g / dl , mcv 114 fl , and platelets 193,000/l . examination of a bone marrow aspirate and biopsy specimen revealed nor mocellular bone marrow with progressive trilineage hematopoiesis , mild erythroid hyperplasia , with megaloblastoid features , and vacuolated erythroid cells , highly suspicious for myelodysplastic syndrome ( figs1 and 2 ) . additional laboratory evaluation included rheumatoid factor 12 , esr 30 , crp 0.4 , serum zinc level 86 g / dl ( 60130 ) and serum copper < 10 g / dl ( 70140 ) . a cbc after 1 month revealed wbc 7600/l , anc 5400/l , hemoglobin 12.2 g / dl , mcv 120 fl , and platelet count 246,000/l . ceruloplasmin and cytochrome oxidase are both copper - containing enzymes essential for heme synthesis 3 . typical morphological findings in peripheral blood and bone marrow aspirate in copper deficiency can mimic myelodysplastic syndrome 1 . bone marrow aspirate in both conditions may show dysplasia in the erythroid precursors such as large size , nuclear multilobulation , and nuclear budding as well as the presence of ring sideroblasts . unlike myelodysplastic syndrome , the bone marrow aspirate in copper deficiency characteristically shows cytoplasmic vacuoles within erythroid and myeloid precursors 2 . furthermore , karyotyping in cases of copper deficiency does not reveal cytogenetic features characteristic of myelodysplastic syndrome . copper is present in common foods including meat , fish , nuts , seeds , and legumes , and therefore deficiency from dietary inadequacy is rare 4 . most copper absorption occurs in the stomach and proximal duodenum , although the more distal small intestine can also absorb copper . problems with copper absorption leading to deficiency should be considered in patients who have history of gastric bypass or gastrectomy , are receiving chronic parenteral nutrition or tube feeding , or on zinc supplementation and develop pancytopenia and/or myeloneuropathy . however , per the mayo clinic the daily recommended intake for adults is 1.52.5 mg / day . copper is available in both intravenous ( cupric chloride ) and tablet forms . in this patient , we hypothesize that the long - term use of jejunal feeding led to a nutritional deficiency of copper and consequent pancytopenia . currently , there is a national shortage of intravenous copper ; therefore we elected to attempt correction of his copper deficiency with higher doses of oral copper . copper deficiency represents a rare but treatable cause of anemia and neutropenia that may be mistaken for myelodysplastic syndrome . therefore , patients with a history of abdominal surgery who present with new found anemia and neutropenia with bone marrow biopsy showing dysplastic changes with no cytogenetic abnormalities should have copper levels checked . recognition of copper deficiency as the cause of these blood abnormalities is critical to appropriately treat this disorder .

key clinical messagecopper deficiency is a rare cause of pancytopenia that may be mistaken for myelodysplastic syndrome . cytoplasmic vacuolization in erythroid and myeloid precursors is found on bone marrow examination . patients with a history of abdominal surgery who present with anemia and neutropenia with dysplastic changes should have copper levels checked .

a 20-year - old boy presented to the emergency department with sudden onset severe left flank pain for a week and no associated vomiting or urinary symptoms . he had a history of per - rectal bleeding 6 months earlier for which he was evaluated elsewhere . an ultrasound done elsewhere the previous day was suggestive of a left perinephric mass , hence a contrast enhanced computed tomography ( ct ) of the abdomen was performed . it revealed an 8 7 cm hyperdense lesion in the perinephric space displacing the kidney anteriorly , with an intensely enhancing lesion adjacent to the hemorrhage , indicating an arterial aneurysm with surrounding hemorrhage [ figure 1 ] . it revealed multiple small left renal arterial aneurysms with no active bleeding [ figure 2a ] . it also revealed similar aneurysms in the superior mesenteric and lumbar arteries [ figure 2b ] . anti - nuclear antibodies , hepatitis b and c serology were negative , and complement levels were normal . a diagnosis of pan was made and he was started on steroids and mycophenolate mofetil , as well as anti - hypertensive medication . he was well at 9 months follow - up and ct angiogram showed no beading of the renal vasculature [ figure 3 ] . computed tomography of the abdomen showing perinephric hematoma ( arrow ) with adjacent intensely enhancing lesion suggestive of an aneurysm ( arrowhead ) abdominal arteriography ( a ) left renal arteriogram showing multiple small aneurysms involving segmental and interlobar branches of renal artery ( arrows ) ( b ) aortogram showing involvement of superior mesenteric and lumbar vasculature ( arrows ) follow - up computed tomography angiogram with no beading of renal vasculature ( arrows ) spontaneous perinephric hemorrhage ( wunderlich syndrome ) is a rare condition with a wide - ranging etiology . zhang et al . in their meta - analysis reported on 165 such cases . of these , 101 ( 62.2% ) were secondary to the rupture of renal tumors , with angiomyolipoma being the most common cause . they reported five cases of bilateral spontaneous hemorrhage and all of these were secondary to pan . pan is a multi - system necrotizing vasculitis that involves small and medium sized vessels . the kidneys are affected in 80% cases with hypertension , proteinuria and ultimately renal functional deterioration being the most common manifestations . it is secondary to the rupture of arterial aneurysms that usually involve the renal artery , and its segmental and interlobar divisions . nephrectomy is associated with a high mortality rate ( 50% ) in the acute setting . angiography with selective embolization allows preservation of renal parenchyma and is now the treatment of choice in cases of active bleeding . a reduction or resolution in aneurysms has been noted following immunosuppressive therapy and is probably due to the decline in inflammation of the vessel wall . per - rectal identified gastrointestinal manifestations , and the need for a surgical consult as an independent predictor of mortality in pan .

we report the case of a young man who presented with spontaneous left perinephric hematoma and per - rectal bleeding . evaluation revealed renal and superior mesenteric arterial aneurysms secondary to polyarteritis nodosa ( pan ) . computed tomography and angiographic findings are presented . the aetiology of spontaneous perinephric hemorrhage along with relevant features of pan are discussed .

one of the few well - understood features of intensive care unit ( icu ) delirium is its association with poor patient outcome . risk factor stratification is essential to the understanding , prevention and treatment of any disorder , and is a cornerstone of scientific endeavor in clinical research . the largest study on icu delirium risk factors to date is published in the previous issue of critical care . delirium in the critical care setting is said to occur in 22% to over 80% of patients . such broad variations in delirium incidence may be partly attributable to differences in predisposing risk factors , which may differ between icus . co - morbidities present prior to icu admission have particularly seldom been considered in icu delirium studies ; several of them are discussed in the paper by van rompaey and colleagues . physicians hold opinions on the risks and benefits of environmental factors ( for example , physical restraints ) affecting the patient once in the icu ; however , little is known about these factors . human interaction , such as the effect visitors may have on delirium , remains unexplored in the critically ill patient . these environmental features , discussed in van rompaey and colleagues ' paper , are important not only because they are inexpensive but also perhaps because they less likely to harm patients than poorly studied pharmacological interventions . the patients described in the study were no longer intubated and thus were perhaps less ill ; in addition , not all centers collected all risk factor data . van rompaey and colleagues nevertheless provide confirmation that several previously identified risk factors remain significant in this multicenter study , challenge other risk factors , and add several risk factors not previously described . alcohol use is not screened for routinely , validated alcohol withdrawal scales remain under - used , and intervention for withdrawal is seldom incorporated into treatment plans . this knowledge is all the more surprising because the relevant studies identify alcohol abuse as a significant risk factor for developing delirium . whether alternatives to routine care ( that is , benzodiazepines titrated to symptoms , rather than antipsychotics ) in these high - risk patients are of any benefit is unknown . age has been considered a risk factor for delirium in non - icu populations and by some icu investigators . when van rompaey and colleagues consider pre - existing cognitive dysfunction , tobacco use and alcohol , age does not appear to confer an additional risk for delirium . if dementia is a risk factor but age is not and studies considering pre - icu admission co - morbidities would indicate this is true prevention or prophylaxis of this common and morbid disorder in the icu should consider different interventions than , say , similar initiatives on the wards . several previously described predisposing factors ( psychoactive drugs , sedatives and opiates ) are revisited and their relative contribution to the risk of icu delirium is tempered by the case mix , and the addition of other variables . the most novel elements in this study , however , are the environmental risks for icu delirium . visible daylight , visitors and not being physically restrained seem to result in a lower incidence of delirium . it is refreshing to know that in the high - tech environment of icus , someone still asks the question ' which inexpensive and nonmorbid approach is useful ? ' combinations of pharmacotherapy and nonpharmacologic interventions such as psychotherapy are used in a broad range of psychiatric disorders . in some situations , mental health is purportedly based on the ' functioning of a high - order nervous system in constant and complex relation with the personal and social environment ' . several of the points raised by the authors ( such as the benefit of company ) offer simple venues for nonpharmacologic intervention . the rise in odd ratios for delirium in physically restrained patients yet make sense when one considers how fearful delirious patients are . other reports have recently corroborated that nonpharmacologic interventions may impact on delirium ; early physiotherapy and ambulation appears to be associated with a decrease in delirium days in a recently conducted randomized , controlled study . all recommended or clinically used antipsychotic drugs have been validated for the treatment of schizophrenia in young populations over short periods of time . the potential for harm with these drugs is all the more worrisome given that their effectiveness and side effect profiles in older or critically ill populations are largely unknown . there is an urgent need for carefully conducted studies differentiating cognitively intact patients from those with subsyndromal delirium or frank delirium , which will integrate stratification based on risk factors such as those described by the authors . only then can we move forward with the disorder , which arguably causes patients , their families and their caregivers the most distress during critical illness .

icu delirium is associated with poor patient outcome . risk factor stratification is essential to the understanding , prevention and treatment of this disorder . alcohol consumption , smoking and prior cognitive impairment appear strongly correlated with delirium risk . several potentially modifiable associations deserve prospective study : these include administration of sedatives and opiates ; multiple catheters ; as well as minimizing physical restraints and enabling visitors .

being an invasive procedure , it is associated with complications that can be managed conservatively or require surgical intervention . in our case report , we discuss a patient who developed surgical emphysema following a therapeutic colonoscopy without any associated abdominal or chest symptoms and was managed conservatively . this case report highlights one of the rare presentations of colonoscopic perforations and provides a feasible management option for clinicians . a 60-year - old female presented to our endoscopy suite after being referred by her general practitioner for investigation of bleeding and passage of mucus per rectum . her past medical history included being diagnosed with ulcerative colitis 26 years ago during her first pregnancy for which she was treated with steroids and has been relatively healthy since then . this colonoscopy showed quiescent colitis confirmed by biopsy . during the present colonoscopy , she had biopsies taken from her rectum , sigmoid , descending and ascending colon given the findings of pseudopolyps . this was not accompanied by shortness of breath , chest pain or abdominal pain . on examination , her oxygen saturation was 99% on room air , with normal respiratory and heart rate . she had marked crepitus over her clavicles , supraclavicular fossa and most of her neck . her erect chest x - ray revealed air under both hemidiaphragms and significant pneumomediastinum ( fig . 1 ) . a computed tomography ( ct ) scan of her chest , abdomen and pelvis was requested . this ct - scan revealed very extensive spreading of surgical emphysema in the retroperitoneum , extending into the mediastinum with small volumes of free intraperitoneal gas ( figs 24 ) . figure 2.ct scan showing an extensive pneumoretroperitoneum . figure 3.ct scan of abdomen showing free intraperitoneal gas compressing the splenic flexure . this consisted of keeping her nil by mouth for 24 h , intravenous fluids and antibiotics . her cervical emphysema gradually resolved and she was discharged 72 h after presentation without any surgical intervention . the rate of perforation following colonoscopy ranges from 0.03 to 0.12% [ 1 , 2 ] . first , it can result from mechanical perforation with the colonoscope and is usually recognized at the time of endoscopy . reported that advanced age , significant comorbidity , obstruction as an indication for colonoscopy and performance of invasive interventions increased the risk of perforation . also reported that older age , male sex , having a polypectomy and a low volume endoscopic service increased the risk of perforation . our patient presented after a therapeutic colonoscopy with cervical emphysema as the initial complaint without any abdominal or chest complaints even though her imaging revealed both pneumoperitoneum and pneumomediastinum . fortunately , she did not exhibit signs of the fourth pneumothorax and did not require a chest drain . this is in stark contrast to previously published case reports where patients developed pneumothoraces requiring chest tube drainage [ 6 , 7 ] . briefly , air in the retroperitoneum can track upwards entering the mediastinum via the hiatus for the aorta and ivc . the air can then continue along fascial planes into the neck resulting in cervical emphysema . additionally , air within the peritoneum can enter the mediastinum via the oesophageal hiatus or can pass through openings in the diaphragm to enter the pleural cavity resulting in pneumothorax . conservative management of perforation following colonoscopy is a safe and acceptable option in patients not exhibiting signs of peritonitis , adequate bowel preparation or silent perforations . additionally , perforations resulting from therapeutic procedures are usually small and are best managed conservatively .

colonoscopy is an invasive procedure used in the detection of colon cancer , inflammatory bowel disease and investigation of bleeding from the rectum . in addition to diagnostic procedures , colonoscopy also has therapeutic indications such as polypectomy and dilation of strictures . we present a case of a patient who presented with cervical emphysema following a therapeutic colonoscopy . the patient had no abdominal or chest pain , shortness of breath and was managed conservatively . perforation following colonoscopy is a rare complication ; however , it is essential that doctors recognize and are aware of the different presentations and management options for this complication .

kaposi 's sarcoma ( ks ) is a spindle - cell malignant low - grade vascular tumor associated with human herpesvirus 8 ( hhv-8 ) . it was first published by the dermatologist moritz kaposi in the journal archiv fr dermatologie und syphilis , in 1872 . in the current literature , four main types of ks are described : ( 1 ) epidemic or aids - related ks , ( 2 ) ks in immunocompromised patients , ( 3 ) classic ks , and ( 4 ) endemic or african ks . chronic lymphocytic leukemia ( cll ) is presented with an increasing number of functionally incompetent mature lymphocytes which are of monoclonal origin , mainly in the peripheral blood , bone marrow , and all throughout the reticuloendothelial system . herein we report an interesting case of ks which was diagnosed during the course of cll as a secondary malignancy . a 64-year - old female case presented with complaints of bluish and indurate lesions all over both lower extremities ( fig . after initial evaluation , biopsy was done from the lesions and ks was diagnosed ( fig . the patient has had a history of cll since the year of 1999 but she has not needed any active drug treatment since 1999 . she did not have any complication such as bacterial infection or hemolytic anemia throughout the disease course . there was no risk factor in her personal history that disposed the patient to aids . quantitative immunoglobulin m and a levels were decreased , whereas immunoglobulin g level was normal . although all tests were negative for systemic involvement of ks , ks lesions have an allover pattern in both extremities , so we planned to administer systemic chemotherapy which consisted of anthracycline , vincristine , and bleomycine . after three cycles of systemic chemotherapy , the lesions had completely disappeared and there was no toxicity of chemotherapeutics . although in the literature , there is a strong relationship between immunosuppression after solid organ transplantation and ks , there is no strong data that shows a relation between cll and ks . patients with cll are prone to the viral , bacterial , and opportunistic infection even without taking any chemotherapeutics . although immunosuppression in cll can dispose the patient to the infection , can it cause tending to the formation of secondary malignancy ? reported that ks is 5 times more common in the cll population than the normal population . in this surveillance analyzed 16,367 patients with cll , so we can not just say that the increased incidence of ks in cll patients is an incidental finding . median latency time between diagnosis of cll and preceding ks was 75 months in this unique study . it is true for all types of ks including aids - related ks , ks in immunocompromised patients , classic ks and endemic or african ks . control of hhv-8 infection is mediated by an antiviral t - cell response to hhv-8 proteins . latent hhv-8 infection of endothelial cells can cause subsequent conversion of endothelial cells into the spindle cells . then the proliferative phase started , and vegf secretion increased from the infected endothelial cells . this activation plays a pivotal role in the development of the final tumor . is this a mechanism that causes ks in cll patient ? we know that there is an imbalance in circulating t - cell subpopulations in cll . so theoretically , latent hhv-8 infection can be reactivated in cll patients and can trigger ks lesions . in conclusion , cll can dispose to the development of ks but the exact mechanism is not explained as much as ks preceding solid organ transplantation . so physicians should be very careful about the probability of ks in newly and progressively developing skin lesions in cll .

cutaneous manifestations can occur in the wide range of internal malignancy . they can occur by metastases or local spread , direct infiltration , or a site of primary malignancy itself . sometimes these manifestations are related with an underlying malignancy but they do not contain malignant cells as paraneoplastic dermatological syndromes . chronic lymphocytic leukemia ( cll ) is the most common leukemia all over the world . cutaneous lesions occur in up to 25% of patients . most commonly seen cutaneous lesions in cll are those of infectious or hemorrhagic origin . skin cancer risk was also increased eightfold in cll when compared with normal population , so cutaneous lesions in cll can be the first manifestation of secondary skin malignancy . herein , we report an interesting case of kaposi 's sarcoma which was diagnosed during the course of cll .

trichoadenoma is a rare benign tumor , with multiple cystic structures closely resembling the infundibular structures of the hair follicle . it presents as a non - specific nodule over the face or buttocks , however , unusual sites such as the neck , upper arm , thigh shoulder , and shaft of the penis may also be affected . ours is a case of a 60-year - old female with a trichoadenoma over the left labia majora , which presented as a slow growing nodular plaque . a 60-year - old married female patient presented to us with asymptomatic gradually increasing skin lesion on vulva since 2 years . examination revealed a firm , nontender , nodular plaque of 3.5 cm 2.5 cm along with a few shiny papules along the right labia majora [ figure 1a ] . the left labia majora , labia mijora , clitoris , and perianal areas were not involved . differential diagnoses considered were fibrosis lymphangioma circumscriptum , giant trichoepithelioma , sarcoidosis , lupus vulgaris , and deep fungal infection . ( c ) post - treatment - 2 months , healing with post - inflammatory hyperpigmentation histopathology from the lesion showed numerous horn cysts present throughout the dermis surrounded by eosinophilic cells [ figure 2a ] . few of the cyst walls were lined by eosinophilic epidermal cells and contained keratin [ figure 2b ] ; thereby leading to a diagnosis of a less known adnexal tumor trichoadenoma . ( a ) 10 view : showing numerous horn cysts present throughout the dermis . ( b ) 40 view : showing cyst walls lined by eosinophilic epidermal cells , containing keratin trichoadenoma of nikolowski is a rare , follicular tumor considered as a neoplastic process by some authors and benign malformation by some others . clinically it presents as a solitary slow growing grayish nodule measuring up to 1.5 cm in diameter , seen over the face ( 57.5% ) and buttocks ( 24.2% ) . other uncommon sites of involvement are the neck , upper arm , thigh , shoulder and shaft of penis . it may also present as a chronic discharging nodule or as an ulcerated growth . rare case reports of trichoadenoma in association with intradermal melanocytic nevus , sebaceous carcinoma , basal cell carcinoma , syringocystadenoma papilliferum have also been recorded . verrucous variants of trichoadenoma have also been reported . though it is a tumor of adulthood , infants as old as 20 months numerous horn cysts are present surrounded by eosinophilic cells . in some instances , a single layer of flattened granular cells is interpolated between the horn cysts and surrounding eosinophilic cells . moreover , the histological similarity of trichoadenoma with trichoepithelioma suggests the development of immature hair structures . however , because the cyst wall consists of epidermoid cells and keratinization may take place with the formation of keratohyalin , it has been suggested that the tumor differentiates largely toward the infundibular portion of the pilosebaceous unit . the keratin profile expression of this tumor supports the theory that it differentiates towards the follicular infundibulum and the follicular bulge region . , we used co2 laser at 9.0 watts with 100 mm hand piece ( 1.0 mm spot size ) in superpulse mode to ablate the growth under local anesthesia [ figure 1b ] . there has been no recurrence so far and the normal vulvar anatomy has been maintained . the trichoadenoma is a mystifying follicular tumor . though it is known to occur on the face and buttocks , newer cases with unusual manifestations are being reported . to the best of our knowledge , a histopathologically proven large trichoadenoma over the vulva has not been reported .

trichoadenoma of nikolowski is a rare , benign , well differentiated , slowly growing tumor of the hair follicle which was first described in 1958 by nikolowski . it usually occurs as a solitary nodular lesion between 3 and 15 mm in diameter . it commonly occurs on the face or the buttocks . herein we report a case of a slowly growing nodular plaque over the vulva of a 60-year - old female , histopathologically proven to be a trichoadenoma . the lesion was completely removed by ablative carbon dioxide laser ( co2 ) .

lymphangiomas are rare benign tumors that are preferentially located in the head and neck in children ( 1 ) . lymphangiomas in the peritoneal cavity are extremely rare occurring most commonly in the mesentery , followed by the omentum , mesocolon , and retroperitoneum . the etiology is unclear , but they are considered primarily to congenital in origin ( 2 ) . we report the case of a young woman with appendicular pain caused by a cystic lymphangioma of the right iliac fossa : a rare but possible differential diagnosis . a 17-year - old female patient presented with a dull abdominal pain in the right iliac fossa and flank of a 1-month duration . an abdominal examination revealed hyperactive bowel sounds and tenderness during palpation in the central abdominal quadrant and right iliac fossa in mc burney point . neoplastic markers ( afp , cea , ca 19 - 9 , ca 15 - 3 , ca125 , tps ) were negative . axial and coronal t2-weighted tse images showed the presence of a fluid - filled cystic lesion in the retrocaecl adipose tissue . an ultrasonographic and ct scan of the abdomen revealed the presence of a cystic mass ( max diameter 60 mm ) in the retrocaecal adipose tissue that was in proximity of the caecum , appendix and ileum . in addition , the ct scan revealed the presence of multiple hypodense formations in the body and tail of pancreas . during the hospital the exam revealed in the retrocaecl adipose tissue the presence of a fluid - filled formation with a diameter of 60 mm that appeared indissociable from the caecum for the presence of a tissue connection ( fig 1 ) . other cystic lesions ( communicating with pancreatic duct ) were present in the whole pancreas and in the right kidney . fluid - filled cystic lymphangioma with polycyclic edge and internal septa ( intraoperative specimen ) the patient underwent exploratory surgery : at laparotomy a yellowish oval formation with a diameter of 6 cm was found arising from the mesentery of terminal ileum with partial adhesion of the appendix . the lesion had a cystic aspect and was fluid - filled ( fig 2 ) . the patient has been followed - up for two years with semestral clinical examination and abdominal eco scan and no recurrence occurred . intra - abdominal mesenteric lymphangioma is an uncommon tumor that usually manifestes in early adulthood . this tumor has an incidence of approximately 1/20,000 and 1/250,000 hospital admissions , and it is more frequent in men than women with a m / f ratio of 3:1 ( 3 ) . in intraperitoneal sites , the small bowel mesentery ( 70% ) is the commonest localization , with 5060% of all cysts located in the ileal mesentery . ( 4 ) the etiology of mesenteric lymphangioma is considered to be congenital , with abnormal embryonic development of the lymphatic system causing sequestration of lymphatic tissue ( 1 ) : infact in our patient also other cystic lesions ( kidney and pancreas ) were found at abdomen ct / mri . radiological investigations are a useful diagnostic tool , but definitive diagnosis is confirmed by histopathology after a complete surgical resection . ( 5 ) at immunochemistry : the lining mesothelial cells are immunoreactive for cytokeratin and negative for factor viiiis . double staining with prox1 and cd31 is the most reliable method for characterizing lymphangioma endothelial cells.(6 )

pain of the right iliac fossa in young girls can relate to several pathologies of the gastrointestinal and genitor - urinary tract . on the other hand , lymphangiomas in the peritoneal cavity are extremely rare . we report the case of a young woman with pain in the right iliac fossa caused by a cystic lymphangioma . a 17-year - old caucasian female patient presented with abdominal pain in the right iliac fossa and flank of a 1-month duration . the patient was apyrexic and laboratory tests were negative . ct / mri of abdomen showed a cystic mass ( max diameter 60 mm ) in the retrocaecl adipose tissue in connection with the caecum and appendix . the patient underwent a total excision of the lesion and appendicectomy . histopathological examination was consistent with the diagnosis of mesenteric lymphangioma . this case shows a rare but possible cause of right iliac fossa pain in young women to be considered in the differential diagnosis .

he narrated that when he was in second grade ( age 7 ) , he and a fellow student were irritating his neighbor in the classroom . the patient mentioned that the point broke off in his thigh and was never extracted . he complained that the site still hurt intermittently and after all this time , he wanted the lump and the reported pencil point to be removed . examination of the anterior left thigh skin revealed a palpable 0.5-cm subdermal / subcutaneous elliptical lump in the skin . there was a tinge of black to blue to it as if it had been tattooed . the patient was explained the indications , risks and benefits of the procedure and he provided consent . a time - out was performed . firm tan dermal - based soft tissue nodule with associated dark graphite precipitate was removed . in the midst of the removed tissue , a 4-mm cone - shaped pencil ( graphite ) two buried absorbable sutures were placed and three simple interrupted 4 - 0 vicryl sutures were used to close the wound . a triple antibiotic ointment was applied and the wound dressed in sterile fashion . the patient was put on cephalexin to prevent secondary infection . the surgical wound healed without complications or significant scarring . the literature reports on hyperpigmented nodules in the skin secondary to a retained pencil core . hatano et al . in 2000 reported on a pencil core granuloma from a pencil tip injury that had occurred about 30 years prior . fukunaga et al . in 2011 summarized 9 previously reported cases of pencil core granulomas , the oldest of which was 58 years old . our case is unique in that we were able to recover the cone - shaped pencil tip and still write with it ( fig . 1 ) . in previous examinations of pencil core granuloma , giant cells and epithelioid cells had been observed . histologic examination in our case similarly revealed foreign body reaction with abundant entrapped dark black pigment and chronic reparative changes , including dense sclerosis and focal granulation tissue formation ( fig . furthermore , one report suggested more rapid growth of lesions and greater degree of tissue injury by colored pencils when compared to non - colored graphite pencils . a number of authors have commented on temporal changes in the granuloma , going from a relatively unchanged appearance to a rapid growth phase , ultimately leading to excision . the decision to explore and excise a pencil core granuloma can be thus based on changes in the skin lesion and/or concern for possible malignancy .

a pencil core with an intact pencil tip was excised from the thigh of a 60-year - old male 53 years after a puncture wound . histologic examination of the excised pencil core and the surrounding tissue revealed a foreign body reaction with abundant entrapped dark black pigment and chronic reparative changes , including dense sclerosis and focal granulation tissue formation .

( sun tzu , the art of war , c.500 bc ) the challenge of conducting high - quality clinical studies in the critically ill population is widely recognised . heterogeneity in patient populations and clinical practice , diagnostic uncertainty , concerns with consent , and the overlapping nature of the presenting illnesses are amongst the inherent difficulties . influential studies in this population therefore increasingly involve multicentre , collaborative efforts using rigorously defined inclusion criteria and outcome measures such projects aim to obviate the aforementioned difficulties and to reduce the influence of individual intensive care units ( icus ) and case - mix variations on the findings . ultimately , the quality of the data justifies the logistical challenge involved . for this reason , the recent article from the newly - formed irish critical care trials group is a welcome announcement of a further such alliance . the data presented describe 1,029 patients admitted to icu services in a 10-week period in 2006 , covering a slight majority of all icu beds in ireland . the data include national specialist centres and university teaching hospitals , as well as regional units . the overall pattern depicts a busy service , with 78% of admissions being emergent in nature , a mean sequential organ failure assessment score of 5.4 , and 70% of patients needing mechanical ventilation . previous research in ireland showing an icu bed occupancy rate of 97% , an unscheduled discharge rate of 23% , and frequent cancellation of elective surgery is consistent with this pattern . the icu readmission rate of 7.5% is perhaps attributable to these service realities as indeed may be the failure to collect data in four of the 14 units that entered the study , including 23% of relevant patients . while accepting that the missing patient data compromise the validity of the findings , the outcomes nonetheless appear compatible with international standards and indeed are broadly indicative of the success of modern intensive care medicine . the outcomes in the subgroups are more striking : over 75% of readmitted patients survived and , of the 93 patients with five or six systems failing on admission , just over one - half survived . compatibility with international outcomes is perhaps most evident from those diagnostic categories where standard , consensus definitions are established . for severe sepsis , the icu mortality of 24% compares with a reported 35% in england , wales and northern ireland for severe sepsis in the first 24 hours . for acute lung injury / acute respiratory distress syndrome , the irish mortality was 32% and the irish critical care trials group has previously shown that this is consistent with modern international studies in the protective ventilation era . although the numbers are relatively small ( n = 289 ) , an icu mortality of 38% for renal failure compares unfavourably with the hospital mortality of 26.3% reported by hoste and colleagues using the same criteria . nonetheless , the study arguably further validates these rifle ( risk - injury - failure - loss - end stage ) descriptors as outcome predictors in acute renal dysfunction . seasonal and regional variations can not be detected as data from a short collection period are pooled together . the use of icu mortality alone as a measure of outcome is not ideal , and more meaningful outcome assessment tools including hospital mortality should be utilised in future projects ; for example , patients who were refused readmission and who might have gone on to die in hospital wards will appear as survivors in such a crude analysis . future scientific publications should also avoid the irritation of new data introduction in the discussion of the findings . the value of the present data will be more fully realised when the irish critical care trials group produces further , hypothesis - testing studies . having taken advice from the anzics critical care trials group , the irish critical care trials group set out to achieve this epidemiological study to provide baseline information for research planning . the data provide insight into disease prevalence ( for example , of acute respiratory distress syndrome ) , and enable planning for the study duration and resource allocation once power analysis has indicated the size of the study population required . the demonstration of the willingness of team members to cooperate and of the capacity of information systems to gather and collate such information is a further key to such studies and collaborations . the authors identify an urgent requirement for audit resources to maintain the ambition shown by this study . participation in the uk 's intensive care national audit and research centre would be one option , opening up the possibility of a uk / irish database . the ability demonstrated by the irish critical care trials group study to establish a research ethos that straddles the relatively contentious border linking the irish republic with the united kingdom is scientifically encouraging . icu : intensive care unit ; rifle : risk - injury - failure - loss - end stage .

quality research , requiring large numbers of participants , in the intensive care unit ( icu ) population requires multicentre collaboration . although logistically challenging , this methodology reduces the influence of individual units and has greater validity and broader relevance to patients and practitioners . the nascent irish critical care trials group opens additional such opportunities . in the accompanying epidemiologic study , the group present data gathered over 10 weeks of 2006 describing 1,029 patients , from 10 irish icus representing over one - half of ireland 's critical care bed capacity . the data depict a busy service , with 78% of admissions being emergent and with a moderately high ( 7% ) readmission rate . while recognising that there were missing data , the outcomes in organ failure and sepsis where international definitions exist and the icu survival rate ( 83% ) were consistent with international standards . the achievement of this planned first epidemiological step lays the foundation for the conduct of prospective scientific studies . these studies might occur in ireland or in cooperation with other audit / scientific groups such as the uk 's intensive care national audit and research centre , the european critical care research network , or others . this brings us a small step closer to the prospect of global , high - volume studies in critical care .

grid laser treatment was performed in both eyes seven months ago . in spite of seven months of follow - up after grid laser with good systemic control , persistent csme was found in both the eyes with central macular thickness ( cmt ) of 311 m and 452 m in the right and left eye respectively [ figs . intravitreal bevacizumab was given in the left eye and the right eye was kept under observation . four weeks after bevacizumab injection , cmt was reduced to 355 m in the left eye with persistent csme observed with maximum macular thickness ( mmt ) of 461 m [ fig . intravitreal dexamethasone implant was injected in the right eye whereas second bevacizumab injection was given in the left eye . at six weeks of follow - up 1e ] whereas cmt and mmt were 306 m and 420 m respectively in the left eye [ fig . persistent macular edema ( me ) in the right eye after 7 months follow - up of grid laser with central macular thickness ( cmt ) 311m . intervention - none . ( b ) persistent me in the left eye after 7 months of grid laser . ( c ) right eye macular sd - oct after 4 weeks of follow - up ( me worsened and cmt increased to 350 m ) . ( d ) left eye macular sd - oct after 4 weeks of 1st intravitreal avastin ( me improved and cmt and mmt reduced to 355 m and 461 m respectively ) . ( e ) right eye macular sd - oct after 6 weeks of implant ( me subsided and cmt became 261m ) . ( f ) left eye macular sd - oct after 6 weeks of 2nd intravitreal bevacizumab . persistent me with cmt 306 m and mmt 420 m ( sd - oct - spectral domain optical coherence tomography ) high intraocular pressure was recorded in the right eye ( 26 mmhg ) and patient was kept on antiglaucoma medications . in the present case , dexamethasone sustained - release implant was able to successfully bring down the cmt to the normal level in refractory csme within six weeks whereas persistent csme was observed even after using two bevacizumab injections in the other eye . as this is a single case with a very short follow - up , it is very difficult to comment on the efficacy of dexamethasone implant per se but comparison with fellow eye ( bevacizumab ) in similar systemic metabolic conditions makes it an interesting observation even at short term . it is important to evaluate the efficacy of dexamethasone sustained - release implant for the long term because that will give a clue towards its cost - effectiveness compared to the existing therapies . it should not be concluded with this report that dexamethasone sustained - release implant is a better therapeutic option than bevacizumab because there was difference in the nature of macular edema in both the eyes i.e. right eye had early edema in comparison to left eye and both the therapies caused reduction in the macular thickness . recent results of resolve and read 2 study have shown successful use of ranibizumab in cases of dme . as far as the frequency of ranibizumab is concerned , the read 2 study has shown reduction in the frequency of ranibizumab injection to 2.9 when it is combined with laser , compared to 5.3 and 4.4 in ranibizumab and laser alone patients respectively during 18 months of follow - up . future comparative clinical studies with a large sample size and long - term follow - up after fda approval of dexamethasone sustained - release implant for dme will be able to provide better results and guidelines .

we report on the significant improvement of central macular thickness in a case of clinically significant macular edema after dexamethasone 0.7 mg sustained - release intravitreal implant ( ozurdex ; allergan , inc , irvine , ca , usa ) . patient presented to us with persistent clinically significant macular edema ( csme ) in both eyes . right eye received dexamethasone implant and left eye received two intravitreal bevacizumab injections 1.25 mg/0.05 ml ( avastin ; genentech inc . , south san francisco , ca , usa ) with an interval of four weeks . after six weeks of follow - up , dexamethasone implant in the right eye showed normal macular thickness whereas persistent macular edema ( me ) was found even after second intravitreal bevacizumab injection in the left eye .

acinar cell carcinoma ( acc ) of the pancreas is a rare tumor that is estimated to account for 12% of all pancreatic exocrine tumors [ 1 , 2 ] . because of the lack of clinical experience with acc , its pathophysiology is not well understood . according to previous reports , acc generally shows expansive growth without invasion into the plexus , portal vein , bile duct and pancreatic duct [ 3 , 4 ] . to our knowledge , no previous reports have described hepatic metastasis from acc primarily in the form of a tumor thrombus . we encountered a very rare case of hepatic metastasis with an unusual clinical presentation : a bile duct tumor thrombus ( bdtt ) from pancreatic acc . a 66-year - old woman had undergone total pancreatectomy due to pancreatic acc ( t3n0m0 , stage iia ) 7 years prior to clinical presentation . computed tomography ( ct ) imaging showed a tumorous lesion measuring 7 cm in length and 1 cm in diameter and extending along the intrahepatic bile duct ( b6 ) . during ct imaging , the lesion showed mild enhancement in the early phase and modest washout in the late phase ( fig . ct arteriography ( cta ) showed early enhancement , which corresponded to the tumorous lesion detected by ct(fig . f - fluorodeoxyglucose positron emission tomography / ct ( fdg - pet / ct ) showed high fdg uptake within the corresponding area ( fig . the tumor was primarily confined to the intrahepatic bile duct ( b6 ) without definitive mass formation in the hepatic parenchyma . tumor markers , including cea , ca19 - 9 and dupan - ii , were all within normal limits . since pathological evaluation of the fine needle biopsy specimen from the bdtt revealed histological features that were compatible with those of the patient 's acc resected 7 years before , the lesion was diagnosed as a hepatic metastasis primarily in the form of a bdtt originating from the prior acc . we initiated chemoradiation therapy ( crt ) , consisting of a total radiation dose of 50 gy and the intravenous administration of gemcitabine ( 1,000 mg / m ) concurrently on days 1 , 8 , and 15 during each 4-week cycle . although a modest effect was observed 13 weeks after the initiation of crt , with a decrease in blood flow and size of the bdtt ( fig . 2a ) , the lesion showed an increase in size 19 weeks after the initiation of crt ( fig . thus , surgical resection , including subsegmentectomy ( s6 ) of the liver and complete removal of the bdtt , was performed . pathological evaluation of the resected specimen confirmed a hepatic metastasis primarily consisting of a bdtt that originated from an acc of the pancreas ( intraductal polypoid growth variant ) . the detailed pathological evaluation of the bdtt and the comparison of this tumor 's characteristics with those of the primary pancreatic lesion that was resected 7 years before are presented in a separate report . although our experience is only anecdotal , the case reported here provides several novel observations in regard to the clinical features associated with pancreatic acc . first , it underscores the need for a long - term follow - up for pancreatic acc cases , even after curative resection . this case involved a liver metastasis that was detected more than 5 years postoperatively , which is unique as hepatic metastases normally occur within a relatively short period following resection of the primary lesion [ 7 , 8 ] . second , aggressive surgical resection combined with crt for hepatic metastasis from pancreatic acc should be considered as a treatment option in selected cases . however , the treatment strategy for metastatic or recurrent acc remains controversial even though various treatment options , including chemotherapy , crt and surgical resection , have been reported [ 7 , 8 , 10 , 11 , 12 ] . reported a case of hepatic metastasis from acc that was successfully treated with an aggressive surgical approach . the advantage of crt prior to surgical resection for hepatic metastasis from acc is unclear . however , a preoperative crt strategy might be beneficial especially in marginally resectable cases because a partial response to crt was observed in the case presented here . third , although multimodal approaches may be useful for selected acc cases [ 10 , 11 , 12 ] , careful repeated re - evaluations during the preoperative treatment period are necessary to improve the chances for a curative resection as demonstrated by our current case , in whom the bdtt showed regrowth after a partial response to crt . further studies with a larger number of patients are required to fully understand the pathophysiology of acc and to evaluate the benefits of multimodal treatment strategies for recurrent acc .

pancreatic acinar cell carcinoma ( acc ) is a rare tumor , and its pathophysiology has not been well understood . treatment strategies for hepatic metastasis originating from acc remain controversial . we report the case of a 66-year - old woman who had undergone total pancreatectomy from acc 7 years prior to clinical presentation . contrast - enhanced computed tomography imaging revealed a tumorous lesion measuring 7 cm in length and 1 cm in diameter and extending along the intrahepatic bile duct ( b6 ) , which showed mild enhancement in the early phase and modest washout in the late phase . this lesion was diagnosed as hepatic metastasis primarily in the form of a bile duct tumor thrombus originating from the prior acc by the pathological evaluation of the fine needle biopsy specimen . the patient underwent preoperative gemcitabine - based chemoradiation therapy followed by subsequent surgical resection , which included subsegmentectomy ( s6 ) of the liver and complete removal of the bile duct tumor thrombus . the patient has had no recurrence during the past 8 months since her last surgery . multimodal treatment including preoperative chemoradiation therapy might be beneficial especially for marginally resectable cases of acc .

extravasation refers to the unintended leakage of a liquid formulation of a drug from blood vessels into surrounding tissues . extravasation of anticancer drugs administered by intravenous infusion constitutes a serious adverse event associated with chemotherapy . in particular , the extravasation of drugs with vesicants , such as anthracyclines , can cause necrosis of the surrounding tissues , often requiring surgical procedures . in addition to careful observation during treatment , a better understanding of the risks of tissue damage triggered by extravasation is essential to ensure the safety of patients who receive cancer chemotherapy . the package insert of doxil ( pegylated - liposomal doxorubicin ) cautions that this preparation is an irritant . however , medical procedures for the management of extravasation reactions and their actual outcomes have not been well documented . we describe here a patient in whom extravasation of a large volume of doxil was successfully treated by an immediate subcutaneous corticosteroid injection . a massive extravasation of a doxil injection ( janssen pharmaceutical , tokyo , japan ) accidently occurred during an intravenous infusion of the drug in a 54-year - old woman with heavily treated metastatic ovarian cancer . the patient was scheduled to receive an intravenous infusion of 75 mg of doxil in a 250 ml 0.9% nacl solution , delivered via a 24-gauge cannula ( surflo i. v. catheters , terumo , tokyo , japan ) placed in the right forearm . before and at the beginning of the infusion , oncology nurses in the outpatient chemotherapy unit confirmed good patency of the vein by checking the blood backflow ; a free flow of the infusion solution was also confirmed . the patient reported a swelling ( 10 x 20 cm ) with mild erythema gradually spreading around the infusion cannula placed in the right forearm ( fig . 1 ) . she experienced no pain , but did feel numbness . in accordance with the institutional guidelines for the management of the extravasation of vesicant drugs , the infusion was terminated immediately , and less than 0.1 ml of the infusion solution and a small amount of blood were slowly aspirated back through the original cannula with the use of a disposable syringe . after removal of the original cannula , 200 mg of corticosteroid ( solu - cortef , pfizer , tokyo , japan ) and 50 mg of lidocaine ( 1% xylocaine , astrazeneca , tokyo , japan ) diluted with 0.9% nacl solution to a total volume of 20 ml were injected subcutaneously via a 27-gauge needle into the extravasation site and surrounding tissues . the affected site was then topically applied with clobetasol propionate ( dermovate ointment , glaxosmithkline , tokyo , japan ) and cooled with an icepack for 30 minutes . immediately after the extravasation of doxil . three days after extravasation the swelling decreased , and erythema of the extravasation site became faint . four weeks after extravasation , the erythema had faded , and a mild epithelial desquamation remained around the extravasation site ( fig . the erythema had resolved , and the wound had healed with no apparent sequela . there were no serious complications such as tissue necrosis or ulceration . this case report provides valuable information on the characteristics and successful management of extravasation reactions caused by doxil , an intravenous anticancer drug used to treat refractory ovarian cancer . in contrast to conventional liposomes , the introduction of this novel delivery system allows pegylated - liposomal doxorubicin to evade detection and destruction by the body s immune system . the drug thus remains in the systemic circulation for a prolonged period , with an elimination half - life of approximately 55 hours . though doxorubicin is thought to be preferentially released from the pegylated - liposomes into tumor tissues , the exact mechanism involved is not fully understood . owing to a modification of the formulation , extravasated doxil is categorized as an irritant , even though doxorubicin per se is a well - known vesicant . although the package insert states that doxil is an irritant , we took all necessary precautions and administered all the treatments currently available in japan because the extravasation was massive , and doxorubicin per se is classified as a vesicant . other antidotes , such as dimethyl sulfoxide ( dmso ) , hyaluronidase , or dexrazoxane , have been used to treat extravasation reactions , but they are not currently approved in japan . although the mechanism by which corticosteroids ameliorate extravasation reactions remains unclear , these drugs have been used to manage extravasation injury caused by various drugs . in our patient , an immediate subcutaneous corticosteroid injection apparently contributed to preventing tissue necrosis and other potentially serious complications .

abstracta massive extravasation of pegylated - liposomal doxorubicin ( doxil ) accidentally occurred , affecting the right forearm of a 54-year - old woman with metastatic ovarian cancer who was receiving an intravenous infusion of the drug . in accordance with the institutional guidelines for vesicant drugs , a corticosteroid preparation was immediately injected subcutaneously into the surrounding tissues . clobetasol propionate and an ice pack were then topically applied to the affected region . there were no serious complications at the extravasation site , such as tissue necrosis or severe pain , and only a transient erythema of the skin and desquamation remained after 2 months .

iatrogenic injury of the lumbar arteries after spinal surgery is a rare complication5 - 9 ) . it may result in the formation of a pseudoaneurysm and consequent acute or delayed retroperitoneal hemorrhage . because pseudoaneurysm develops gradually from a focal arterial wall disruption however , this condition may lead to a sudden and massive retroperitoneal hemorrhage and hypovolemic shock . we recently treated a patient who showed a large retroperitoneal hematoma caused by pseudoaneurysm that arose nine days after posterolateral fusion ( plf ) . a 55-year - old female patient with lower back pain and neurogenic intermittent claudication , who previously underwent l4-l5 plf due to spinal stenosis , was presented to our hospital . she experienced small thalamic infarction 5 years ago , and since then , she started antiplatelet and antihypertensive medications . there was minimal bleeding during the surgery , and post - operative complaints only include mild back pain . however , on the 9th post - operative day , the patient complained of a sudden onset of severe abdominal pain and distension . moreover , the patient was hemodynamically unstable ( 8.5 g / dl hemoglobin concentration ; 90/60 mm hg blood pressure ; and 120/minute pulse rate ) . a late abdominal complication was suspected , and an emergency computed tomography ( ct ) scan of the abdomen with contrast enhancement was performed . the patient was resuscitated with blood transfusion , intravenous fluids for hypovolemic shock and an interventional radiologist was urgently consulted . lumbar spinal angiography showed the delayed hematoma due to rupture of the 2nd lumbar artery pseudoaneurysm ( fig . after we confirmed the successful embolization through the final angiogram , the patient was sent to the ward . since then , the patient 's postoperative progress proceeded normally with recovery of the hemodynamic parameters . two weeks after the embolization , follow - up abdominal ct showed the decreased retroperitoneal hematoma ( fig . a few authors reported iatrogenic lumbar artery pseudoaneurysm as a consequence of spinal surgery4 - 8 ) . moreover , there were some reports about lumbar artery pseudoaneurysm after trauma1 - 3,11 ) . however , there were few reports about lumbar artery pseudoaneurysm after plf . in 1953 , watkins12 ) first introduced posterolateral intertransverse fusion and the indications for the use of this technique have been broadened to include degenerative disc disease and low back pain . nowadays , this technique is supplemented by the provision of a transpedicular fixation device and is widely used in lumbosacral spinal surgery . preparation of the bone fusion bed , which is the most important step in plf , was done through decortications of the transverse process by drilling . in the present case , the fracture of the right l3 transverse process was connected , thus the injury of the lumbar artery may have occurred close to the transverse process during drilling . we believe that this iatrogenic arterial wall injury was responsible for the formation of the lumbar artery pseudoaneurysm with the consequent delayed retroperitoneal hemorrhage . the lumbar arteries of l1 to l4 are small paired vessels that originate from the dorsal aspect of the abdominal aorta at the level of the transverse processes . the lower lumbar arteries can occasionally originate from a common trunk near the midline of the posterior aorta ( fig . these vessels run laterally along the bodies of the lumbar vertebrae and divide into anterior and posterior branches at the medial border of the psoas muscle and anterior of the transverse process1 ) . in the present case , we thought that the lumbar artery was injured at the anterior of the transverse process ( fig . lee et al.4 ) reported the case of a lumbar artery injury combined with a transverse process fracture after a fall . thus if the patient showed retroperitoneal hemorrhage along with a transverse process fracture , the clinician should consider the possibility of a lumbar artery injury . in case of retroperitoneal hemorrhage due to lumbar artery injury , endovascular embolization is effective in controlling hemorrhage as it avoids the risks associated with another anesthesia , surgical incision , and the locating and controlling the bleeding1,7,9,10 ) . in our case , a transcatheter embolization has successfully stopped retroperitoneal hemorrhage due to rupture of lumbar artery pseudoaneurysm . although plf is a safe and widespread surgical technique , major complications are possible if proper attention is not given to the preparation of fusion bed such as drilling or decortication of a transverse process .

a 55-year - old female patient presented with lower back pain and neurogenic intermittent claudication and underwent l3-l4 posterolateral fusion . to prepare the bone fusion bed , the transverse process of l3 and l4 was decorticated with a drill . on the 9th post - operative day , the patient complained of a sudden onset of severe abdominal pain and distension . abdominal computed tomography revealed retroperitoneal hematoma in the right psoas muscle and iatrogenic right l3 transverse process fracture . lumbar spinal angiography showed the delayed hematoma due to rupture of the 2nd lumbar artery pseudoaneurysm and coil embolization was done at the ruptured lumbar artery pseudoaneusyrm . since then , the patient 's postoperative progress proceeded normally with recovery of the hemodynamic parameters .

congenital heart disease encompasses a variety of lesions that may include communication / s between the left and the right side of the heart . such communications may cause volume overload in the short and medium term , possibly resulting in heart failure , or irreversible complications in the long term such as eisenmenger 's syndrome . previously , such defects ( typically atrial ( asd ) and ventricular septal defects ( vsd ) and patent ductus arteriosus ( pda ) ) were closed surgically . over the last decade , a vast variety of devices have been developed to close such defects through the transcatheter route . we report one patient who had self - limiting haemolysis after implantation of an amplatzer perimembranous vsd device . our patient was born via normal vaginal delivery at 38 weeks gestation after an uneventful preganacy . at the routine 6 week visit , she was noted to be failing to thrive and a 3/6 systolic murmur was noted . diuretics were commenced and she throve . at 4 years of age , she was admitted to the paediatric surgical ward with fever , abdominal pain and vomiting . surgical exploration of the abdomen under antibiotic cover was normal , as was an echocardiogram at this time . fever persisted and a repeat echocardiogram showed tricuspid valve endocarditis with a large vegetation that eventually eroded part of the valve resulting in significant regurgitation . the endocarditis was treated with antibiotics and transcatheter closure of the vsd was done successfully using a 14 mm amplatzer perimembranous vsd device at 5 years of age . one week after discharge , she was readmitted due to jaundice and dark coloured urine . haemoglobin was 9.5g / dl , liver function tests were deranged ( bilirubin 80umol / l ; direct bilirubin 12umol / l ; gamma gt 80 u / l ; alt 58 u / l ; alkaline phosphatase 707 u / l , with urobilinogen in the urine . viral studies , inclujding cmv , ebv , hepatitis a and b , were all negative . she was discharged home as after two weeks as her jaundice cleared and her liver function tests improved . haemolysis has been documented after amplatzer device closure of pda,1 asd,2 and vsd.3 the one reported case after vsd closure resulted in transient renal failure.3 hemolysis has also been associated with the use of amplatzer devices to close paravalvar mitral valve leaks after mitral valve replacement.4 conservative treatment is usually sufficient but reintervention of some form may occasionally be necessary , such as intradevice coil deployment in order to completely eliminate any degree of residual left to right shunting.5

over the last few years , a vast variety of devices have been developed to close various septal defects through the transcatheter route . haemolysis has been documented after amplatzer device closure of patent ductus arteriousus , atrial septal defect , and ventricular septal defect . we report one patient with self - limiting haemolysis after implantation of an amplatzer perimembranous vsd device .

ultrasound has been widely used for rapid assessment of critical illness and is regarded as a visual stethoscope . there are few reports on the diagnosis of intracranial lesions by ultrasound in icu patients . here we present a multiple trauma patient who had undergone craniectomy and in whom recurrent intraventricular hemorrhage was rapidly detected by ultrasound . a 29-year - old man was transferred from a local hospital 9 hours after a 5-meter fall . his glascow coma score ( gcs ) was 4 and both pupils were unequal in size . the diagnosis was severe cerebral contusion and subdural hemotoma in the left temporal and parietal lobe , bilateral lung contusion and left clavicle fracture . he received an urgent decompressive craniectomy and evacuation of hematoma , and was then admitted to the emergency icu . in the following days , his condition improved and his gcs reached 8 , although this suddenly decreased to 6 on day 12 . bedside ultrasound was performed instantly and showed acute intraventricular hemorrhage ( ivh ) , which was confirmed by subsequent computed tomography ( ct ) ( figure 1 ) . bedside ultrasound and a head ct scan were arranged immediately , both showing recurrence of ivh ( figure 1 ) . the family refused further treatment and he then became brain dead ( figure 1 ) . computed tomography and ultrasound images of the head in a multiple trauma patient with craniectomy and recurrent intraventricular hemorrhage . ( a , b ) head computed tomography ( ct ) before craniectomy showing severe cerebral contusion and the deformed left lateral ventricle . ( c , d ) head ct on the fi rst day post - craniectomy showing the absence of the skull and the normal left lateral ventricle . ( e ) ultrasound image corresponding to the ct scan shown in ( d ) ; ( f ) the normal doppler pattern of the left middle cerebral artery ( lmca ) . ( g - i ) head ct and ultrasound images after the fi rst intraventricular hemorrhage ( ivh ) : ( g , h ) hematocele in both lateral ventricles ; ( i ) slightly abnormal doppler pattern of the lmca . ( j - l ) head ct and ultrasound images after the second ivh : ( j , k ) both enlarged lateral ventricles with hematocele compared with ( g ) ; ( l ) reverse blood fl ow during the diastolic period of lmca . the absence of the skull may facilitate the ultrasound to detect the brain , and intracranial morphological abnormal signs such as dislocation of the midline , intracerebral hematomas , size of lateral ventricular and hydrocephalus may be clearly displayed on the screen . this rapid and non - invasive method can complement the results of ct and may help to reduce delays in surgical management . caricato and colleagues found that ultrasound in patients with decompressive craniectomy was as effective as ct to evaluate intracerebral disorders . in this multiple trauma patient , ivh happened twice after the craniectomy and was immediately identified by ultrasound . meanwhile , ultrasound may detect the characteristics of cerebral blood flow , which helps to assess intracranial pressure and cerebral perfusion pressure . ct : computed tomography ; gcs : glascow coma score ; ivh : intraventricular hemorrhage .

ultrasound may be a useful tool to evaluate intracranial abnormalities in critically ill patients undergoing decompressive craniectomy . we present a multiple trauma patient who had undergone craniectomy and in whom recurrent intraventricular hemorrhage and patterns of cerebral blood flow were rapidly detected by ultrasound .

hba1c is a retrospective analyte of the carbohydrate metabolism reflecting the mean blood glucose levels during the last 6 to 8 weeks . clinical studies revealed a close relation between late diabetic complications and the concentration of glycated proteins in blood . therefore hba1c determination is widely used to assess the metabolic status and to monitor the medical treatment of diabetic patients . the diabetes control and complications trial ( dcct ) and the united kingdom prospective diabetes study ( ukpds ) used hba1c as one of the main indicators for the quality of diabetes management , . hba1c is meanwhile recommended for screening of diabetes , since hba1c concentrations in blood are not affected by acute metabolic alterations in the patients . standardization of hba1c determinations affords the establishing and implementation of a reference measurement procedure with the highest accuracy possible . at the end of the 80s a certified reference material of hba1c was not available and an high performance liquid chromatography ( hplc ) peak of hba1c analysis developed by goldstein and co - workers became the reference for the calibration of hba1c measurement . the hplc procedure was accepted as a common voluntary consensus standard for the hba1c determination in the dcct- and ukpds - studies . moreover , the procedure was applied as standard protocol for analytical devices for hba1c measurements . the national glycohemoglobin standardization program ( ngsp ) became the basis for the calibration of hba1c measurements in the usa . according to this calibration hba1c values are reported in % of blood haemoglobin concentration . further investigations showed that the separation of hba1c from other proteins by hplc was incomplete and the specificity was not appropriate for international standardization . at the end of the 90s , the international federation of clinical chemistry ( ifcc ) had established a working group to develop an international reference measurement procedure for hba1c traceable to a standard of higher metrological order . the principle of the ifcc reference measurement procedure was the proteolytic treatment of the blood sample with endoproteinase glu - c cleaving glycated or non - glycated hexapeptides from the n - terminal -chains of the haemoglobin . the ratio of the glycated and non - glycated n - terminal hexapeptides of the haemoglobin -chain expressed in percent was defined as the ifcc unit . the decision created considerable confusion , since from now on there were two different calibrations with the same unit on the market . the results between the laboratories of the working group were regularly monitored by intercomparison surveys . the quality of measurements , expressed in terms of inaccuracy and imprecision , was improved during the measurement campaigns . from the collected data of the ifcc working group a master equation was developed to convert values from ifcc units into ngsp units and vice versa . since 2007 , laboratories in europe , australia , and japan calibrated their analytical systems for hba1c according to the ifcc reference measurement procedure and started to report their results in mmol / mol to avoid confusions ; us laboratories remained with ngsp calibration and express hba1c values in ngsp % . the effort of the ifcc working group resulted in a remarkable improvement with respect to the ngsp consensus calibration . however , the key problem of the ifcc reference measurement procedure revealed to be the quality of hba1c and hba0 calibrator preparations and their traceability to the si standard . during the inter - comparison studies a more robust lc - ms reference measurement procedure improving the reproducibility of measurement was established , . in a recent study the traceability of hba1c , measurement results to si units has been achieved by isotope - dilution mass spectrometry , using glycated and non - glycated -n - terminal hexapeptides of defined purity and stated uncertainty of measurement as calibrators and deuterated -n - terminal hexapeptides as internal standards . the traceability is accomplished by hydrolysis of the unlabelled hexapeptide standards and the determination of the amino acid concentration by lc - id - ms calibrated with certified amino acid standards ( figure 1 ( fig . 1 ) ) . in a recent article little et al . claim the development of hba1c standardization from chaos to order during the last 15 years with the ngsp and ifcc standardization efforts and discuss problems arising from the implementation of the ifcc reference system . unbroken chain of comparison using a reference measurement procedure of highest metrological order and , if possible , a reference material of highest metrological level which is traceable to si units . with this iso standard the traceability of measurement the new reference measurement procedure is appropriate to set target values in external quality assessment schemes . in figure 2 ( fig . 2 ) the dispersion of routine values around the different target values is demonstrated in a recent external quality assessment scheme ( eqas ) for hba1c . the three possible target values ( consensus value , ifcc reference measurement procedure value , and idms value ) are given in this scheme . the new calibration using the lc - id - ms procedure leads to slightly higher hba1c levels than the recent calibration according to the ifcc reference measurement procedure . in summary , a new standardization of hba1c measurement has been established at the highest level of accuracy , with stated uncertainty of measurement and with traceability to si units following the ifcc reference measurement protocol using lc - id - ms as procedure of highest metrological order .

glycated haemoglobin ( hba1c ) measurements are used in clinical studies and for the management of diabetic patients . various efforts were made to standardize the hba1c measurements with consensus standards and standards based on a reference measurement procedure with external calibration . according to iso 17511 a standard should meet highest accuracy possible , have a defined uncertainty of measurement and the calibration should be traceable to si units . for hba1c this has been realized using a lc - id - ms procedure based on the existing reference measurement procedure .

nocturnal enuresis ( ne ) is defined as nighttime bedwetting after developmental age when bladder control should have been established . anatomical and functional integrity of noradrenaline , dopamine and serotonin systems is required for continence . iatrogenic ne is an adverse event commonly reported in patients taking several central nervous system agents , in particular antipsychotics , ( risperidone and clozapine ) , ssri and some antiepileptics ( sodium valproate ) . despite enuresis being reported as a very rare adverse event in summary of product characteristics of some antihistamines , no single case report is available to date in literature . we herein report a case of a child presenting with ne after treatment with the antihistamine drug cetirizine . a 6-year - old female child presented with recurrent episodes of rhinitis , bronchitis and bronchospasm since the age of 2 years . her family history was positive for allergic diatheses . the rest of her past medical history was unremarkable with normal developmental milestones having been reached . she never experienced episodes of ne after bladder control was achieved at the age of 3 years . during an episode of persistent dry coughing , which was unresponsive to oral betamethasone 1 mg / day , she was prescribed add - on oral cetirizine 5 mg / day in the evening for a week . on day two months later , she was again treated with oral cetirizine at the same dosage for another episode of cough and bronchospasm . a total score of 9 ( probable ) was obtained on the naranjo causality scale . in particular , a subtle regulation between noradrenaline , serotonin and dopamine pathways is required for appropriate bladder control . functional imbalance of these neurotransmitters within basal ganglion structures appears to be central in the pathophysiology of enuresis and urinary incontinence . the substantial role of dopaminergic circuits is supported by the frequent neurogenic detrusor overactivity and voiding dysfunction in patients with parkinson 's disease or other extrapyramidal dysfunctions , due to nigrostriatal dopamine depletion . noradrenergic and serotoninergic systems exert their effects on continence directly , but also by facilitating or inhibiting dopaminergic pathways , respectively . overall , pharmacological agents depleting or blocking norepinephrine or dopamine cause incontinence or enuresis , while drugs increasing these pathways induce urinary retention [ table 1 ] . experimental studies proved that the histaminergic system directly influences the serotonin transmission in the central nervous system , by the presynaptic inhibition of 5-hydroxytryptamine release . therefore , antihistamine treatment , acting on 5-hydroxytryptamine regulation , could involve a central serotonin disinhibition , leading to an imbalance of the serotonin / dopamine ratio . drugs reported to be associated with enuresis in particular , cetirizine in this patient could have enhanced serotonin pathways originating from raphe nucleus , which exert an inhibitory effect on dopamine release from the mesocortical tract . thus , this disrupted serotonin modulation can lead to a relative dopaminergic depletion , which eventually causes enuresis .

nocturnal enuresis is a common disorder in childhood , but its pathophysiological mechanisms have not been fully elucidated . iatrogenic nocturnal enuresis has been described following treatment with several psychotropic medications . herein , we describe a 6-year - old child who experienced nocturnal enuresis during treatment with the antihistamine cetirizine . drug rechallenge was positive . several neurotransmitters are implicated in the pathogenesis of nocturnal enuresis , including noradrenaline , serotonin and dopamine . antihistamine treatment may provoke functional imbalance of these pathways resulting in incontinence .

schwannoma also called as neurilemmoma is a benign neoplasm of the schwann cells surrounding the nerves . . however , intraosseous schwannoma are extremely rare in jaw bones and represent less than 1% of benign primary tumors of the jaws . a 42-year - old man hailing from dakshina kannada district in karnataka , india , reported to us with a slowly growing swelling on the left side of lower jaw since the previous 4 years . three years ago teeth in the same region became loose and were extracted at a primary health center . the patient had visited the same health center a number of times because of the swelling . medications were prescribed each time but there was no decrease in the size of the swelling . a diffuse swelling was observed on the left lower third of the face extending to the left submandibular region with normal overlying skin . intraoral examination revealed bicortical expansion of the edentulous ridge in the same region with reduced buccal sulcus depth . residual cyst , fibro - osseous lesion , ameloblastoma , dentigerous cyst , and odontogenic keratocyst were listed as the possible differential diagnoses . a mandibular lateral occlusal radiograph was made , which showed buccolingual expansion in the same region and orthopantomogram revealed the presence of large radiolucent area ( 5 7 cm ) on the left side of the mandible involving the body and ramus [ figure 1 ] . a sclerotic rim extending 1 cm below the lower border of the mandible was observed . anteriorly the lesion extended to the distal aspect of premolar and posteriorly to the ramus . a biopsy was performed from the intraoral site , middle of the mandibular residual alveolar ridge and evaluated histopathologically . it revealed the presence of spindle - shaped cells with elongated nuclei arranged in typical circular ( antoni a ) and linear pattern ( antoni b ) along with small cyst - like spaces and eosinophilic verocay bodies [ figure 2 ] . based on these histopathologies , it was diagnosed as schwannoma . since the lesion was massive and only a thin margin of the base was intact , which could lead to pathological fracture , the entire left segment of the mandible was resected barring the condyle . the left side was then reconstructed with bone plate and stabilized with interdental wiring for 3 weeks before being discharged from the hospital . the patient was followed - up for 6 months during which the patient had of localized numbness on the left side of the lower lip . the patient was also advised prosthetic rehabilitation but failed to keep up with further appointments . ( left side ) mandibular lateral occlusal radiograph revealing buccolingual expansion of the body of mandible on the left side . ( right side ) orthopantomogram showing large radiolucent area in the left body and ramus region of mandible photomicrograph ( hematoxylin and eosin stain ; original magnification 100 ) showing cells with elongated spindle - shaped nuclei ( antoni a type ) and also homogenous acellular areas ( verocay bodies ) a recent review of literature revealed that only 44 cases of intrabony schwannomas have been reported . three possible mechanisms of bone involvement by schwannoma have been explained : ( 1 ) a tumor may arise within the bone ; ( 2 ) a tumor may arise within the nutrient canal and produce canal enlargement ; or ( 3 ) a peripheral tumor causing bone enlargement . in our case a combination of the first 2 mechanisms could be the cause of bone involvement . in most of the reported cases the same nerve was involved in our case on the left side of the mandible . radiographically schwannomas present as well - defined unilocular radiolucency with sclerotic borders but sometimes features , such as cortical expansion , root resorption , spotty calcification , and peripheral scalloping can be evident . microscopically spindle - shaped cells in antoni a and antoni b arrangement interspersed with hyalinized verocay bodies are characteristic features . surgical treatment was carried out in our patient , which is the most widely used method to prevent recurrence . intraosseous schwannoma although extremely rare , knowledge of the clinical radiological and histopathological features is extremely important for prompt diagnosis and appropriate management .

schwannoma , a benign nerve sheath tumor is relatively rare in occurrence and even rarer in sites , such as jaw bones . there are only 45 reported cases of intraosseous schwannoma of the jaws reported in the literature . we report a rare case of mandibular schwannoma in a 50-year - old indian male . the clinical features resembled that of a residual cyst , fibro - osseous lesion or an odontogenic tumor / cyst . radiological differential diagnoses of ameloblastoma or odontogenic keratocyst was made based on the findings of the orthopantomogram . the lesion was examined histopathologically and a final diagnosis of schwannoma arising from the inferior alveolar nerve was made . the aim of this report is to add information to the existing sparse literature on intraosseous schwannomas of the jaw .

malrotation of the midgut is an abnormality in embryological development of gastrointestinal tract . by the fourth intrauterine week the gastrointestinal tract is in the form of an endoderm lined tube divided into fore , mid- and hindgut . mid- and hindgut defined by their blood supply the superior and inferior mesenteric arteries respectively . by the fifth week of life the midgut begins a process of rapid enlargement , physiological herniation and rotation . with rapid expansion of liver and kidneys , expansion of the midgut intestinal loop can not be contained within the abdominal cavity ; this results in temporary physiological midgut herniation through the umbilical cord with superior mesenteric artery forming the axis . this process forms c of the duodenum and places it behind the superior mesenteric vessels . hernial reduction occurs by week 10 with the jejunum reducing first and lying to the left and subsequent distal portions lying progressively to the right . the ceacum descends from position in the right upper quadrant forming the descending colon with its mesentery gradually disappearing . a 17-year - old male was seen in the emergency department with 10-year history of abdominal colic which is relieved by vomiting , along with dehydration . there was no history of jaundice , fever , steatorrhea , or bleeding per rectum . the patient had been treated with proton pump inhibitors , prokinetic agents by general practitioners , without any relief . his ultrasound examination and standing x - ray of abdomen physical examination was normal except minimal abdominal tenderness in epigastric , right hypochondriac region along with mild dehydration . his liver function tests , renal function tests , amylase , hemogram , and urinalysis were normal . duodenum appeared connected to small intestine seen on right side giving a whirlpool appearance due to rotation of gut around the superior mesenteric artery . superior mesenteric vein was seen on left of superior mesenteric artery [ figures 1a and b ] . stomach and duodenum appear connected to small intestine seen on the right side giving a whirlpool appearance due to rotation of gut around the superior mesenteric artery . superior mesenteric vein is seen on the left of superior mesenteric artery surgical referral was made ; he was treated with four - port laparoscopic ladd 's procedure . the peritoneum to the right of the ascending colon and caecum was incised and the anteriorly situated bands were stripped to free the duodenum . he was discharged within 2 days eating a normal diet and made a good postoperative recovery . at 3 months he was gaining weight and had no further vomiting . midgut mal and nonrotation refers to failure in counter clockwise rotation of the midgut which results in misplacement of the duodeno jejunal junction to the right of the midline ; in addition the small bowel mesentery has narrow vertical posterior attachment which is prone to volvulus . other anatomical abnormalities include peritoneal ( ladd 's ) bands running from the right colon to the lateral abdominal wall and an extensively mobile ceacum that fails to descend . acute presentation is with volvulus of midgut or ileoceacum occurring most frequently in neonate with likelihood decreasing with age . in most of the reported cases of this presentation , patients present with bilious vomiting in the first month of life because of duodenal obstruction or a volvulus . pathophysiology of these chronic symptoms may relate to the compressive effects of peritoneal bands running from ceacum and ascending colon to the right lateral wall . the short mesentery allows the small bowel to twist around the narrowed sma pedicle to create a distinctive whirlpool appearance . surgical management of intestinal malrotation was first described by ladd in 1936 and remains mainstay of management today . it involves reduction of vovulus if present , division of abnormal peritoneal bands ( duodeno colic , dodenojejunal - ileocolic ) , and placement of the small bowel to the right of the abdomen and ceacum to the left . increasingly laparoscopic ladd 's procedures are being performed and have been shown to be effective where there is no acute volvulus.[79 ] this minimally invasive approach allows for earlier oral intake and discharge from hospital .

abnormalities in midgut rotation occur during the physiological herniation of midgut between the 5th and 10th week of gestation . the most significant abnormality is narrow small bowel mesentery which is prone to volvulus . this occurs most frequently in the neonatal period , less commonly midgut malrotation presents in adulthood with either acute volvulus or chronic abdominal symptoms . it is the latter group that represents a diagnostic challenge . we report a case of a 17-year - old male patient who presented with 10-year history of nonspecific gastro - intestinal symptoms . after extensive investigation the patient was diagnosed with midgut malrotation following computed tomography of abdomen . the patient was treated with a laparoscopic ladd 's procedure and at 3 months he was gaining weight and had stopped vomiting . a laparoscopic ladd 's procedure is an acceptable alternative to the open technique in treating symptomatic malrotation in adults . midgut malrotation is a rare congenital anomaly which may present as chronic abdominal pain . abdominal ct is helpful for diagnosis .

autism spectrum disorders ( asd ) , according to the diagnostic and statistical manual of mental disorders 5 edition ( dsm-5 ) , are neurodevelopmental pathologies impairing both social competencies and patterns of behavior . until today it is probably also for this reason that in the last decades complementary and alternative medicine ( cam ) has spread widely among families of asd children , although apart from melatonin , there is no scientific evidence of their effectiveness . one of the most commonly used cam practices in asd children is the supplementation of omega-3 that are essential fatty acids present in the following foods : fish , seafood , meat , eggs , vegetable oils , cereal - based products . we describe the case of a child with asd who seemed to respond to omega-3 supplementation in a relevant and lasting manner . family history was positive for asd in two first - degree paternal cousins ; previous language delay in the paternal line . the boy came to our observation for the first time at the age of 2 years 10 months . already at that time , there were qualitative anomalies in terms of intersubjectivity ( limited exchange capacity ) , communication ( lacking both the verbal and nonverbal ) , and interests ( perseverative activities such as hitting objects against each other ) , so as to justify a diagnosis of asd , according to the dsm-5 criteria . autism diagnostic observation schedule , module 1 , showed an overall result above the cutoff for autism , with a score above the cutoff for autism in language and communication , as well as in reciprocal social interaction . the childhood autism rating scale 2 edition standard version ( cars2-st ) showed results consistent with mild to moderate symptoms of asd ( see later ) . after obtaining an informed consent , at the age of 4 years 11 months an oral treatment with omega-3 at the dosage of 1 g a day was started . since then , significant improvements were observed in the clinical picture . the child appeared more active and responsive to solicitations , verbal ( comprehension and expression ) and nonverbal communication skills increased , personal autonomy improved , oppositional behaviors as well as hyperactivity and inattention decreased . these improvements have been quantified through cars2-st [ figure 1 ] : while the total score was 36.5 ( cutoff for the presence of mild to moderate symptoms of asd = 30 ) before omega-3 treatment start , it decreased to 33 at the most recent assessment , 22 months after omega-3 treatment start . according to cars2-st , improvement of scores involved the following items : relating to people ; visual and listening response ; verbal and nonverbal communication ; activity level ; and general impression [ figure 1 ] . therefore , omega-3 supplementation seemed to have a favorable impact on the quality of life of the child . it should be noted that the applied behavior analysis intervention , already underway when the treatment with omega-3 was started , was not subsequently modified . various attempts to suspend the omega-3 supplementation during the 22 months of follow - up failed because of significant symptom worsening ( restlessness , agitation , decrease of responsiveness to teaching ) , which then disappeared after the resumption of the treatment . there were no side effects attributable to omega-3 . at the time of the most recent evaluation , at the age of 6 years and 9 months profile of the scores of the 15 items included in the childhood autism rating scale , 2 edition standard version , respectively before ( black horizontal bars ) and 22 months after ( white horizontal bars ) the start of omega-3 treatment . note that score 1 corresponds to the normal behavior , while score 4 corresponds to the most atypical behavior the interesting hypothesis of van elst et al . seems to be a good theoretical basis for treatment with omega-3 supplementation in children with asd . they speculated that the increase in asd prevalence during the last decades is related to dietary modifications of fatty acid composition , characterized by a higher ratio omega-6/omega-3 . in particular , the omega-3 deficit , especially in the early stages of life , may cause changes of myelination , neurogenesis , synaptogenesis , neurotransmitter turnover , brain connectivity , cellular differentiation and development , inflammatory reactions , cognitive functioning , and behavior . but so far , based on the results of randomized clinical trials , evidence - based medicine negates the effectiveness of omega-3 in asd children in particular on symptoms such as deficits in social interaction and communication , hyperactivity , stereotypies . the empirical value of a case report like ours is obviously not comparable to that of a randomized clinical trial , primarily due to the risk of mistakenly considering a placebo effect as a real effect of the drug . we were aware of this possible bias when describing our case nevertheless , considering anecdotal experiences , including that of our patient , and nonrandomized trials , the presence of a subgroup of asd patients who are really responders to omega-3 can not be excluded . these responders might not appear when evaluating the omega-3 effects in a sample taken as a whole . further , considering the high heterogeneity of asd phenotypes and etiologies , it seems to be very unlikely that a given treatment produces the same results in all affected individuals . we believe that the question about the effectiveness of omega-3 in asd is still open and that it requires carrying out studies that check for the possible presence of a subgroup of asd individuals responders to this treatment . should the actual presence of these responders be determined , it would surely be very important to identify what the characteristics are that distinguish them from nonresponders .

one of the most commonly used complementary and alternative practices in children with autism spectrum disorder ( asd ) is the supplementation of omega-3 . we describe the case of a child with asd who seemed to respond to omega-3 supplementation in a relevant and lasting manner . so far , based on the results of randomized clinical trials , evidence - based medicine negates the effectiveness of omega-3 in asd children . nevertheless , considering anecdotal experiences , including that of our patient , and nonrandomized trials , the presence of a subgroup of asd patients who are really responders to omega-3 can not be excluded . these responders might not appear when evaluating the omega-3 effects in a sample taken as a whole . studies that check for the possible presence of this subgroup of asd individuals responders to omega-3 are necessary .

spinal meningocele is associated with spina bifida cysts and may occur at any age . however , intrapelvic meningocele is rare and , without spinal compression , this type of meningocele does not cause any discomfort . for this reason , intrapelvic meningocele has not attracted much attention for a long time . here , we report a case of an asymptomatic intrapelvic meningocele found incidentally during a sigmoidectomy due to megacolon in a 30-year old male patient . a 30-year - old male , admitted because of constipation , was scheduled to undergo sigmoidectomy . preoperative magnetic resonance ( mr ) scan indicated that a cyst was in front of the third sacral vertebrae ( s3 ) ( fig . after sigmoidectomy , this cyst was resected . however , on the first postoperative day , 150 ml of amber liquid was drained from pelvic drainage tube . over the following several days 1c and 1d ) . because the patient had just undergone sigmoidectomy , conservative treatment consisting of three main steps was considered : drainage , anti - infection therapy ( cefpiramide combined with tinidazole ) and posturing by raising the position of the lumbosacral region to reduce liquorrhoea . at the beginning , we drained the cerebrospinal fluid ( csf ) via a pelvic drainage tube . however , because the volume still exceeded 150 ml / d , we placed another drainage tube into the subarachnoid space . on the first day , approximately 300 ml csf drained from the subarachnoid tube , and 50 ml drained from the pelvic cavity . over the following three days , the volume of liquid from the pelvic cavity began to reduce generally . the subarachnoid drainage tube was removed on the fourth day and there was no liquorrhoea over the next two days . mr scan reveals the presence of a cyst 3 cm in diameter ( arrow ) in front of s3 in a 30-year - old male patient ( a : coronal section ; b : median sagittal section ) . histological examination ( c : 40 ; d : 100 ) showed that the cyst was mixed with nerve collagen tissue ( h&e ) . intrapelvic meningocele is defined as a csf - filled sac within the spinal meningeal wall which protrudes into the intrapelvic cavity through an enlarged intervertebral foramen or bone defect . acquired meningocele is relatively common as a laminectomy complication , while congenital meningocele is commonly associated with neurofibromatosis , . the clinical manifestation of an intrapelvic meningocele is closely related to its size and relationship with surrounding structures . it may include back pain , urinary and fecal incontinence , and lower limbs weakness or paralysis . a small lesion may be asymptomatic and discovered incidentally , as it was in our case . for small- and medium - sized lesions , the most common surgical management is resection of the meningocele and repair of the dural defect through laminectomy , . in this case , surgery was performed with removal of the meningocele pouches and shunting of the cyst to the subarachnoid region . however , liquorrhoea occurred after the operation . although liquorrhoea is a common occurance with cystectomy , it may result in infection of the central nervous system . specific posture and drainage in combination with anti - infection therapy were performed instead of surgery . this conservative method can also treat liquorrhoea effectively and at the same time help patients avoid a second operation .

asymptomatic intrapelvic meningocele is rare . here , we report the case of a 30-year - old chinese man who underwent sigmoidectomy due to megacolon . during the operation , an intrapelvic cyst was found and resected . meningocele was confirmed by histological examination . the patient recovered well postoperatively with the exception of liquorrhoea . conservative therapy was initiated , including draining , anti - infection and specific posture maintenance . during the following week , liquorrhoea was generally relieved and the patient was discharged . this is the first known report of liquorrhoea associated with intrapelvic meningocele resection successfully treated by conservative therapy . our case indicates that conservative treatment may be considered for similar cases so that a second surgery is avoided .

a 52-year - old healthy man presented with intermittent ocular pain of indistinct duration in his right eye . his best - corrected visual acuity ( bcva ) was 20/20 , and the intraocular pressure in the right eye was 10 mmhg . slit - lamp and gonioscopic examination of the right eye showed an irregularly - shaped pigmented mass at the temporal iris root ( fig . ultrasound biomicroscopy showed a ciliary body mass with extension into the iris root and medium internal reflection ( fig . the basal diameter of the mass was 1.5 mm and the height was 1.3 mm . magnetic resonance imaging of the head and orbit was unremarkable , and positron emission tomography of the ciliary body identified no significant fluorodeoxyglucose uptake in the orbit or the imaged body to suggest malignancy . 2b ) , and the tumor was positive for melanocytic markers ( melan a and hmb-45 ) . a macrophage marker ( cd 68 ) confirmed the presence of melanophages in a scattered pattern . the proliferative activity , estimated by the ki-67 labeling index , was less than 1.0% , implying a benign tumor . histopathologic features were consistent with melanocytoma of the ciliary body with extension into the iris root . slit - lamp photography taken on postoperative day 7 showed a small iridectomy site with good overall cosmesis ( fig . the patient 's bcva remained 20/20 with an intraocular pressure of 12 mmhg . during one year of follow - up , no signs of tumor recurrence were seen , and the patient reported resolution of the intermittent ocular pain in the involved eye . melanocytoma of the ciliary body is typically identified at a late stage , as its location behind the iris eludes early detection with routine ophthalmic examination . only after ciliary body melanocytomas extend into the iris root can they be visualized on anterior segment examination . in a report of ten patients with ciliary body melanocytomas , including a review of 30 additional cases , 85% of patients displayed extension into the iris root or the trabecular meshwork , but only 12% had elevated intraocular pressure . our patient exhibited extension of his ciliary - body melanocytoma into the iris root , but his preoperative intraocular pressure was within the normal range . in this series of ten patients , three were symptomatic , one of which reported soreness of the involved eye . likewise , our patient presented with intermittent ocular pain , which could have been related to the presence of tumor in the ciliary body . the differential diagnosis of a pigmented tumor of the ciliary body includes adenoma , adenocarcinoma , medulloepithelioma and , most importantly , malignant melanoma . although melanocytoma can usually be distinguished histopathologically , it can be difficult to clinically differentiate before surgery , especially from a malignant melanoma . advised surgical removal rather than observation for management of pigmented lesions of the ciliary body due to both the difficulty in clinically distinguishing a melanocytoma from a malignant melanoma and the frequent occurrence of necrosis ( 36% ) seen in ciliary body melanocytomas . if serious intraocular complications are not present , such as massive necrosis of a large tumor , severe destruction of chamber angle structures , or massive hemorrhage , and if technically feasible , iridocyclectomy under lamellar scleral flap seems an apt choice for the surgical treatment of ciliary body tumors . in summary , melanocytoma of the ciliary body is a rare tumor that can invade chamber angle structures and present as a pigmented mass at the iris root . we present a case of ciliary body melanocytoma which presented as an iris mass with intermittent pain that was successfully managed with an iridocyclectomy .

we report a case of ciliary body melanocytoma in a korean patient , which presented as an intermittently painful pigmented iris mass and was successfully managed by iridocyclectomy . a 52-year - old healthy man presented with an irregularly - shaped and heavily - pigmented mass at the iris root of his right eye . visual acuity of the right eye was 20/20 with normal intraocular pressure . ultrasound biomicroscopy showed a 1.5x1.3-mm ciliary - body mass with extension into the iris root . iridocyclectomy with scleral resection under a lamellar scleral flap was performed , and the histopathologic features of the resected tissue were consistent with melanocytoma of the ciliary body . the patient 's visual acuity remained 20/20 with good postoperative cosmesis . during one year of follow - up , no signs of tumor recurrence were seen , and the patient reported resolution of the intermittent ocular pain in the involved eye .

nonunion of a fracture can pose a significant challenge in terms of skeletal stability , soft tissue biomechanics and potential limb loss . they most commonly occur as a consequence of infection , mal - alignment , metabolic and endocrine abnormalities . however , in otherwise healthy young individuals , such etiologies are rare and alternative pathological factors should be considered . herein , we present the first reported and successfully treated case of tibal fracture nonunion as the result of posterior tibial artery ( pta ) pseudoaneurysm . a 20-year - old man with no past medical history presented to the emergency department with non - displaced and closed transverse fracture of the left mid - tibial shaft and an intact fibula induced by a tackle during a football match . on examination , he had no neurovascular deficit and all crural pulses were palpable . his fracture was treated conservatively with a non - weight bearing above knee plaster cast later replaced by a below knee cast for a total period of 6 months . upon examination during his attendance to the facture clinic , the subsequent magnetic resonance imagining ( mri ) confirmed a nonunion of the fracture along with an ovoid 2.7 cm posterior compartment vascularized mass causing bowing of the interosseous septum with no evidence of avascularity of the bony margins ( fig . 1 ) . an endovascular approach was adopted , and the pseudoaneurysm was injected with 2 ml of thrombin , filled with multiple coils and deployment of an angiography balloon ( in the sac ) ( 4 mm 6 cm ) for 7 min ( fig . 2 ) . this resulted in complete cessation of the pseudoaneurysm with normal flow in the pta ( fig . follow - up x - ray ( 6 months ) demonstrated complete union of the fracture site with no associated complications . figure 1:mri images demonstrating the site , size and the origin of the pta pseudoaneurysm . figure 2:angiographic image , demonstrating the site of the nonunion , coil insertion to the pseudoaneurysm and extravasated contrast . mri images demonstrating the site , size and the origin of the pta pseudoaneurysm . angiographic image , demonstrating the site of the nonunion , coil insertion to the pseudoaneurysm and extravasated contrast . pseudoaneurysms of the pta are rare , and they mainly occur following sudden acceleration and/or deceleration type injury , open fractures and , in few cases , can be iatrogenic ( manipulation of fracture , surgery and ilizarov k - wire ) [ 2 , 3 ] . currently , no report in the literature suggests a single isolated and close fracture of tibial bone to be responsible for pseudoaneurysms of pta . they are mainly asymptomatic and present as a pulsatile lump but in their deeper anatomical course could result in tibial nerve compression causing paresthesia , neuralgia and weakness of the foot and toe muscles . in 39% of reported series , they present with signs and symptoms of lower limb ischemia following pseudoaneurysms thrombosis and/or distal embolization [ 4 , 5 ] . , the patient did not exhibit any neurovascular deficit , and it was an incidental finding following investigations for the tibial fracture nonunion . furthermore , fracture nonunion in a healthy young individual without any risk factor is rare and should raise suspicions about alternative and infrequent pathologies . the management of pseudoaneurysms of pta is varied , and they depend on the individual 's symptomatology and the subsequent course of the pseudoaneurysm . currently , there is no consensus on their overall management ; therefore , indications for their open versus endovascular repair are subjected to various debates . open repair ( ligation and/or excision and interposition vein grafting ) in deep anatomical areas may subject the patient to extensive dissection , longer operative time and higher incidences of morbidity ( motor and sensory nerve damage , prolonged recovery , deep vein thrombosis ) . endovascular repair is another acceptable option , but this approach has its own limitations . the deployment of any type stent for exclusion of pseudoaneurysm from the main artery in the infra popliteal region is known to possess low patency and thrombotic incidences . the use of thrombin , coils and glue remains another alternative , but this mainly depends on the pseudoaneurysm neck size , as wider necks fail to contain the above and can result in failure of the procedure and thrombosis of the remaining artery . overall , the approach should be tailored according to the patient 's arterial status and features of the pseudoaneurysm . to the best of our knowledge , deployment of angiography balloon ( as an occlusive technique ) in addition to thrombin and coil for the exclusion of pta pseudoaneurysm has not been reported in the literature . the use of this novel technique could be beneficial in exclusion of wider neck pseudoaneurysms and limitations of the damage to the main vessel as reported in the current case report . in the reported case , the individual did not exhibit any signs and symptoms of pseudoaneurysms and similarly did not raise any suspicion of such pathology till nonunion was detected . once suspected , the best investigative modality is computed tomography angiography ( cta ) followed by magnetic resonance imaging ( mri ) . both of these modalities have sensitivity and specificity of more than 90% for detection of isolated entities . in conclusion , nonunion of fractures in young healthy individuals should raise the suspicion of pseudoaneurysms , despite the absence of clinical signs and symptoms of vascular disruptions . clinicians should have low threshold for cta and/or mri in such circumstances , and the surgical method of repair ( open versus endovascular ) should be in accordance with the patient 's best interest . written informed consent was obtained from the patient for publication of this case report and accompanying images .

pseudoaneurysm of the posterior tibial artery ( pta ) is uncommon , and they mainly occur following high - velocity trauma , open fractures and can be iatrogenic in nature . to the best of our knowledge , this is the first reported and successfully treated case of pta pseudoaneurysm identified as a consequence of tibia fracture nonunion in an otherwise healthy young individual 6 months following the original incident with a novel intraoperative technique .

as in most other developing nations , critical care medicine as a specialty has developed very slowly and only recently in india . the coronary care units were developed in the early to mid-1970s . perhaps the main pioneer of the field of critical care in india was farokh e udwadia , a brilliant physician with international training in pulmonology . in the mid 1970s , udwadia developed the first respiratory care units in the country in two hospitals in mumbai a community hospital and a private one . the most major achievement of these units was not only to bring down the mortality of tetanus , but also to open the eyes of society to the need for critical care services . organized critical care training or programmes did not materialize , however , and it was left to individual interested trainees to go abroad and receive training . although the speciality was being practiced in isolated foci of hospital practices , the first few ripples in this field were created by consultants returning to india after training abroad in the united kingdom , in the united states , and in australia . the initial centres of such activity were mumbai , pune and chennai , and they still remain the centres of academic creativity and administrative ability . these few enthusiastic , trained consultants came together in 1992 to discuss critical care on a common platform , and they formed the national indian society of critical care medicine ( isccm ) . the society had its teething troubles and has now established itself very firmly as a representative body of critical care consultants in india . the current practice of critical care in india is a matter of as much diversity as the country itself . there are three types of hospitals in india that are delivering patient care in india . community hospitals are mostly run by the government and essentially result in no cost to the patients . critical care is a branch that involves a lot of technology and therefore is dependent on finances . hence , there have been limitations to the growth of this branch in community hospitals . additionally , there are more than 1000 district hospitals . only a small proportion ( < 10% ) of all these hospitals , however , patients are levied a charge for these services that is proportional to their income ; there are also a small percentage of beds that are provided for free . as per the current estimation , icus in private tertiary care hospitals are usually very well equipped and thus form the most major contributor to the critical care facilities in the country , albeit at a higher cost to the patient . finally , an interesting segment of health care facilities in india consists of small hospitals or nursing homes . modestly equipped , and managed mostly by medical professionals themselves , these are realities representing the vast middle and lower classes , and they contribute about 40% of available beds for the country . the need and the viability of facilities for critical care are being acknowledged by this segment , and currently the facilities are on the upswing . the certificate course in critical care , the first organized training activity in critical care medicine , was started 4 years ago by the isccm and has been evolving well . a number of hospitals have developed training modules , and more students are coming out of this training programme regularly . the isccm has also been very active in interacting with various medical councils in india . as a result , the postdoctoral fellowship in critical care medicine conducted by the national board of examinations has recently been announced . with this , the first steps for training in critical care on a national level curriculum are now being taken . the training of nurses , technicians , and therapists has begun in some isolated foci but has not evolved into a meaningful training activity . the patterns of medical problems seen in indian icus are dissimilar to those seen elsewhere . a number of tropical infections such as malaria , leptospirosis , tuberculosis , salmonellosis , etc . form a significant proportion of the patients . playing its part in the development of this new speciality , the isccm has taken the lead in the development of a number of other related issues . the cpr training project and the development of an independent , dedicated organization like the resuscitation council of india has been felt by many who have been working in this field . along with other like - minded societies , development of guidelines for the working of icus has been another important issue that the isccm has taken up . the guidelines are currently being formulated . for a country that has it own set of problems the indian journal of critical care medicine is the official journal of the society and is the only mouthpiece of the organization . the society has redesigned and activated its website ( ) , so one can now have access to all the latest news on isccm activities the annual national conference in critical care , conducted by the isccm , has been the high point of academic activities in this field . held in different important cities , this event has been attracting not only the who - is - who in critical care in india , but also many international stalwarts over the past 8 years . good quality original work has now started emerging , and is being accepted for publication by the prestigious international journals . at the recently held world congress , a multicentre study on scoring systems was presented on behalf of the isccm , and indian icus are now being included in the upcoming international simplified acute physiology score ( saps)iii study . for the first time , india will be represented on the executive committee of the world federation of societies of intensive and critical care medicine . one hopes that all the efforts will lead to a humane , scientific and meaningful service for the multitude of critically ill patients . the ' icus worldwide ' series is created in collaboration with the world federation of societies of intensive and critical care medicine ( wfsiccm ) .

critical care practices in india have evolved significantly over the past decade . critical care initially began as a service in major hospitals , but with the formation of the indian society of critical care medicine the development of this speciality has been very rapid . regular conferences , updates , continuing medical education programmes and workshops have emerged , and postdoctoral training programmes have been developed . scientific publications have begun to appear and in spite of the diverse problems and standards , meaningful speciality - related activities have begun . future challenges include the development of guidelines , the consolidation of training activities and research on the outcome of critical tropical problems .

sebaceous carcinoma , first described by allaire in 1891 accounts for less than 1% of all cutaneous malignancies . it is a rare but aggressive malignant neoplasm that arises from sebaceous glands with a tendency for both local recurrence and distant metastases . we report a case of an extraocular type of sebaceous carcinoma of the scalp with no distant metastases . a 65-year elderly man presented with a large asymptomatic swelling over the vertex of the scalp , which gradually progressed over a period of twelve months . examination showed a large swelling of 15 12 5 cm size situated over the vertex of the scalp extending to the occipital and tempero - parietal regions.the tumor showed focal ulceration with yellowish malodorous seropurulent discharge [ figure 1 ] . clinically , a differential diagnosis of cylindroma , angiosarcoma , deep fungal infections , nocardiosis , mycobacterial or atypical mycobacterial infections were considered . a large multi- lobulated swelling with ill - defined margins and irregular surface showing focal ulceration and yellow sero - purulent discharge the culture of the seropurulent discharge revealed staphylococcus aureus and acinetobacter baumanni suggestive of secondary bacterial infection . afb stain and culture for both mycobacteria and atypical mycobacteria and fungal culture were negative . ct scan of the brain showed only age related cerebral atrophy , and there was no evidence of extension of the tumour or metastatic deposits . histopathology revealed an infiltrating neoplasm in the dermis composed of cells arranged in lobules separated by fibrovascular stroma . [ figure 2 ] neoplastic cells were round having moderate amount of eosinophilic to vacuolated cytoplasm and large nuclei with prominent eosinophilic nucleoli . dermis showing a neoplasm composed of cells arranged in lobules separated by fibrovascular stroma ( h and e , 4 ) neoplastic cells having moderate amount of eosinophilic to vacuolated cytoplasm and large nuclei with prominent eosinophilic nucleoli and increased mitoses ( h and e , 40 ) immunohistochemical marker study showed diffuse strong positivity for cytokeratin and focal positivity for epithelial membrane antigen ( ema ) [ figure 4 ] . sebaceous carcinoma is a rare malignant neoplasm , often occurs in adults with a slight male preponderance this malignancy can occur as peri - ocular and extraocular types and the former variant contributes to 75% of sebaceous neoplasms . the upper eyelid is affected two to three times more often than the lower eyelid due to greater number of meibomian glands extraocular sebaceous carcinoma which constitutes 25% of the sebaceous neoplasms has been reported more commonly on the head and neck areas followed by trunk , salivary glands , genitalia , breast , ear canal , and the intra - oral cavity . the most frequent clinical presentation is a painless subcutaneous nodule but it can also present as pedunculated lesions , irregular mass or diffuse thickening of the skin . this protean appearance frequently masquerades as other benign tumors or inflammatory conditions thereby leading to delay in diagnosis , inappropriate treatment , increased morbidity , and mortality . the morphological hall mark of sebaceous differentiation is the detection of sebaceous cells and demonstration of fat in vacuolated tumor cells . special stains such as oil red o may be helpful in confirming the presence of fat , but requires frozen section . other differential diagnosis includes basal cell carcinoma with sebaceous differentiation for poorly differentiated sebaceous cell carcinoma . our case did not show poorly differentiated basaloid cells and other features of basal cell carcinoma . sebaceous carcinoma cells express immunohistochemical markers such as cytokeratin , epithelial membrane antigen ( ema ) , cam5.2 and anti - breast carcinoma associated antigen-225 antibody . the current case showed diffuse strong cytokeratin expression and ema was focally positive in cells with sebaceous differentiation . considering the morphological , histopathological and immunohistochemical marker study findings we favoured a diagnosis of sebaceous carcinoma . the common associations of sebaceous carcinoma are muir - torre syndrome , an autosomal dominant condition comprising of sebaceous neoplasm with one or more low - grade visceral malignancies and nevus sebaceous of jadassohn . distant metastases and recurrence rates are more common in the ocular type of sebaceous carcinoma when compared to extraocular sebaceous carcinoma as seen in our case . recurrence rates of ocular sebaceous carcinoma ranges from 11% to 30% with distant metastases occurring in 3% to 25% though extraocular sebaceous carcinoma is less aggressive , a study with 91 cases of extraocular sebaceous carcinoma showed a recurrence rate of 29% with 21% developing metastases . wide excision and selective use of radiotherapy is the ideal treatment of choice . in summary , this tumor should suggest the possibility of muir - torre syndrome and alert the clinician to search for occult malignancies .

extra ocular sebaceous carcinoma is a rare malignancy when compared to peri ocular variant and these are derived from sebaceous gland epithelium . the aggressive types of extra ocular sebaceous neoplasm are reported with lymph node and visceral metastasis associated with poor prognosis . here we report a case of extensive cutaneous extra ocular sebaceous cell carcinoma confined to large area of scalp proven by immunohistochemistry without intra cranial involvement , distant metastases or evidence of muir - torre syndrome .

a widely accepted definition of diabetic peripheral neuropathy is the presence of symptoms and signs of peripheral nerve dysfunction in people with diabetes after exclusion of other causes ( boulton 1998 ) . it can be classified into several syndromes each with a distinct pattern of involvement of peripheral nerves . diabetes being a very common medical condition , one should be aware of the usual as well as its rare presentations . diabetic neuropathy can manifest in any stage of the disease though it tends to be a delayed manifestation in type 2 diabetes . it can present as an asymmetrical painful proximal neuropathy or it can be symmetrical and distal . autonomic neuropathy is a more serious form of nervous system involvement and requires careful monitoring . sometimes in a single individual a 57-year - old male electronic engineer presented with severe burning dysesthesia and pain in d4 - 12 distribution on the right side of his chest and abdomen . he also experienced troublesome allodynia leading to decreased sleep , excessive day time sleepiness and fatigue . the symptoms according to him started 6 months prior to his present visit after he had a chest tube insertion for drainage of pleural effusion . on the basis of pleural fluid analysis he had elevated blood sugar at that time with fasting blood sugar ( fbs ) 246 mg / dl and postprandial blood sugar ( ppbs ) 360 mg / dl with glycated hemoglobin ( hba1c ) 9.2% . he was first diagnosed to have diabetes 3 years ago and was on irregular treatment for the same . a week later he noticed a right abdominal bulge especially after taking a heavy meal or while trying to get up from a lying posture . this was associated with a burning pain over the right lower chest and upper abdomen . he was extensively investigated for his painful abdominal mass including a gastrointestinal scopy and ultrasonography ( usg ) abdomen , however no abnormality could be detected . meanwhile his blood sugar remained uncontrolled , he stopped insulin and changed to herbal medications . on initial evaluation at our hospital , since all results were within normal limits he was referred to neurology department for further evaluation . meanwhile he also developed a right proximal lower limb weakness associated with wasting of thigh muscles leading to buckling at right knee joint . on examination there was a healed scar at the site of intercostal drain insertion in right fifth intercostal space on the anterior axillary line . he had weakness of hip adduction , knee extension , and hip extension all on the right side . the right abdominal muscles were weak with a protrusion of the abdomen mimicking a mass [ figure 1 ] . right knee jerk was absent and sensation to crude touch was diminished by 20% on the right side extending from d4 dermatome to right knee . abdominal bulge on attempted flexion of neck and upper trunk on investigations , he continued to have high blood sugars on admission . cerebrospinal fluid ( csf ) revealed normal sugar with protein 79 mg / dl and 2 cells / mm , mononuclear . magnetic resonance imaging ( mri ) cervical , dorsal , and lumbosacral spine with contrast showed mild cervical canal stenosis at c3-c6 . nerve conduction velocity study ( ncv ) ; right femoral compound muscle action potential ( cmap ) amplitudes were reduced and paraspinal electromyography studies ( emg ) revealed denervation potential from thoracic and lumbar segments . in view of the initial clinical findings of painful sensory symptoms with unilateral abdominal weakness and proximal right lower limb lower motor neuron weakness we considered the possibility of a multiple radiculoneuropathy . since the patient was on antituberculous treatment ( att ) , spinal arachnoiditis secondary to tuberculosis was the initial diagnosis . however , mri spine with contrast and csf study were normal . on reviewing the history he had uncontrolled sugars from the very onset of his symptoms . this prompted us to think the second possibility of a diabetic truncal neuropathy with a unilateral proximal radiculoneuropathy involving the right lower limb . his troublesome abdominal swelling , which was extensively investigated earlier , was due to an abdominal wall weakness , a finding very rarely seen in diabetic truncal neuropathy . it is not a true hernia as there is no protrusion of viscus through a defect in the abdominal wall on the other hand it is the weak muscle wall which bulges out . pain is in a radicular distribution and is usually accompanied by allodynia and tend to become worse at night leading to insomnia and excessive daytime sleepiness as in our case . the pathophysiology of diabetic truncal neuropathy is still a matter of controversy , however being a painful neuropathy an ischemic cause seems to be the most favored hypothesis . our patient had good symptomatic relief with a combination of gabapentine , tramadol , and amitryptaline . on follow up after 3 months he was totally relieved of pain however , his abdominal bulge persisted for which he was advised muscle strengthening exercises and abdominal binders . the clinician should be aware of this uncommon presentation as it can prevent unnecessary investigations . it is a self - limiting condition and usually resolves spontaneously in 2 - 6 months and has a good prognosis .

diabetic neuropathy has varied clinical presentations . as clinicians we should be aware of the common as well as rare manifestations of this syndrome . diabetic truncal neuropathy presenting as a painful pseudoabdominal mass can easily mislead clinicians who are unaware of this problem . subsequently , this can lead to unnecessary investigations and discomfort to the patient . a good blood sugar control and judicious use of drugs for neuropathic pain along with physiotherapy usually gives good relief . it is mostly a self - limiting condition .

endoscopic mucosal resection ( emr ) is an endoscopic alternative to surgical resection of mucosal and submucosal neoplastic lesions and intramucosal cancers . lesions limited to the mucosa and the superficial layers of the submucosa appear to be the most amenable to endoscopic cure . the techniques for emr can be broadly divided into two groups : suction and non - suction techniques . this topic will provide an overview of useful accessories for colorectal emr such as cap and band . this technique is most commonly performed with a transparent cap ( disposable distal attachment ; olympus , tokyo , japan ) attached to the tip of the endoscope . cap - assisted colonoscopy ( cac ) uses a transparent plastic hood attached to the tip of the colonoscope to flatten the semilunar folds and improve mucosal exposure . several studies have examined that cac facilitated shortening of the cecal intubation time in difficult cases , and was more sensitive for detecting adenomas than was conventional colonoscopy.1,2 a meta - analysis and systemic review suggests that a transparent cap on the end of the colonoscope may give a marginally faster cecal intubation time compared with standard colonoscopy . it also suggests that there is a better polyp detection rate and less pain with the cap.3,4 cac may reduce the time required for colonoscopic emr of each polyp and may also improve the polyp detection rate.5 this technique is most commonly performed using a distal attachment fitted to the distal end of the endoscope , with saline solution with a low concentration of epinephrine injected underneath the lesion . the lesion was snared and drawn into the cap using the suction function of the endoscope , and then ligated and resected using electrocautery . the most serious complication of this technique may be perforation due to the lack of submucosal saline injection and too much suction . therefore , large volume injection , which creates a large bleb and potentially reduces the risk of perforation , is recommended.6 moreover , emr using a cap ( emr - c ) is effective at removal of carcinoid tumor which is diagnosed increasingly each year . it is widely accepted that rectal carcinoid tumors with a diameter of 10 mm or less can be treated with local excision , including endoscopic resection.7 complete resection of rectal carcinoid tumors , however , is difficult to achieve with conventional endoscopic resection techniques because these tumors often extend into the submucosa . resection via polypectomy or conventional emr is often associated with resection margin involvement , which necessitates further intervention.8,9 the rate of positive resection margin for tumor is lower in the group of emr - c than conventional polypectomy group.10 - 12 and secondary endoscopic treatment for remnant lesions of rectal carcinoid tumors after primary emr or polypectomy is technically difficult because of fibrosis of the residual tissues . emr - c , a method to resect the submucosal layer by suction by using a transparent cap , may be feasible as a salvage treatment a variation of the suction technique is the band and snare procedure . during the band ( endoscopic ligator ; conmed , new york , ny , usa ) and snare procedure , tissue is banded using an esophageal variceal banding device and then snared off in the standard fashion.14 after submucosal injection beneath the lesion to elevate it away from the muscularis propria , the lesion was aspired into the ligator device and the elastic band was then deployed . next , snare resection was performed below the band with a blended electrosurgical current ( fig . the resection specimen was then removed by aspiration into the cap or by retrieving it with a grasping forceps . after resection of the specimen , the ulcer floor was endoscopically closed with clips to prevent postoperative bleeding and perforation . as with emr - c , complete resection of rectal carcinoid tumor is important but is difficult to achieve with conventional endoscopic resection techniques because these tumors often extend into the submucosa . resection via polypectomy or conventional emr is often associated with resection margin involvement . among various endoscopic resection techniques , endoscopic submucosal resection with a ligation device ( cac is more sensitive for detecting adenomas and may reduce the time required for colonoscopic emr of each polyp . emr - c and emr with a ligation device ( or esmr - l ) may be a superior method to conventional emr for removing small rectal carcinoids .

endoscopic mucosal resection ( emr ) is an endoscopic alternative to surgical resection of mucosal and submucosal neoplastic lesions . prior to the development of knives , emr could be performed with accessories to elevate the lesion . after the development of various knives , en bloc resection was possible without other accessories . so , recently , simple snaring without suction or endoscopic submucosal dissection using knife in the epithelial lesions such as adenoma or early mucosal cancer has been performed . however , for easy and complete resection of subepithelial lesions such as carcinoid tumor , a few accessories are needed . complete resection of rectal carcinoid tumors is difficult to achieve with conventional endoscopic resection techniques because these tumors often extend into the submucosa . the rate of positive resection margin for tumor is lower in the group of emr using a cap ( emr - c ) or emr with a ligation device ( emr - l ) than conventional emr group . emr - c and emr - l ( or endoscopic submucosal resection with a ligation device ) may be a superior method to conventional emr for removing small rectal carcinoid tumors .

we report here a case of prolonged fever post infected pacemaker ( pm ) and lead extraction , with late diagnosis of a retained lead in the right subclavian vein and superior vena cava , whose removal resulted in clinical resolution . a 49-year - old man underwent removal of a pm due to infective endocarditis . in this procedure , the electrodes were left in place and the pm was implanted on the contralateral site . after 2 weeks , the patient was readmitted with sepsis related to the cardiac device , which was completely removed followed by the implantation of an epicardial pm . as the patient continued to have fever , he was referred to the university hospital , federal university of minas gerais , belo horizonte , brazil , which is a tertiary care center . extensive workup for fever of unknown origin did not clarify the cause of the fever , which remained in spite of the use of different antibiotics including agents to cover enteroccocus sp that was isolated from the blood cultures . a vascular duplex scan was performed and showed the presence of an endovascular foreign body in the right subclavian vein and superior vena cava associated with thrombus . the diagnosis was confirmed by computed tomography ( ct ) ( fig . 1 ) . the patient underwent endovascular removal of the foreign body , which was identified as the distal part of the right ventricular lead , measuring about 10 cm long . after the procedure , he presented clinical and laboratory improvement consistent with complete resolution of the infection , and was discharged from the hospital after completing 28 days of antibiotic therapy guided by blood culture . from the last decades , the indications for implant of cardiac electronic devices continue to expand . as a result , we have observed increasing complications related to the removal of these devices . in spite of the evolution of the extraction techniques from simple traction to extraction with dilators , powered sheaths and laser assistance , percutaneous removal of the device leads is still associated with morbidity and mortality , though in a small proportion of cases , . the more frequent indications for cardiac device extraction are lead dysfunction , pocket infection and systemic infection , and the rate of successful lead remove is over 94% , , . procedural - related major complications include death , cardiac and/or vascular avulsion , pulmonary embolism , stroke , and pacing system - related infection of a previously non - infected site . pericardial effusion or hemothorax , hematoma at the surgical site , arm swelling or vein thrombosis , migration of lead fragment without any sequelae , and blood loss during surgery requiring blood transfusion are considered minor complications of lead extraction . death within 30 days from the extraction is also included among the outcomes of lead extraction procedures . our patient developed blood stream infection due to the persistence of the retained the distal part after percutaneous lead extraction coating cable of the pm associated with thrombus formation in the right subclavian vein and superior vena cava , which may be considered a major complication of lead extraction . as the lead extraction was performed in a low volume center , we hypothesized whether this fact could have contributed to the development of the complication . this possibility is reinforced by the results of a recent meta - analysis , in which the outcome of lead extraction was evaluated according to the center experience based on the volume of procedures carried out ( low , medium , and high volume centers ) , demonstrating that procedure volume is a major determinant of outcomes of transvenous lead extraction . the diagnosis of the cause of the infection presented by our patient was challenging especially because of the lack of local manifestations . the absence of these manifestations also contributed to overlooking the foreign body on imaging methods initially . indeed , we noticed the foreign body on ct scan only after the result of the vascular duplex scan . we conclude that a retained foreign body should be considered in the differential diagnosis of persistent fever after removing a pm system . firstly , it reinforces the need of examining the extracted material after lead extraction ; so that any missing fragment / coating can be identified and looked for . secondly , it shows the importance of having a detailed scheme of analysis of the imaging methods . vascular doppler and chest ct are part of the basic work up for such cases ; thus , a proper scheme of analysis of their results would have allowed diagnosing the presence of a retained foreign body at an earlier stage in our patient .

the implant indication of cardiac electronic devices continues to expand ; therefore , we have observed increasing complications related to their removal . we describe the case of a patient who presented with prolonged bloodstream infection after having undergone removal of a pacemaker . after extensive workup for fever of unknown origin and antibiotic therapy without any improvement , it was possible to demonstrate a foreign body in the right subclavian vein and superior vena cava corresponding to the distal part of the right ventricular lead . endovascular removal of the foreign body and prolonged antibiotic administration was followed by complete resolution of the clinical picture . we ascribed the difficulty in diagnosing the source of the infection especially to the lack of local manifestations .

the patient is a 46-year - old african american male with a history of stroke , diabetes , and hiv , who presented with a sudden onset shortness of breath , palpitations , and chest discomfort . physical examination was unremarkable except for tachycardia and right - sided weakness associated with an episode of stroke 10 years earlier . ct chest angiography demonstrated a large bilateral pulmonary embolism ( pe ) with straightening of the interventricular septum suggesting right heart strain ( fig . right heart strain necessitated a transthoracic echocardiogram ( tte ) that showed a possible thrombus versus mass in both atria . to confirm the diagnosis , the tee showed a pfo with a large thrombus measuring 10 cm lodged in the pfo ( fig . a tee with bubble study , which was done as part of the previous stroke workup , did not show any right - to - left shunt . cardiac surgery was consulted for possible surgical removal of the thrombus and repair of the pfo . after a discussion of the risks and benefits of surgical versus medical management a year later , follow - up tte did not show a pfo or any clot in the heart . the mortality rate in cases of pe with thrombus in transit is very high ( 1 ) . there is no general consensus about the best treatment for a thrombus entrapped in a pfo ( 2 ) . because of the high risk of death from thrombus in transit , a cardiothoracic surgeon should be consulted immediately upon confirming the diagnosis ( 3 ) . if the pfo is not repaired , the patient should remain on anticoagulation indefinitely ( 4 ) . this case is also a reminder that a negative tee does not rule out the presence of a pfo ( 5 ) . the authors have not received any funding or benefits from industry or elsewhere to conduct this study .

in rare cases , thrombus in transit can be entrapped in a patent foramen ovale ( pfo ) . a patient with this condition is at high risk of embolic stroke and death . early diagnosis and treatment is essential to help prevent stroke and death in these cases . there is no universal management guideline for this rare condition . the decision between medical versus surgical treatment should be made individually for each patient . we present a case of thrombus in transit entrapped in a pfo that was treated medically by lifelong anticoagulation .

thirteen f. tularensis isolates collected over the past 10 years in switzerland ( figure 1 ) were subjected to extensive genetic characterization . the species and subspecies designations of all strains were confirmed by real - time pcr that targeted the fopa gene and by amplification of the rd1 region ( 8) , which showed that all strains were f. tularensis subsp . holarctica strains ( 7 ) and the genome sequence of the strain isolated in france , ftnf002 ( genbank accession no . nc_009749 ) , were included in the study to represent the currently known genetic subpopulations within the subspecies . all strains from switzerland were genetically characterized at 6 highly variable loci ( by mlva ) and 14 more stable loci that indicate the classification f. tularensis subsp . the rd analysis was also performed because a 1.59-kb deletion marker , rd23 , was reported to be restricted to strains from france and spain ( 3 ) . the mlva markers ( m3 , m6 , m20 , m21 , m22 , and m24 ) and ftind markers ( ftind 2538 ) were amplified by pcr and then sequenced with an abi prism 3100 genetic analyzer ( applied biosystems , foster city , ca , usa ) and the bigdye terminator cycle sequencing kit ( applied biosystems ) . dna fragment sizes were calculated from the nucleotide sequences of the mlva and ftind markers and used to compare the isolates with previously analyzed strains from the united states , japan , france , and russia ( 3,7 ) . the rd23 marker was assayed by using standard pcr and agarose gel methods as previously described ( 3 ) . a cluster analysis based on the mlva and indel size data was performed by using bionumerics version 3.5 ( applied maths , kortrjik , belgium ) . the unweighted pair group method with arithmetic mean phylogram is based on the combined ftind and multiple - locus variable - number tandem repeat analysis . bootstrap values > 80% are given at the respective nodes and were calculated by using 10,000 iterations . as expected , the indel markers served to place each strain into major branches of the cluster tree , and the more variable mlva markers provided the fine resolution at the tips of the tree . the switzerland strains belonged to the same genetic cluster as the f. tularensis strain ftnf002 from france ( figure 1 ) that in a previous work clustered with strains from spain ( 3,9 ) . moreover , all the swiss strains exhibited the 1.59-kb genomic deletion at the rd23 locus and the unique 464-bp size at mlva marker m24 , which confirmed their close relationship to the french strain ftnf002 as well as to other strains from france and spain ( 3 ) . the finding of f. tularensis strains in switzerland represent sporadic occurrences of tularemia without any obvious epidemiologic connection . the strains originated from 6 hares , 3 monkeys , and 4 persons and were collected at different locations in switzerland over a period of 10 years ( figure 2 ) . the human infections most likely occurred through direct contact with wild animals , through rodent bites , and through consumption of a hare cooked at low temperature . four swiss strains displayed a genetic profile identical to that of the representative french strain ftnf002 ( 3 ) . the other 6 genotypes were closely related to ftnf002 , and all corresponded to the subclade b.br:ftnf00200 as defined by canonical single nucleotide polymorphisms by vogler et al . this cluster , which also contained the strains from the iberian peninsula , seems to have spread throughout central and western europe . moreover , all the swiss strains were susceptible to erythromycin ( mics 0.25 g / ml to 1 g / ml ) , which is a phenotypic marker that has previously been suggested to divide f. tularensis subsp . strains of f. tularensis from switzerland ( central europe ) genetically clustered with strains from france and spain ( western europe ) as determined by the unique 464-bp genetic marker m24 and a specific deletion at marker rd23 . furthermore , strains within the cluster differed at only 2 mlva markers , and 4 other mlva and 14 ftind markers were identical . in a previous study that included strains from the 19971998 tularemia outbreak in spain , the specific m24 allele and the rd23 deletion were found in 49 of 49 strains from spain and france but in only 1 of 189 strains from 7 northern and eastern european countries and japan ( 3 ) . the tularemia outbreak of 19971998 in spain , which resulted in > 500 human cases ( 5 ) , was thus caused by f. tularensis strains that were genetically closely related to strains recovered in switzerland from 1996 onwards , before the beginning of the outbreaks in spain . this genetic relationship shows that factors other than the presence or introduction of a specific clone of the infectious agent per se determined the magnitude of the tularemia outbreaks in spain . for epidemiologists to understand the distribution of f. tularensis ( and other rare disease agents ) in the environment and their propagation across national and geographic borders , surveillance programs that include molecular analyses of these agents should be undertaken in multiple countries , and the resulting data should be shared internationally .

we conducted a molecular analysis of francisella tularensis strains isolated in switzerland and identified a specific subpopulation belonging to a cluster of f. tularensis subsp . holarctica that is widely dispersed in central and western continental europe . this subpopulation was present before the tularemia epidemics on the iberian peninsula .

lesions of the anterior interosseous nerve are rare and comprise less than 1% of all upper extremity nerve palsies . traumatic causes include blunt trauma , forearm fractures , penetrating injury and local pressure from a plaster cast , but has never before been described in association with crutch use . this is the first reported case of the use of elbow crutches causing symptomatic anterior interosseous nerve compression . this case describes a 30-year - old male who developed an inability to pinch with his left hand following the use of elbow crutches for a foot injury . on examination a diagnosis of anterior interosseous palsy was made and the patient was treated conservatively and crutch use was ceased . at six weeks follow - up the patient made a complete recovery with full function of his left hand . this report highlights the importance of adequate education in the safe use of elbow crutches for all patients . the side effects of inappropriate use should be carefully examined for during follow - up care . lesions of the anterior interosseous nerve [ ain ] are rare and comprise less than 1% of upper limb palsies . it is an exclusively motor branch of the median nerve and provides innervation to flexor pollicis longus , the medial part of flexor digitorum profundus involving the index and sometimes the middle finger , and to pronator quadratus . entrapment of the nerve results in a characteristic appearance on pinching , which may be mistaken for a tendon rupture . we present a case of a patient who developed ain palsy following the use of elbow crutches . a 30-year - old fit and healthy male presented to fracture clinic four days after suffering a stubbing injury to his left big toe and superficial abrasion to his left heel . x - rays reviewed an undisplaced fracture of the base of the left little toe distal phalanx . he was given elbow crutches with instructions for partial weight bearing on the affected leg and discharged with a fracture clinic appointment . in clinic , the patient complained of a gradual onset of difficulty in pinching and gripping objects with his left hand since the injury . [ figures 1 and 2 ] movements of the left index and middle fingers were normal and there were no areas of paraesthesia . since there was no history of trauma or predisposing conditions , we believe the patient developed an anterior interosseous nerve palsy following the inappropriate use of elbow crutches over four days . he was subsequently placed in a plaster cast boot and was advised to fully weight bear without the use of crutches . however , by the time he came to a six - week follow - up clinic after the injury , he had made a complete clinical recovery in hand movement and function . many causes for ain palsy have subsequently been described in literature , and can be divided into traumatic and non - traumatic / spontaneous . traumatic causes range from blunt trauma , local pressure from sleeping on the affected arm or poorly applied cast , excessive exercise , penetrating injury , forearm fractures and open reduction and internal fixation . to our knowledge ain palsy can be described as complete , where flexor digitorum profundus and flexor pollicis longus are both affected , or incomplete , where only one muscle is affected . it differs from other lesions of the median nerve in that sensory complaints are essentially absent . as a result of these factors , ain palsy can often mimic single tendon lesions , leading to difficulties with diagnosis . in the literature , there are cases of ain entrapment with unnecessary surgical exploration of fpl tendon . o with the thumb and index finger , and the loss of ability to perform fine tasks despite an intact sensory innervation . spontaneous ruptures of flexor pollicis longus tendon are rare and are generally associated with distinct underlying pathologies , such as rheumatoid arthritis . several clinical tests have been described to distinguish between palsy of fpl and rupture of this tendon[810 ] . electrodiagnosis with nerve - conduction study or electromyography is the investigation of choice to differentiate between nerve palsy and tendon rupture . in the present case , however , the patient made a full recovery before electrodiagnostic studies could be carried out , thereby ruling out the possibility of rupture of flexor pollicis longus . conservative management includes removal of precipitating causes such as crutches and casts , immobilisation and anti - inflammatory medication . we believe all patient should be taught safe and effective usage of elbow crutches when they initially receive them . the side effects of inappropriate use should be carefully examined for during follow - up care .

context : lesions of the anterior interosseous nerve are rare and comprise less than 1% of all upper extremity nerve palsies . traumatic causes include blunt trauma , forearm fractures , penetrating injury and local pressure from a plaster cast , but has never before been described in association with crutch use . this is the first reported case of the use of elbow crutches causing symptomatic anterior interosseous nerve compression.case report : this case describes a 30-year - old male who developed an inability to pinch with his left hand following the use of elbow crutches for a foot injury . on examination he was unable to flex the interphalangeal joint of his left thumb . a diagnosis of anterior interosseous palsy was made and the patient was treated conservatively and crutch use was ceased . at six weeks follow - up the patient made a complete recovery with full function of his left hand.conclusion:this report highlights the importance of adequate education in the safe use of elbow crutches for all patients . the side effects of inappropriate use should be carefully examined for during follow - up care .

osteoporosis is a deficiency of bone mineral density that increases fracture risk . because the number of elderly people in the population is currently increasing the age - specific prevalence of osteoporosis is less than 2% in women younger than 50 years . between the ages of 20 and 40 years our case provides a perspective on the diagnosis of postpartum osteoporosis and complications such as compression fracture . one month earlier , she had a tertiary cesarean section and a left ovarian cystectomy . during the cesarean section , there were no other complications . she had no past surgical or family histories , nor had trauma . on physical examination , she had diffuse back pain . the pain was so severe that she had difficulty in getting up for 7 days . the lumbar spine anteroposterior ( ap ) and lateral revealed recent compression fractures of t12 , l1 , and l3 . a bone scan also revealed increased bone uptake , perfusion , and multiple recent compression fractures of t12-s1 ( fig . bone marrow density determined using dual energy x - ray absorptiometry ( dxa ) revealed that the t - scores for the lumbar vertebrae l1-l4 , femoral neck , and femoral total were significantly low in the patient ( fig . 2 ) . she was given calcium 500 mg daily and a chest brace for non - weight bearing . osteoporosis is a skeletal disease involving decreased density of the mineral portion of the bone . the causes of osteoporosis are most commonly age , menopause , hormone imbalance involving the thyroid or parathyroid , and alcohol intake . osteoporosis is diagnosed according to the 2007 international society for clinical densitometry ( iscd ) official positions or world health organization ( who ) criteria . we previously differentiated the two criteria and revealed that the who criteria are more tolerant than the iscd official positions . because osteoporosis is related to fractures , a fracture - risk assessment tool ( frax ) using clinical risk factors combined with femoral neck bone mineral density ( bmd ) can predict the 10-year fracture risk . we previously reviewed the korean frax model with the general population and recommended that physicians use the korean frax model in the general female population to predict fracture risk . we already found that serum ( 25-[oh]d3 ) concentrations were significantly correlated with age ( p=0.001 ) , duration of menopause ( p=0.005 ) , and osteoporosis ( p=0.044 ) . also , erythrocyte levels of n-3 polyunsaturated fatty acid ( pufa ) and intake of fish were positively correlated with bmd of the femoral neck . however , vitamin d and diet are not included as fracture risk factors . in our analysis of the bmd and previous obstetric history , another study also revealed that postmenopausal women had a correlation between risk factors for osteopenia or osteoporosis and obstetric history including previous gravity . a possible mechanism of postpartum osteoporosis involves ligament laxity due to endocrine changes such as parathyroid hormone ( pth)-like peptide , magnesium sulfate management , prolonged bed rest for preterm labor , and stress or fatigue fractures during delivery . the fetus takes 30 g of calcium from the mother , and hypoestrogenemia during breast - feeding leads to bone mineral loss in women . as the gestational age proceeds , because of their increased number of pregnancies , they have a longer duration of breast - feeding . therefore , correlations between the severity of bmd and breast - feeding duration with osteoporosis are unclear . the most common symptom is severe back pain in the lower thoracic , lumbar region . the most common site in pregnancy- and lactation - associated osteoporosis is the vertebrae , as in our case . in particular , more than 1 standard deviation ( sd ) of bone loss was found . we should recognize the potential risk of postpartum osteoporosis and carefully differentiate back pain after delivery to prevent osteoporotic fractures .

osteoporosis is mainly a problem in postmenopausal women . however , we had a case of postpartum compression fracture associated with osteoporosis . a 42-year - old multiparous woman had undergone tertiary cesarean section without complications 1 month before . she was breast - feeding her baby and had no other bone - related complication history . she did not exercise on a regular basis . she experienced back pain abruptly and was diagnosed with a recent compression fracture of t12 , l1 , and l3 . we evaluated this rare case of postpartum osteoporosis and compression fracture .

esophagopleural fistula ( epf ) is an uncommon condition despite the anatomical proximity of the trachea and esophagus . causes of epf include pneumonectomy for suppurative or tubercular disease and carcinoma of the lung and malignancy of the esophagus . non - malignant epf is due to trauma or infection . the most common infectious cause of epf is tuberculosis , the others being syphilis , mycotic disease , and crohn 's disease . perforation of the esophagus and subsequent fistula formation can occur as a result of foreign bodies , barrett 's ulcer , and more rarely boerhaave 's syndrome . spontaneous development of a fistula between the esophagus and pleura is rarely described in the literature . a 34-year - old male was admitted with a 1-month history of cough , breathlessness , regurgitation during feeding , and chest discomfort . he was a known case of bipolar mood disorder and was on antipsychotics for the last 7 years . general survey was unremarkable . on chest examination , an enlarged right hemithorax with decreased movement of the right chest and stony dull note over the right hemithorax from the fourth , eighth , and ninth intercostal spaces downward along the midclavicular , midaxillary , and scapular chest x - ray revealed right pleural effusion . on thoracocentesis , there was a dry tap . on ultrasonography of the thorax , there was minimal right pleural collection with internal echoes ( not amenable for aspiration ) . on high - resolution computed tomography ( hrct ) of the thorax , an encysted collection ( 7.9 cm 8.5 cm 10 cm ) was noted in the right pleural space showing air and food particles in it with a linear fistulous communication from the distal esophagus , and on oral contrast administration , the contrast medium was seen to enter into the collection [ figures 1 and 2 ] . finally , the upper gastrointestinal ( gi ) endoscopy confirmed a large fisulous tract measuring 9 mm at the distal esophagus without any mucosal abnormality . with the definite diagnosis of epf , the patient was transferred to the department of cardiothoracic and vascular surgery ( ctvs ) where thoracotomy and closure of the fistula were performed . during the procedure , a number of possible causes for the development of epf following pneumonectomy for suppurative disease of the lung have been suggested . anatomically , the esophagus lies much closer to the right hemithorax than to the left , the left being separated from the pleural cavity by the aorta . the possibility of direct epf and extent of mediastinitis are determined by the anatomic relationship of the esophagus and the pleura , the amount of mediastinal fat , and intervening connective tissue . fistula related to empyema was the most commonly reported case in the preantibiotic era . nonetheless , there continues to be isolated reports of posttuberculous pyopneumothorax fistulas . erosion of an empyema into the esophagus and rupture of caseating lymph nodes into the esophagus are the possible mechanisms . in our case , the cause of epf was unclear . evidence of malignancy was undetectable from radiology and no specific infectious agent was isolated at the time of diagnosis and treatment . esophageal injury should be considered when a patient presents with retrosternal chest pain , fever , dysphagia , and dyspnea , especially when the patient gives antecedent history of instrumentation or surgery . the diagnosis of epf can be suspected clinically ; however , for confirmation , imaging is required . the imaging modalities include chest radiograph , ultrasound , barium swallow , contrast - enhanced computed tomography ( ct ) , and magnetic resonance imaging ( mri ) with each modality having its advantages , and chest ct is a very useful modality . leaks of the esophagus are associated with high mortality and the need to be treated as soon as possible . therapeutic options include surgical repair or resection or conservative management with antibiotic therapy and cessation of oral intake . endoscopic treatment with fibrin glue , clip , suturing , and metallic stents has been described . to conclude , spontaneous development of epf is an unusual condition entity with nonspecific clinical presentation . ct of chest is a very useful modality for early diagnosis and management of epf . it should be performed in patients with pleural effusion presenting with nonspecific clinical symptoms before any intervention or drainage .

esophago - pleural fistula ( epf ) is an uncommon condition , despite of an anatomical proximity of these structures . causes of epf include pneumonectomy for suppurative or tubercular disease of lung and carcinoma lung , malignancy of esophagus . benign epf is rare and may be due to trauma or infection . the most common infectious cause is tuberculosis . spontaneous development of fistula between esophagus and pleura is rarely described in literature . we , hereby present a spontaneous case of such a rare entity in a middle - aged male .

gastrointestinal bleeding ( gib ) is a well - described complication of the left ventricular assist device ( lvad ) placement and has been reported to occur in 1940% of patients with the heartmate ii device . the mechanism of gib in these patients may be secondary to alterations in vascular endothelial physiology , hemodynamic alterations , hematologic imbalances , coagulopathy , acquired von willebrand disease , and angiodysplastic lesions . the patient had a heartmate ii lvad placed 3 years prior to presentation for medically refractory heart failure . he presented to the emergency department with light - headedness and hematochezia for several days . his inr was 2.8 , creatinine was 2.3 mg / dl , and hemoglobin was 10.4 from baseline of 12 to 13 g / dl . he underwent esophagogastroduodenoscopy revealing a gastric ulcer with a clean base with no evidence of active bleeding . a colonoscopy revealed a likely malignant tumor in the cecum , diverticulosis , and nonbleeding colonic angiodysplastic lesions [ figure 1 ] . the cecal mass was biopsied ; it appeared to be actively bleeding and uncontrollable endoscopically . colonoscopic image of apparent cecal mass with surrounding blood he was taken emergently to the operating room . a midline laparotomy was undertaken and he was found to have a cecal mass that was perforated into a retroperitoneal abscess . the right colon was mobilized to the hepatic flexure and the abscess cavity was debrided . a right colectomy was performed with a stapled side - to - side ileocolonic anastomosis . a drain was placed in the abscess cavity , fascia was closed , and the skin and subcutaneous tissues were packed with gauze . it revealed a transmural defect of 1.0 cm 0.5 cm with organizing hematoma and acute on chronic inflammation . the patient 's postoperative course was complicated by prolonged ileus necessitating total parenteral nutrition and pelvic abscess that required percutaneous drain placement . the patient 's postoperative complications eventually resolved , and he was discharged to home with home health . gib is more common in lvad patients than patients with mechanical valves on anticoagulation , showing that lvad physiology is a major factor . it is difficult to determine the risk associated with warfarin , since inr is variable in different studies and aspirin is routinely used . causes of gib in a meta - analysis of 136 patients showed angiodysplasia ( 29% ) , gastritis ( 22% ) , peptic ulcer disease ( 13% ) , diverticular bleeding ( 6% ) , colonic polyps ( 5% ) , colitis ( 3% ) , and unknown or other causes ( 22% ) . location of bleeding by pooled event rates in this study was 48% for upper gib , 22% for lower gib , 15% for small intestinal bleeding , and 19% was unable to be localized or not evaluated . to our knowledge , this is the first report of a gib masquerading as a colonic malignancy in a patient with an lvad . there has been a shift from pulsatile to continuous flow pumps such as the heartmate ii . continuous flow pumps have shown improved durability , decreased thromboembolic events , and improved survival at the cost of increased incidence of gib . found that history of previous gib was independently associated with gib with lvad placement , while a meta - analysis of 1839 lvad patients found that older age and elevated creatinine were associated with gib whereas prior history of gib or differences in lvad settings were not associated with differences . when treating the gib is a cessation of anticoagulants , correction of underlying coagulopathy , resuscitation , and acid suppression . capsule endoscopy or push enteroscopy may be used to help with localization in difficult cases . potential therapeutic options include endoscopic treatments , embolization , and surgical intervention , when less invasive means prove to be unsuccessful . octreotide has been tried , but there is no strong evidence to support its routine use . depending on the severity of the gib , lesion , and treatment , inr goal may be lowered to reduce the risk of future events or anticoagulation may even be withheld when necessary . there is a theoretical higher risk of thromboembolic events when anticoagulation is reduced . in a study by morgan et al . , patients with an episode of gib transitioned to lower dose warfarin ( inr 1.52.0 ) and aspirin ( n = 8) or with aspirin only ( n = 5 ) therapy had no thromboembolic events . previous gib , advanced age , continuous flow lvads , and renal failure are the risk factors . treatment includes reversal of anticoagulation , resuscitation , localization , endoscopy , and surgery , if needed .

there are many complications associated with the left ventricular assist devices ( lvads ) , including gastrointestinal bleeding ( gib ) . we present a case of a pseudo colonic mass visualized on colonoscopy during workup for gib in an lvad patient necessitating a right colectomy with final pathology negative for malignancy . a review of the literature in regards to the pathology , diagnosis , and treatment of this interesting condition is included .

38 cases ( 13.92% ) had varied symptoms of anxiety , irritability , sleeplessness , agitation , unprovoked aggressive behavior , and mild depression following the detection of hiv positivity table 1 . hiv seropositive status and psychological symptoms a detailed analysis of the seropositive cases from jan 2010 to aug 2010 revealed that out of 1977 cases 74 ( 3.74% ) were found positive and almost all the cases had symptoms of anxiety , irritability , depressive symptoms , and agitation [ table 2 ] . a closer look of the positive cases revealed that the rate of positivity was higher in females than males . a total of 6135 who were subjected to pre - test counseling from jan 2008 to aug 2010 . a total of 5688 ( 92.71% ) underwent tests for hiv status [ table 1 ] . out of these , significance of the test was explained to the cases and if they found positive what steps they ultimately take to get themselves treated with antiretroviral therapy , testing of family members , and the universal precautions that they have to resort to were highlighted . 273 ( 4.80% ) cases were found positive for hiv by western blot , 246 ( 90.10% ) cases were put on art , and 1 case ( 0.37% ) was detected to have developed had stage ii [ figures 14 ] . mri brain showing hyper intense areas in frontal and temporo - parietal areas mri brain showing hyper intense areas in internal capsule and thalamus mri brain showing subcortical lesions in frontal and temporal regions mri brain showing subcortical lesions in frontal and temporal regions 38 cases ( 13.92% ) had varied psychiatric symptoms of anxiety , irritability , sleeplessness , agitation , unprovoked aggressive behavior , and mild depression following the detection of hiv positivity . neuropsychiatric manifestations following detection of hiv virus has been described since the earliest reports on aids . it is now recognized that neuropsychiatric signs and symptoms can result from direct effects of hiv on the brain and cns or from indirect effects such as opportunistic infections or tumors associated with immunosuppression , cerebrovascular disease , systemic toxicity , and complications of antiretroviral therapy . as understanding of the broad range of neuropsychiatric manifestations of hiv has grown , new classification and diagnostic criteria has replaced the earlier more inexact terms such as hiv encephalopathy and adc . hiv virus enters the cns early in the course of illness and induces neural injury / inflammation . although hiv does not productively infect neurons , replication of the perivascular macrophages and microglia alters the usual functioning through cascade of neurotoxic molecular events . histologically major changes are seen in the subcortical areas of the brain and include pallor and gliosis ; multinucleated giant cell encephalitis , and vacuolar myelopathy [ ref figures 14 vide infra of the had stage ii case ] . hiv rna may be present throughout the cns and hiv - associated neuropathies are consequently observed in many brain regions including white matter tracts , neo - cortex , basal ganglia , and limbic system . the preferential impact of hiv infection on fronto - striatal circuits results in prototypical pattern of deficits , including slowed information processing efficiency , executive dysfunction , and deficient episodic memory encoding and retrieval . clinically , impaired ability to concentrate , increased forgetfulness , difficulty in reading , increased difficulty in performing complex tasks especially dual tasks or divided attention paradigm , deficits in working memory , verbal and spatial working memory process , apathy , reduced spontaneity , inertia , irritability , lack of initiative , and social withdrawal . among the motor deficits are unsteady gait , poor balance , tremor , and difficulty with rapid alternating movements . increased tone and although had is the initial aids defining illness in 3% of hiv + ve patients , it is a clinical dilemma in undiagnosed hiv cases in the early stages . the hiv patients can hope for a better living with the easy availability of drugs made available by the government . education of the ill effects of the hiv remains the mainstay with regard to control of this dreaded infection .

background : considerable clinical research has been conducted to increase our knowledge in understanding the underlying neuropathological significance of hiv viral infection.aim:to find the incidence of hiv associated dementia in a suburban part of india.materials and methods:6135 prospective cases from january 2008 to august 2010 were subjected to pretest counseling . those willing were tested for hiv status using western blot test.results:5688 ( 92.71% ) underwent for detection of hiv.273 ( 4.8% ) were tested positive . 246 out of these ( 90.10% ) were put on art . 1 ( 0.37% ) was detected to have had stage ii.38 cases ( 18.92% ) had varied psychiatric symptoms.conclusion:haart has considerably reduced morbidity in hiv infection .

clay - shoveler 's fracture is isolated spinous process avulsion fracture classically observed in the lower cervical or upper thoracic spine . this type of fracture is most common in those who engaged in hard physical labor3,6,7 ) . recently , several cases of isolated spinous process fractures that are caused by motor vehicle accidents or sports injuries have been reported ( table 1 ) . isolated spinous process fractures are most common at the level of t1 , followed by c7 , t2 , t3 , and c61,7 ) . among them , multiple spinous process fractures are very rare1 ) . we present a case of multiple isolated spinous process fractures with twelve contiguous levels of the cervico - thoracic spine , which extended from c4 to t8 . to the best of our knowledge , this case is the most multiple spinous process fractures so far reported in the literatures . a 34-year - old man was transferred our hospital from local clinic after motorcycle accident . initial spine x - rays showed multiple spinous process fractures in the cervico - thoracic spine . computed tomography ( ct ) scanning of the cervicothoracic spine showed twelve contiguous spinous process fractures ranged from c4 to t8 ( fig . cervico - thoracic spine magnetic resonance images ( mri ) revealed that spinal cord was normal and there was no additional soft tissue injury ( fig . after 3 months , we evaluated follow - up cervico - thoracic ct scanning which did n't show union of spinous process fractures yet . after 6 months follow - up , visual analogue scale score was gradually improved from 6 to 1 . clay - shoveler 's fracture may occur through direct trauma on the flexed spine or through shear forces2,3,4,5,6,7 ) . muscle and ligamentous stress is transmitted through the supra - spinous ligaments and results in an avulsion fracture of the spinous processes2 ) . our case is an unusual example for multiple spinous process fracture that can be produced by several mechanisms , such as a direct blow to the posterior aspect of lower neck and the thorax , upper back hyperextension , and hyperflexion injuries against the supraspinous ligament after motorcycle crash . a double spinous process sign at the affected level can be detected on simple antero - posterior radiographs6,9 ) . downward displacement of fractured spinous process fragments ( spinolaminar breach sign ) can be seen on lateral radiograph views3,6,7 ) . isolated spinous process fracture considered as a warning sign for more severe spinal injury , so it should be evaluated carefully to detect more severe spinal injuries7 ) . ct and mri of the cervical and thoracic spine may be useful for detecting more serious spine fractures and further injuries to surrounding structures including spinal cord and ligament3,6 ) . generally , this type of fractures are treated conservatively without the need for surgical intervention3,5,8 ) . immobilization and restriction of physical activity for 4 to 6 weeks generally result in good outcome3,8 ) . our patient shows good clinical outcome after 4 weeks immobilization and restriction of physical activity . we report a case of twelve isolated spinous process fracture of the cervico - thoracic spine after motorcycle accident .

the clay - shoveler 's fracture is an isolated avulsion fracture of the lower cervical or upper thoracic spinous process . among them , multiple spinous process fractures are very rare . we present 34-year - old male patient who have multiple spinous process fractures with twelve contiguous levels of cervico - thoracic spine ( from c4 to t8 ) after motorcycle accident . this case is multiple isolated spinous process fractures with good clinical outcome .

road traffic accidents constitute the most common cause . among the penetrating injuries , gunshot and stab injury are the main causes in the western world . in india , proper incidence and causes are not known . though in western literature it is unheard of the arrow injury , it is a common finding in india , especially in the tribal areas . delivery of a baby with wound caused by penetrating injury in mother is not commonly encountered . here , we are presenting a case of intra uterine injury of a fetus caused by an arrow striking his mother at term . a male baby of 2.7 kg weight was brought to outpatient department with the history of delivery with an injury on the right cheek [ figure 1 ] . the cesarean section was done as the mother had a penetrating arrow injury on her lower abdomen due to an assault 9 h before . she came to hospital with the arrow still penetrating in her body [ figure 2 ] . baby with the wound on the face mother with the arrow there was a four cornered star shaped injury of 2.5 cm diameter on the right cheek . as general anesthesia could not be arranged immediately , the procedure was done under local anesthesia . trauma during pregnancy is a unique event because two patients , the mother and her fetus , are at risk and need special evaluation and management . penetrating injury as a cause of maternal - fetal injury during pregnancy is found in one third of such cases . gunshot , stab wounds , and attempt of illegal abortion are the most common causes of penetrating trauma . in penetrating trauma , probability of maternal organ damage is about 1540% and fetal injury is about 70% . if compared with blunt trauma fetal injury is more likely in penetrating trauma . fetal death after gunshot injury is 71% and stabbings 42%.4 muzumdar and his colleague described a bizarre case from canada where a baby had head injury when mother herself fired an air gun through her vagina at term . following a spontaneous precipitous vaginal delivery the baby had convulsion and was found to have intracranial hemorrhage with a metal pellet near the right lateral ventricle . the newborn had a right temporal swelling and it was diagnosed as a subcutaneous hemangioma . but at 2 years of life , it was symptomatic and diagnosed as growing skull fracture with dural tear due to the prenatal injury . patient needed neurosurgical procedure . though the injury in the baby and its management was simple in our case , it is presented here because of its fascinating nature and the interesting backdrop . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed .

injury of a pregnant lady risks both mother and fetus . various modes of injuries are possible . but arrow injury is not usually heard of in today 's world . we have reported a male child delivered with a cut injury on the face . it was caused by a penetrating arrow hitting his mother in her lower abdomen at term . the injury of the baby was repaired successfully .

a 37-year - old chinese male presented to the department of ophthalmology and visual sciences , alexandra hospital , singapore , with a history of poor vision in the left eye since childhood . visual acuity was light perception in the left eye and 20/20 in the right eye . fundus examination in the left eye revealed mnf extending from the optic disc up to the equator [ fig . 1 ] . montage fundal photograph of the left eye showing extensive retinal myelination manifest refraction was performed with a best - corrected visual acuity ( bcva ) of 20/20 in the right eye and of light perception in the left eye , with a spherical equivalent of -3.50 diopter sphere ( ds ) and of -12.50 ds , respectively . to further investigate the cause of poor visual acuity , the patient was subjected to electrophysiology testing , which included a pattern perg , pattern visual - evoked potential ( vep ) and a full - field erg , all conducted under the international society for clinical electrophysiology of vision ( iscev ) protocol.[68 ] in the perg findings , p50 and n95 amplitudes were normal in the right eye ( p50 = 6.2 , n95 = 9.5 ) , suggesting normal macula and retinal ganglion cell function , but undetectable in the left eye ( no observable response ) . pattern reversal vep was also undetected in the left eye ( no observable response ) . these findings are suggestive of poor visual function in the left eye . in the full - field erg findings , the scotopic b - wave ( rod response , a result of the depolarization of the on - bipolar neurons ) was within normal limits in amplitude , although higher in the right eye compared with the left . the maximal and photopic responses were also within normal limits for both eyes [ table 1 ] . the perg has been shown to be important in clinical and research applications , as it is useful in providing information about retinal ganglion cell function , including macular function . therefore , by using a similar stimulus to that for the vep , the perg can aid in its interpretation when the vep is abnormal . in this case , an undetectable perg in the left eye is consistent with abnormal ganglion cell function , which correlates with amblyopia in that eye . however , in addition to studies that suggest that mnf may be considered an oligodendrocytic choristoma , further studies have also shown the disappearance of mnf in patients who had later developed branch retinal artery occlusion due to ischemic atrophy of the ganglion cell and nerve fiber layer . there has also been an implication of the loss of ganglion cells post - central retinal artery occlusion ( crao ) , where the photopic negative response in cases of crao have been shown to be severely affected despite preservation of the cone b - wave . this makes it possible , although not easy to establish due to the lack of previous ophthalmic records , that the abnormal retinal ganglion cell function in this patient may be directly associated with the extensive mnf in the retinal nerve fiber layer rather than a result of the secondary amblyopia that had developed as a result of the extensive myelination . this suggestion may be reinforced from findings in a recent case report where two out of the three patients studied were not only amblyopic but also demonstrated consistent changes in the perg . compared with our patient , the p50 and n95 waves in the perg were recordable but decreased amplitude and increased in latency although the mnfs described were only mainly peri - papillary and not extensive . in conclusion therefore , electrophysiological testing can play an important role in the assessment of visual function in a patient with poor visual acuity associated with extensive myelination , and this diagnostic modality may be able to provide a guide to visual prognosis in such cases .

an observational case report of electrophysiological abnormalities in a patient with anisomyopic amblyopia as a result of unilateral extensive myelinated retinal nerve fibers ( mnfs ) is illustrated . the electrophysiological readings revealed an abnormal pattern electroretinogram ( perg ) but normal full - field electroretinogram readings in the affected eye . the visual - evoked potential was also undetectable in that eye . our findings suggest that extensive mnfs can be associated with electrophysiological abnormalities , in particular the perg , which can aid in diagnosing the cause of impaired vision when associated with amblyopia .

nutrition ( that is , eating food ) is not only a prerequisite for survival ; it is also a frequently enjoyable and social function . as a consequence , when the ability to swallow is impaired ( dysphagia ) people with neuromuscular disease may develop acute respiratory failure and be admitted to critical care for mechanical ventilation . the article by macht and colleagues in a previous issue of critical care throws more light on this area of clinical uncertainty and begins to answer some questions and at the same time opens up further avenues for exploration . the presence of dysphagia is frequently under - recognized and suboptimally managed , especially in patients discharged from critical care back to a medical or surgical ward following extubation . in a retrospective study of patients with acute respiratory failure and pre - existing neuromuscular disease , macht and colleagues reported that 93% of those patients discharged from critical care following extubation and transferred to a surgical or medical ward had post - extubation dysphagia , which in 33% of cases was considered severe . of those with moderate or severe dysphagia , 66% were discharged from the hospital with dysphagia still present . in a similar cohort , without pre - existing neuromuscular disease , 84% had dysphagia , and only 17% were assessed as being severe . in both cohorts , those patients with dysphagia had a prolonged length of hospital stay and increased morbidity ; mortality was not increased in the recent study . as in many retrospective studies , only a minority of all the patients admitted and mechanically ventilated on critical care were included ; 9% in the neuromuscular group and 25% in the non - neurological group were included in the studies . this is a recurring problem with many studies in this area ; the numbers are small , raising questions about generalizability of results . what is the true incidence of post - extubation dysphagia ? could the number with severe dysphagia be higher than suggested , as many patients did not undergo a bedside swallow examination ( bse ) . how common is aspiration , silent or otherwise ? in a small cohort of 35 patients studied by tolep and colleagues , 16 had neuromuscular disorders ; of these 16 , 14 had an abnormal videofluoroscopy ( vf ) ( modified barium swallow ) ; and of these 14 , 4 were aspirating . leder , using fiberoptic endoscopic evaluation of swallowing ( fees ) , noted that 17 out of 52 patients aspirated , the majority silently . aspiration pneumonia is the most common form of hospital - acquired pneumonia and carries a mortality of 20% to 65% . the development of complications , including pneumonia , following aspiration is dependent on many factors , including the physiological condition of the patient , and by definition many of these patients are at the limits of their physiological reserve , the frequency of aspiration , the contents of the aspirate and the quality of mouth care . macht and colleagues are concerned that the bse will not detect silent aspiration , and this is true by definition , but does that matter ? we all aspirate , particularly at night when the frequency of swallowing is greatly reduced or ceases . many studies investigate preselected patients , who have been referred for assessment , whereas those studies investigating un - selected patients do not find any particular association with aspiration . silent aspiration is detected only by instrumentation ( vf / fees ) and , when detected in this way , appears to have no clinically apparent affect and this would not be logistically practical in all patients and should be reserved for those patients who are most at risk of infection or continually develop chest infections or desaturation . this cohort of patients with neuromuscular disease was a heterogeneous population , and how optimal was the management of the dysphagia ( as good clinical management can reduce pulmonary complications ) , or was mouth care very diligent to prevent aspiration pneumonia secondary to infected saliva ? the etiology of post - extubation dysphagia is likely to have multiple factors , including progression of the underlying neuromuscular disease critical care syndrome and trauma to the airway and muscle wasting / weakness . it is possible that this study by macht and colleagues missed significant variation in the occurrence of dysphagia because of the heterogeneity of the cohort or because the subgroups were too small for separate analysis . therefore , future studies in critical care populations should be undertaken prospectively and need to be adequately powered to detect clinically significant differences , and determine predefined sub groups must be identified for analysis . the data collected need to explore all possible etiology factors , including trauma due to a difficult intubation ( 1.5% to 8.5% of intubation are classed as difficult ) , and answer questions such as these : was pre - intubation dysphagia that had not been clinically apparent present , and what was the functional ability of the patient prior to intubation ? this study has begun to answer some of the questions around the complex problem of post - extubation dysphagia in neuromuscular disease . this should spur others on to further elucidate the relationship between extubation dysphagia , neuromuscular disease , and acute respiratory failure . bse : bedside swallow examination ; fees : fiberoptic endoscopic evaluation of swallowing ; vf : videofluoroscopy .

post - extubation dysphagia is a common and serious problem . the presence of neuromuscular disease at the time of intubation is likely to increase this . until recently , the prevalence and the association with length of intubation had not been clarified . results published in this journal suggest that 93% of extubated patients with neuromuscular disease had post - extubation dysphagia , which in 33% of cases was considered severe . the number of days ventilated was the single predictor of severe dysphagia and a consequent prolonged hospital stay . further work to build on these results to unravel the complex interplay between disease , trauma , and other unknown factors will be required .

endobronchial fibroepithelial polyps are a rare neoplasm with only a few case reports in the english literature ( 1 - 4 ) . histologically , these lesions are composed of collagen fibers in a stroma covered by normal respiratory epithelium ( 3 ) . we report a case of a fibroepithelial polyp of the bronchus , of which a lobulating contour is well visualized at computed tomography ( ct ) . however , a non - contrast chest ct scan ( brilliance 6 , philips healthcare , cleveland , oh , usa ) revealed a 1 cm , endobronchial polypoid nodule abutting the anterior wall of the left main bronchus , which had a soft - tissue attenuation of 40 hu . the endobronchial nodule showed a conspicuous lobulating contour that resembled an appearance of a blackberry ( fig . at bronchoscopic examination , a lobulated endoluminal nodule with a glossy surface was arising from the anterior wall of the left main bronchus , resulting in significant luminal narrowing . the polypoid nodule was removed using an electrosurgical snare during rigid bronchoscopy under general anesthesia . at gross examination of the polypectomy specimen , the tumor measured 1.2 1.0 cm in diameter with a lobulating contour and a glossy surface ( fig . histopathologically , the nodule showed marked papillary projections , which resulted in the gross morphology of a lobulating contour of the polyp ( fig it consisted of fibrovascular stroma covered by normal respiratory epithelium , enabling the histological diagnosis of a fibroepithelial polyp ( fig . a fibroepithelial polyp is a common type of tumor in the skin or the genitourinary tract . especially , it is the most common benign tumors of the ureters ( 5 - 8 ) . however , fibroepithelial polyps of the bronchus are rare with only a few case reports in the english literature ( 1 - 4 ) . according to these case reports , it manifests as an endobronchial polypoid nodule that is covered with normal respiratory mucosa , consisting of fibrovascular stroma with or without few inflammatory cells and adipocytic components ( 1 - 4 ) . some reports have classified fibroepithelial polyps as inflammatory , while others have argued that the inflammation is a result of the intermittent respiratory tract obstruction caused by its mass effect ( 4 ) . histologically , fibroepithelial polyps of ureters are known to be predominantly composed of urothelial epithelium and fibrovascular stroma , showing a finger - like or polypoid growth pattern ( 8) . such a papillary growth pattern on histology may result in a lobulating contour of the nodule on gross morphology , which can be readily identifiable on radiological images . the gross appearances of fibroepithelial polyps arising in the tracheobronchial tree and the ureter described in the previous case reports are summarized in table 1 . the majority of appearances of the polyps suggested their lobulating contours , and many of those figures actually shown in reports revealed quite a similar appearance of the nodules to that of our case . although these appearances had not been emphasized in those reports , the lobulating contour of the polyp was clearly identified on ct images in our case , and this feature had been derived from a typical papillary growth pattern of the fibroepithelial polyp . it has been known that an endobronchial nodule can develop in a variety of neoplasms including squamous cell carcinoma , adenoid cystic carcinoma , mucoepidermoid carcinoma , neurogenic tumor , leiomyoma , hamartoma and lipoma , and so on . therefore , we think that an awareness of the implication of such a lobulating contour of an endobronchial tumor might be important in the differential diagnoses of various tracheobronchial polypoid nodules . in summary , we report a case of a fibroepithelial polyp of the bronchus in which a lobulating contour of the endophytic nodule was well identified at ct scan , and we believe that its lobulating shape represents a typical papillary growth pattern of the tumor on histopathology . although endobronchial fibroepithelial polyps are a rare neoplasm , such a characteristic lobulating contour of the polyps might be helpful in the differential diagnosis from other various endobronchial polypoid nodules .

a fibroepithelial polyp of the bronchus is a rare , benign , and endobronchial tumor , histologically consisting of fibrovascular stroma covered by normal respiratory epithelium . we report a case of a fibroepithelial polyp arising from the left main bronchus . on ct , a characteristic lobulating contour of the endobronchial nodule was well visualized , which histopathologically represented a typical papillary growth pattern of the nodule . such a lobulating contour of the nodule might help make a correct diagnosis of this rare disease among other various endobronchial neoplasms .

in our current study , we found that early exponential growth phase cultures of p. aeruginosa elicited the most robust net release and presence of a functional flagellum was essential for this process . forced contact of immotile p. aeruginosa with pmns restored their ability to trigger maximal net extrusion . p. aeruginosa flagellin alone was unable to induce net release . in a genetic complementation study we found that both , motab and motcd loci of p. aeruginosa thus , we identified flagellar swimming motility as a novel microbial factor crucial to pmn activation and net formation . although it is undocumented whether nets are present at early stages of cf lung disease , bacterial motility - fueled net formation likely occurs at this initial phase because early cf clinical isolates of p. aeruginosa typically express flagellum and pmns are also present ( fig . 1 ) . the interaction of pmns with early forms of p. aeruginosa must be critical to determine later progression of cf lung disease . important questions to be answered are why nets released at this early stage would be incapable of clearing p. aeruginosa infection and instead drive bacterial adaption toward an aflagellated , biofilm - forming phenotype . over the course of cf lung disease , p. aeruginosa down - regulates its flagellum expression . in chronic cf airways , p. aeruginosa mainly exist in 3-dimensional , suspension biofilms also called non - attached aggregates ( fig . 1 ) . these suspension biofilms surrounded by pmns represent the characteristic clinical picture in chronic cf airways . biofilms are dynamic structures , and motile , flagellated bacteria likely break free from biofilms in chronic cf , possibly interacting with pmns ( fig . 1 ) . this is supported by recent data showing that p. aeruginosa flagellin is detected in sputa of chronic cf patients . a minor population of flagellated p. aeruginosa in chronic cf airways could also have been marginalized so far because this topic has not been intensely investigated yet , and this population is hard to study and could have been overlooked in the presence of much more abundant biofilm - bound bacteria accumulating well - characterized mutations ( muca , lasr ) . conclusions with respect to the population structure of p. aeruginosa in cf have largely been generalized based on results obtained on single bacterial isolates , although high levels of phenotypic diversity among p. aeruginosa isolates within individual cf patients have already been noted . a small population of flagellated p. aeruginosa could be found in cf airways while most p. aeruginosa are present in form of alginate - producing , elastase - negative bacteria . muca mutations drive the mucoid , biofilm - forming phenotype , lasr mutations contribute to pmn recruitment while outbreaks of flagellated bacteria from biofilms could be mainly responsible for pmn activation and net release . pmns quickly and easily recognize motile , flagellated forms of p. aeruginosa and launch their robust effector mechanisms including net release in response to them . on the other hand , bacterial biofilms therefore , the way motile p. aeruginosa interacts with pmns , the cell type representing most of cells found in chronic cf airways , is likely an important factor in influencing the progression of cf lung disease despite the fact that planktonic forms of p. aeruginosa are outnumbered by those found in biofilms ( fig . 1 ) . overall , we speculate that p. aeruginosa motility - driven pmn activation has clinical relevance not only at initial but also later stages of cf airway disease .

abstractneutrophil extracellular trap ( net ) formation represents a unique effector function of neutrophils ( pmn ) . the mechanism of net release in response to bacteria is largely unknown . we studied the process by which pseudomonas aeruginosa , an opportunistic pathogen , interacts with primary pmns , and found that flagellar swimming motility of the bacterium is essential for inducing net extrusion . cystic fibrosis ( cf ) lung disease is associated with p. aeruginosa infection and pmn - dominated inflammation . although nets are abundant in cf airways , the main factors triggering net release in cf remain unclear . our study implicates that motile p. aeruginosa is a strong net - inducer in cf . in early stages of cf lung disease flagellated , motile isolates of p. aeruginosa are characteristic and their interactions with pmns could lead to net formation . in chronic cf , p. aeruginosa down - regulates its flagellum expression to avoid recognition by the immune system and forms biofilms . flagellated bacteria , however , are released from biofilms and could interact with pmns to form nets . although flagellated forms likely represent only a small fraction of the total p. aeruginosa load in chronic cf , net release induced by them could have a significant impact on inflammation and lung function since flagellated forms trigger the most robust response of the immune system including pmns.overall , we speculate that net formation driven by motile p. aeruginosa could be a novel , significant contributor to pathogenesis at both , early and late stages of cf lung disease .

new , large genomic data sets are providing more in - depth insights into the diagnosis and treatment of disease . in the past decade , new and innovative methods have continued to add value to the underlying data and uncover the secrets of the genome . visual data inspection by experienced researchers is an important quality control element in the analytical process . unfortunately , this part of the process is tedious and time consuming , and the increasing volumes of high - throughput sequencing data of various types and platforms are proving to be a major analytical challenge . here , we report a visualization tool that allows researchers to explore their data at a very rapid speed and significantly reduce the burden of reviewing tens and hundreds of thousands of variant calls . areas with systematic read errors can be quickly identified , and inefficient attempts to verify results in noisy regions can be avoided . alview is a fast and portable visualization tool . the core code interfaces with heng li et al s samtools library1 for parsing bam files . the program is written in platform - independent c. peculiarities specific to an operating system are isolated with if defined ( ifdef ) directives ; so , for instance , when microsoft visual c provides alternate support for a portable operating system interface ( posix ) standard function , a handcrafted , native interface work around is supplied . for graphical user interface ( gui ) frameworks , alview uses win32 interface for windows , the gtk2 interface for linux , and bsd unix - based systems and cocoa for apple mac os x. samtools1 is written to posix standards , but different microsoft visual compilers provide various levels of support for these unix style standards . as a result , the source code for third - party libraries that were modified for windows is provided to facilitate compiling and linking alview on windows . the main code for alview , in the file alviewcore.cpp , is written to be portable between operating systems and emphasizes speed of execution . the code can be compiled as a stand - alone executable and must be linked with the zlib2 and samtools1 libraries . sequence reads are processed via custom samtools callback functions arranged in in - memory data structures and represented by an aesthetic , annotated image . the image is then output to the screen as a native graphics object or to the disk as a standard image format file . alview can also be compiled as a webserver daemon that uses the common gateway interface ( cgi)3 standard . the cgi version produces interactive html output and uses dynamic html54 features , including zoom in by selection via a jquery5 library . the cgi webserver alview version loads a list of permitted - to - access bam files from a user - maintained text file ; so custom lists of bam files of interest are easy to generate and use . the source code is free and open to modification so that users and local system operators can implement their own security . the alview cgi webserver version provides modifiable url access , so that , for instance , cells in a spreadsheet can link to viewable results for any sample or location . stand - alone alview accepts parameters that specify bam file name and genomic coordinates . invoking alview in a script can create a slideshow of interesting regions . for example , fields in a single nucleotide polymorphism ( snp ) detection output file can be used to specify a series of calls to alview to generate images for each purported polymorphism or mutation . the burden of reviewing ten and hundreds of thousands of mutation calls can therefore be significantly reduced . the source code is available at github.6 the readme file there points to links for selected executables and complete download packages that include the associated reference genome data . a live webserver version of alview for examining public human cancer short - read datasets is available at https://cgwb.nci.nih.gov/cgi-bin/alview . alview source code and executables for several operating systems are available at the national cancer institute ( nci)/national cancer informatics program s ( ncip s ) github site : https://github.com/ncip/alview . nci retains the copyrights to national cancer institute and associated images , which may not be used in forked projects . alview provides a solid substructure that allows for various types of access to short - read data across different operating systems . figure 1 . demonstrates the various navigation and information buttons available in the web version of alview and shows how selection via mouse provides zoom in capabilities . alview is a trim , fast , precise tool and complements existing programs such as the integrated genomics viewer ( igv),7 bamview,8 and gbrowse 2.0.9 the benefits of alview are extreme speed and a sharp focus on exploring short reads . different implementation philosophies can influence memory usage and performance but provide useful alternative paths to solving similar problems . igv provides much more functionality than alview by supporting many other input file types other than bam sequence read files . igv s java implementation provides write once , run anywhere portability via implementations of the java virtual machine . alview s implementation relies on low - level operating system and native gui toolkit api calls . igv requires registration for download for running off of disk , whereas alview does not . desktop igv may require internet for full , easy , simple operation , whereas alview does not require network connection ( though it may call user - invoked external webpages ) . 7 intel core i52400 cpu at 3.10 ghz and 8 gb ram , restarts of igv v2.3 took from 12 to 18 seconds . restarts of alview took a small fraction of one second . for a small view of a genomic region , the java platform se binary for igv took up 292 mb , while alview took up 11 mb .

the name alview is a contraction of the term alignment viewer . alview is a compiled to native architecture software tool for visualizing the alignment of sequencing data . inputs are files of short - read sequences aligned to a reference genome in the sam / bam format and files containing reference genome data . outputs are visualizations of these aligned short reads . alview is written in portable c with optional graphical user interface ( gui ) code written in c , c++ , and objective - c . the application can run in three different ways : as a web server , as a command line tool , or as a native , gui program . alview is compatible with microsoft windows , linux , and apple os x. it is available as a web demo at https://cgwb.nci.nih.gov/cgi-bin/alview . the source code and windows / mac / linux executables are available via https://github.com/ncip/alview .

the first symptom of soft rot on apple fruit was a water - soaked appearance to the affected tissue . the diseased parts later disintegrated into a mushy mass of disorganized cells that sloughed off . white mycelia formed on infection sites of apples and gradually covered the fruit with tufted whisker - like gray sporangiophores and sporangia ( fig . sporangiospores , sporangia , and sporangiophores were observed under a light microscope ( table 1 ) . the fungal colonies that grew on potato dextrose agar were initially white and cottony , then became heavily speckled with sporangia , and finally became brownish - grey to blackish - grey and spread rapidly with stolons fired at various points to the substrate by rhizoids ( fig . the optimum temperature for mycelial growth was 30 , with good growth still apparent at 37. sporangiospores were unequal , numerous , irregular , sub - globose or oval , angular with striations , and 4~8 m ( fig . smooth - walled , simple or branched , non - septate , long , and arose from stolons opposite rhizoids usually in groups of 3~5 or more . sporangia were globose , white at first , and then turned black with many spores , mostly 40~200 m ( fig . 2c ) . columella were globose to sub - globose , pale brown , and mostly 85~110 m ( fig . twelve apple fruits were artificially inoculated with a representative fungus using the wound infection method . a conidial suspension ( 0.1 ml ; 3 10 conidia / ml ) of the causal fungus was placed on the surface of apple fruit . the inoculated fruit was kept in a moist chamber with 100% relative humidity at 30. after a 3 day incubation , the same fungal symptoms were reproduced : soft rot was observed on inoculated fruits that was identical to symptoms observed at the commercial markets ( fig . 1c and 1d ) . to confirm the identity of the causal fungus , the its rdna of the isolate was amplified and sequenced using its1 ( 5'-tccgtaggtgaacctgcgg-3 ' ) and its4 primers ( 5'-tcctccgcttattgatatgc-3 ' ) , as described by white et al . . a phylogenetic analysis was performed using mega4 with the neighbor - joining method and the tajima - nei distance model . previously published its sequences from r. oryzae strains were included for reference , and mucor miehei ( genbank accession no . the present isolate was placed within a clade comprising r. oryzae references isolates ( fig . soft rot of apple caused by r. stolonifer has been reported previously , but soft rot caused by r. oryzae has not been recorded in korea . the representative culture of the causal fungus was deposited in the korean agricultural culture collection ( kacc 45815 ) , national academy of agricultural science , suwon , korea . based on the mycological characters , molecular data , and pathogenicity testing of the host plant , the fungus was identified as rhizopus oryzae went & prisen geerligs .

soft rot in apple caused by rhizopus oryzae was found for the first time in korea . a detailed description of the specimen is given along with its internal transcribed spacer rdna sequence . the fungus was identified as rhizopus oryzae based on the mycological characteristics , molecular data , and pathogenicity testing .

hematohidrosis is known to be precipitated by stress , strain , or any sort of exertion , and it may occur in individuals with underlying bleeding disorders . it may occur in individuals suffering from extreme levels of stress . around the sweat glands , there are multiple blood vessels in a net - like form , which constrict under the pressure of great stress . then , as the anxiety passes , the blood vessels dilate to the point of rupture and goes into the sweat glands . as the sweat glands produce a lot of sweat , they push the blood to the surface , which comes out as droplets of blood mixed with sweat . we hereby report a case where bloody sweat discharged from the forehead , face , and body episodically in a healthy young girl who did not have any underlying disorders . a 12-year - old girl visited us with a history of bleeding from the intact skin over the forehead , scalp , cheek , nose , and trunk [ figures 1 and 2 ] for the last 2 years . the bleeding occurred in episodes , once or twice a day , sometimes more frequently , especially on waking up in the morning . no preceding history of stress or anxiety and no preceding episode of tingling sensation were found . each episode started with mild watery secretion over the forehead or other body parts , followed immediately with bright - red colored secretion . each episode lasted for about 10 - 15 min , and the patient remained perfectly alright during the post - episode period until the next episode . no history of ingestion of any anticoagulants , dyes , or other drugs was obtained from her . she did not have any history of major medical or surgical illness in the past . sweat admixed with blood from the right side of the face sweat admixed with blood from nose her general physical examination and systemic examinations did not reveal any abnormality . the skin over the forehead was normal . on gross examination , the secretion was bright - red in color , less viscous than blood , and it was not frank blood . on collection of the secretion and examination of its smeared preparation under a microscope , plenty of erythrocytes were observed ; the sample was benzidine test positive [ figure 3 ] and alkaptonuria negative . her routine hemogram , blood counts , platelet count , bleeding time ( 2 min ) , clotting time ( 3 min 30 sec ) , active partial thrombin time ( 25 sec ; normal range 24 - 32 sec ) , prothrombin time , liver function tests , and renal function tests were within normal limits . peripheral smear of the secretion showed multiple red blood cells ( rbcs ) along with numerous gram - positive cocci and bacilli [ figure 4 ] . benzidine test of the secretion , confirming presence of blood peripheral smear of the secretion showing rbcs and numerous cocci and bacilli psychiatric analysis revealed intelligent quotient ( i.q . ) between 60 and 70 and a loss of insight . the patient was given atropine transdermal patch over the involved sites and she noticed gradual improvement in both the severity and frequency of the episodes . we followed the patient for 2 months after stopping the atropine treatment . during this period hematohidrosis also known as hematidrosis , hemidrosis , and hematidrosis is a condition in which capillary blood vessels that feed the sweat glands rupture , causing them to exude blood ; it occurs under conditions of extreme physical or emotional stress . various causative factors have been suggested by holoubek , like component of systemic disease , vicarious menstruation , excessive exertion , psychogenic , psychogenic purpura , and unknown causes . acute fear and intense mental contemplation are the most frequent causes , as reported in six cases in a study . in our case , one hypothesis proposed for etiopathogenesis of hematohidrosis , as suggested by some authors , is that multiple blood vessels present in a net - like form around the sweat gland constrict under pressure of stress . as the anxiety passes out , the blood vessels dilate to the point of rupture . the blood goes into the sweat glands , which push it along with sweat to the surface , presenting as droplets of blood mixed with sweat . severe mental anxiety activates the sympathetic nervous system to invoke stress - fight reaction to such a degree as to cause hemorrhage of the vessels supplying the sweat glands into the ducts of the sweat glands . they concluded that pathological basis for hematohidrosis might be distinctive vasculitis , but direct immunofluorescence study did not reveal any abnormality in our case . biopsy during symptom free period did not reveal any blood - filled vascular spaces , intradermal bleeding or abnormality in hair follicle , sebaceous , or sweat glands . benzidine test is an important tool in diagnosis , where hemoglobin in blood reacts with hydrogen peroxide - liberating oxygen , which then reacts with organic reagent producing a green to blue colored compound . we believe that a further insight in the etiopathogenesis may help develop more comprehensive management protocol of this rare and unusual condition . 1 . in our case , no underlying precipitating factors such as stress , strain , or any bleeding disorder was identified .

hematohidrosis is a very rare condition in which an individual sweats blood . it may occur in an individual who is suffering from extreme levels of stress . various causative factors have been suggested like component of systemic disease , vicarious menstruation , excessive exertion , psychogenic , and unknown causes . fear and intense mental contemplation are the most frequent causes . it may also occur in bleeding disorders . we here report a case where bloody sweat was discharged from the forehead , face , and body episodically in a 12-year - old healthy girl with no bleeding disorder or any other underlying cause . all investigations done were within normal limits , except low intelligent quotient and loss of insight . the patient was given atropine sulphate transdermal patch with marked improvement in severity .

necrobiotic xanthogranuloma ( nxg ) was first described as a separate entity by kossard and winkelmann in 1980 . the disease is characterized by multiple , plaque- like , yellow - brown lesions , with a predilection for the periorbital area ( spectacle - like distribution ) . about 100 cases have been reported in world literature . to our knowledge , this is the first case of isolated cutaneous nxg being reported from india . a 65-year - old female presented with a 3-year history of gradually enlarging , periorbital , asymptomatic lesions . however , it recurred in a few months and was accompanied by new larger nodules . the patient 's medical history was not pertinent.on examination , there were multiple , yellowish brown , firm nodules distributed symmetrically in the periorbital area . the size of the nodules varied from 3 to 7 cm in their widest dimensions . the surface showed a few telangiectasias and mild atrophy [ figures 1 and 2 ] . yellow brown firm periorbital nodules superficial telangiectasias and mild atrophy with this history and clinical findings , a provisional diagnosis of a xanthomatous disorder was made . histopathological examination of a surgical wedge biopsy showed ill - defined large foci of dense granulomatous infiltrate of large histiocytes and histiocytic foamy cells with lymphocytes and few plasma cells . several histiocytic giant cells resembling touton giant cells were present [ figures 3 and 4 ] . however , typical findings of necrobiosis were not seen . dense granulomatous infiltrate of histiocytes laboratory investigations were normal for blood counts , electrolytes , glucose levels , lfts , except for s.triglycerides and s.cholesterol levels which were mildly elevated . immunohistochemistry was positive for cd 68 [ figure 5 ] and negative for cd 1a and s100 . thus , a final diagnosis of isolated cutaneous nxg without paraproteinemia or associated disorders was made . the patient was maintained on a regular follow - up of every 15 days for the first 2 months and then once a month for 9 months . nxg is a rare , progressive , histiocytic disease that features destructive cutaneous lesions , a close association with paraproteinemia , and multisystem extracutaneous manifestations . nxg is a disease of adults with an average age of onset in the sixth decade . nxg begins as papules and nodules that slowly enlarge into indurated plaques ranging from 0.5 to 25 cm . characteristic lesions have a red - orange or yellow color as well as telangiectasias , atrophy , scarsand ulcers . the most common site of involvement is the face , particularly the periorbital region ( 85% of cases ) ; the trunk and proximal extremities are other affected sites . ophthalmological complications affect approximately 50% of cases and include orbital masses , conjunctival involvement , keratitis , scleritis , ectropion , uveitis and blindness . nxg may involve other extracutaneous sites , including the heart , lungs , kidneys , liver , spleen , intestines , skeletal muscle and central nervous system . paraproteinemia is closely associated with nxg , with approximately 80% of patients demonstrating a monoclonal gammopathy of igg- on serum protein electrophoresis ; furthermore , 10% of these patients develop multiple myeloma . hepatosplenomegaly , lymphadenopathy , an increased erythrocyte sedimentation rate , leukopenia , hypocomplementemia , thrombocytopenia and cryoglobulinemia are other common findings . theories regarding pathogenesis include deposition of immunoglobulins and lipid complexes with a foreign body giant - cell reaction and monocyte activation with intracellular lipid accumulation . histopathological examination shows a palisading xanthogranuloma with areas of necrobiotic , degenerated collagen that infiltrates the mid - dermis with extension into the subcutaneous fat . multiple xanthomatized histiocytes , touton giant cells , other bizarre giant cells , cholesterol clefts and lymphoid follicles are often present within granulomas . improvement in skin lesions has been seen with low - dose chlorambucil , melphalan with or without prednisolone , methotrexate , localized radiotherapy , systemic glucocorticoids , interferon -2a , and plasmapheresis with hydroxychloroquine . topically nitrogen mustard , bncu , intralesional corticosteroids and intravenous immunoglobulin have also been tried . surgical excision has also been advocated . in our patient , we decided to surgically excise the lesions in view of absence of systemic involvement .

necrobiotic xanthogranuloma ( nxg ) is a very rare , progressive variant of non - langerhans cell histiocytosis . it is known to be associated with multisystem involvement and paraproteinemias . a 65-year - old female presented with chronic , slowly growing , asymptomatic periorbital nodules . the lesions had recurred after local excision elsewhere . no systemic involvement or paraproteinemias were detected . a provisional diagnosis of isolated cutaneous nxg was made which was confirmed by histopathology and immunohistochemistry staining . the lesions were surgically excised with excellent cosmetic and functional results . there was no recurrence over a period of 9 months . to our knowledge , this is the second case of nxg reported from india and the first without any systemic manifestations .

atrial fibrillation ( af ) with progressive atrial dilation combined with inadequate anticoagulation exhibits a high risk of thromboembolic events . we present the unusual case of a ball thrombus originating from the left atrium ( la ) with remaining continuity to the left atrial appendage ( laa ) causing syncope and cerebral ischemia . an 81-year - old female presenting with syncope and aphasia was admitted to the emergency department of our hospital . the patient was diagnosed with permanent af four years prior to presentation and was managed with rate - control therapy . however , for unknown reasons , there was no anticoagulation at the time of presentation . so far , except for af and arterial hypertension ( treated by angiotensin receptor blockers and diuretics ) , the patient was free from other independent risk factors for la thrombus formation ( no previous cardiac or embolic history ) . the head computed tomography scan of the patient , who was normotensive at this time , revealed acute focal ischemia at the left temporal lobe . transthoracic echocardiography showed the presence of a 4 3.5 cm measuring , free - floating , nonhomogeneous mass in the la . there was a rate - dependent dislocation of this mass to the mitral valve orifice ( fig . this caused intermittent obstruction of the left ventricular outflow tract , and was associated with presyncopal symptoms . in addition , echocardiography demonstrated severely dilated atria , presumed to be due to long - standing af . the lv showed concentric hypertrophy with a preserved ejection fraction . after excluding significant coronary stenosis by coronary angiography , the primary operative finding was a spherical purple gelatinous mass measuring 4.5 4 cm , partially free floating in the la , but attached to the laa by a thin bridge ( fig . excision of the mass including the shaft was performed , followed by suture ligation of the laa . histopathological examination confirmed a layered , partly necrotic , mostly organized , parietal thrombus ( not displayed ) . surgical inspection confirmed the echocardiographic findings of moderate mitral regurgitation , so additional valve repair was not indicated . moreover , a maze procedure was considered , but because of the patient s critical status and cerebral ischemia , it was deferred . at the 6-month follow - up evaluation , there were no signs of relevant postoperative complications or neurologic postischemic sequelae . the patient was anticoagulated during this period , and no new thrombus formation was identified on repeat echocardiographic examination ( fig . in this report , we present the case of a patient with syncope and acute cerebral ischemia due to embolization from an unusual la ball thrombus with preserved attachment to the laa not visible on transthoracic echocardiography . thrombus formation and embolization are the most threatening complications of an inadequate anticoagulation in the presence of af . as in this case , the presence of a mass in the la often presents a diagnostic challenge for cardiologists . echocardiography can help distinguish between solid benign or malignant mass , and organized or disorganized thrombus formation . shape and echogenicity are important characteristics that help to distinguish among these potential etiologies of atrial mass . to date , there have been few reports of the presence of an la ball thrombus without mitral valve disease.13 most frequently , the presence of a ball thrombus suggests mass formation by continuous increase in size independently from the la wall . therapeutic decision - making and optimal treatment strategy selection , ie , thrombolysis versus surgical intervention , require prompt evaluation of clinical parameters and evaluation of the thrombus morphofunctional characteristics . several authors have reported recurrent ischemic episodes after thrombolysis in the presence of la thrombus.4,5 when a free - floating thrombus is identified in the la , emergent cardiac surgery for embolectomy is often required because of the high risk for sudden circulatory collapse or systemic embolization.3,6 as in the case we present here , accurate trans thoracic echocardiographic evaluation of the mass provides critical information required for initiating timely therapeutic procedures.7 continuous anticoagulation treatment for patients with af remains extremely important to prevent embolic complications.8 selection of the optimal treatment strategy for atypical la thrombus is a challenging clinical scenario . unfortunately , no guidelines exist to direct the selection of the most appropriate evaluation and treatment for these cases . the use of transthoracic echocardiography in emergency situations can be a cost - effective and time - saving tool impacting immediate therapeutic decision - making .

few cases of a left atrial thrombus without mitral valve disease have been reported . we present an unusual case in which a patient presented to the emergency department with syncope and acute cerebral ischemia caused by a ball thrombus originating from the left atrium ( la ) . an emergency bedside echocardiogram showed the la ball thrombus intermittently obstructing the mitral orifice and , at times , compromising the left ventricular outflow tract . this thrombus was determined to be the source of cerebral embolization resulting in acute ischemia . surgical excision of the mass was performed . at operation , the thrombus was found to be tethered to the left atrial appendage . this tethering was not apparent on the echocardiographic images , where the thrombus appeared to be free floating . this case demonstrates the utility of transthoracic echocardiography in establishing the etiology of emergent conditions seemingly unrelated to acute cardiac disease , in this situation a neurologic presentation with syncope and cerebral ischemia .

fibrous dysplasia is not hereditary in nature and it caused by mutation in the gnas1 ( guanine nucleotide binding protein , alpha stimulating activity polypeptide ) gene ( 20q13.2 ) and this gene encodes a g - protein which results in overproduction of camp in the affected tissues . furthermore , there is increased the proliferation of melanocytes thus results in cafe - au - lait spots . fibrous dysplasia has three clinical patterns namely monostotic , polyostotic , craniofacial form . about 3% of lesions associated are with skin pigmentation and hyperfunctioning endocrine disorders known as the mccune albright syndrome . the skull base and proximal metaphysic of femora are two sites most commonly involved . in the skull fd involves skull bases and facial bones . in childhood fd presents as facial asymmetry or a bump , but symmetric expansion of malar prominences and/or frontal bosses may be seen . due to abnormal growth and deformity of craniofacial bones may result in encroachment on cranial nerves . female patients experience increased pain level during pregnancy and during the menstrual cycle because of estrogen receptors found in fd . bianco et al . demonstrated that fd is a disease of bone marrow stromal cells ( bmsc ) . the bmscs form structural framework upon which hematopoiesis occurs in the bone marrow and a subset of bmsc are multipotent stem cells capable of differentiating into multiple cells including osteoblasts , osteocytes , chondrocytes , bone marrow adipocytes and other cells . in fd bmsc differentiate along osteogenic lineage , but differentiation is arrested and instead undergo proliferation giving rise to fibro - osseous masses of tissue . g - protein is central in cell originating pathway leads to the generation of intracellular second messenger , camp / protein kinase a signaling . all mutation in gs alpha identified in association with fd is the 201 position . in > 95% cases arginine is replaced by either cysteine or histidine ( r201c or r201h ) . this result in inhibition of intrinsic gtpase activity of gs alpha protein and it is this aspect that leads to constitutive , ligand - independent generation of intercellular camp . gene amplification techniques such as polymerase chain reaction is now possible to test for genetic mutation in peripheral blood samples . actively formed lesions in adolescents have greatly increased isotope update that corresponds closely to radiographic extent of the lesion . some characteristic feature is bar - shaped pattern , whole - bone involvement and close match between the size of the lesion on radiograph and the size of the area of uptake . the extent of the lesion is visible clearly on computed tomography , and cortical boundary is depicted more clearly than radiograph . the thickness of cortex , endosteal scalloping and periosteal new bone reaction and homogeneity of the poorly mineralized lesional tissue are well demonstrated . delicate trabeculae of immature bone with no osteoblastic rimming enmeshed within a bland fibrous stroma of dysplastic spindle - shaped cells without any cellular features of malignancy . variable number of immature , nonstress oriented , disconnected dysplastic trabeculae floating in a sea of immature mesenchymal cells that have little or no collagen about them . change is noticed . the malignant transformation rate is unknown , but it is likely to be not > 1% . cancer is more likely to occur in polyostotic disease , and most common histological types were osteosarcoma , fibrosarcoma and chondrosarcoma . there are also reports suggesting that the malignant transformation may be more common in mazabraud 's syndrome ( fd in association with intramuscular myxomas ) . the findings in various studies showed that high dose intravenous pamidronate decreases pain and the markers of bone metabolism .

fibrous dysplasia ( fd ) is a benign intramedullary fibro - osseous lesion . fd is a bone developmental anomaly characterized by replacement of normal bone and marrow bone by fibrous tissue . it involves any of the bones as single lesion ( monostotic ) or in multiple bone lesions ( polyostotic ) or all of the skeletal system ( panostotic ) . long bones are most commonly involved , which mostly identified incidentally and clinically appears asymptomatic . clinical , radiographical and histopathological findings will help in confirming the lesion . there are many treatment option available , but still management of fd remains challenging .

a 65-year - old woman was referred to our department because of a recently diagnosed left atrial ( la ) mass . she presented with a five - month history of fever , malaise , and myalgia and had been diagnosed with ventricular septal defect ( vsd ) two years earlier . on physical examination , two - dimensional transthoracic echocardiography showed an la cyst ( 2423 mm ) that was attached to the interatrial septum and detected the trace of blood flow at the margin of the cyst ( fig . 1 ) . the vsd was a small perimembranous type and the pulmonary blood flow to systemic blood flow ratio ( qp / qs ) was 1.2 . the patient underwent surgical treatment . the cystic mass attached to the left side of the interatrial septum was white - pink - yellowish and oval in shape . the cystic mass was widely excised with the atrial septum , leaving the defect in the atrial septum , and the small vsd was closed with two pledgeted sutures without a patch . the defect on the atrial septum was closed with an artificial patch ( polytetraflouroethylene , gore - tex patch ; wl gore & assoc , flagstaff , az , usa ) . histologically , the mass consisted of myxoid cells in abundant loose and myxoid stroma , and the tumor cells were round or polygonal with eosinophilic cytoplasm ( fig . la myxoma is usually a solid , round or polypoid mass without a cystic structure attached to the interatrial septum . the size of the mass ranges from 1 to 15 cm and is usually about 5 to 6 cm . the mass has solid content with a consistency varying according to the amount of collagen it contains . the preoperative diagnosis of a cardiac mass has been enabled by echocardiographic examination . in this case , it was difficult to establish the diagnosis of the mass preoperatively because of its cystic structure without any content . however , we believed that it was not a simple cyst and that surgical excision was needed because she had had constitutional symptoms . myxomas originated from the mesenchymal cells of the septal endocardium and can present as villous , papillary , sessile or pedunculated pattern and were usually divided into round and polypoid type and the majority of them are solid . the types of cystic masses that can appear in la are as follows : myxoma , hydatid cyst , and interatrial septal aneurysm [ 5 - 8 ] . owing to their many structural similarities , it is difficult to establish an accurate diagnosis using preoperative echocardiography . major complications like stroke , rupture , and hemodynamic problems may develop if they are not removed . therefore the surgical excision of an la cyst is reasonable for establishing a definitive diagnosis and preventing sequelae .

cardiac myxomas are the most common primary benign tumors of uncertain etiology . they usually present as polypoid or oval - shaped masses projecting into a heart chamber from the interatrial septum and have a soft , gelatinous consistency without a cystic structure . we report a case of left atrial myxoma with a single cystic form .

prognosis of advanced or metastatic pancreatic adenocarcinoma is poor , with a median overall survival between 6.8 and 11 months for all histological types . herein we report a case of a patient treated by the combination of gemcitabine plus oxaliplatin ( gemox regimen ) with a complete radiological response . a 50-year - old male complained of epigastric and right hypochondriac region pain in april 2001 . a ct scan showed multiple cystic tumors in the liver and spleen , a pseudocystic tumor on the pancreatic isthmus and an unlimited mass on the pancreatic corpus and tail with peripancreatic effusion . the octreoscan and tumor marker ca 19.9 concentration were normal . in may 2001 , chemotherapy with gemcitabine 1,000 patient experienced grade 1 urticaria at cycle 4 and grade 2 thrombopenia , justifying a 75% dose reduction at cycle 7 . evaluation after 4 cycles showed partial response with a 50% reduction of the liver and spleen tumors . after 9 cycles , a complete tumor response was obtained . since 2002 , physical examination and biology have remained normal . the initial biopsy was reanalyzed , and the initial diagnosis was confirmed . at the age of 61 years , a localized prostatic adenocarcinoma was diagnosed , treated by radical prostatectomy . in 2013 , a personal and family history of breast cancers ( sister at 34 years , mother at 60 years and the grandmother ) and a bladder cancer ( brother ) led to a genetic consultation with brca1 and brca2 genetic testing . the patient as well as a cousin and his two sons were found to carry a brca2 mutation . in january 2015 , the patient had no clinical symptoms , a sequelary segmentary portal hypertension and well - epithelialized varicose vein on gastroscopy and he was relapse free . the mean age at diagnostic is 60 years , with a male preponderance ( 65% ) . the most common diagnosis is due to mass effect such as abdominal pain , jaundice or weight loss . this tumor is often associated with pancreatic panniculitis ( weber - christian disease ) , perhaps due to high serum lipase concentrations . data on chemosensitivity are only available from case reports . different regimens of chemotherapy ( gemcitabine , lv5fu2 , oxaliplatin , cisplatin , irinotecan , docetaxel and paclitaxel ) were tested alone or in combination . nevertheless , the combination of gemcitabine plus oxaliplatin was not investigated . objective response rates were described with folfirinox , folfox , lv5fu2-gemcitabine , lv5fu2 , weekly paclitaxel and lv5fu2-cisplatin . however , no complete response was observed . acc is associated with a better prognosis than ductal adenocarcinoma at all stages of disease . in metastatic disease , schmidt et al . described a 5-year overall survival rate of 17.2% versus 2.8% for ductal carcinoma . in unresected patients , wisnoski et al . reported an overall survival of 25 versus 3 months . the longest survival observed in the literature was 11 years compared to 14 years in our patient . brca2 mutations are known to be associated with pancreatic cancer , representing 2% of the total pancreatic cancer cases . nowadays , there are ongoing trials testing parp inhibitors that show promising results with regard to platinum - based chemotherapy regimens . we reported the first case of metastatic acc treated with the gemox regimen . despite the lack of study in these patients , a chemotherapy regimen with oxaliplatin seems to be the most effective combination with long survival . in these patients

pancreatic acinar cell carcinoma ( acc ) is a rare entity . herein we present the case of a 50-year - old male patient with an unlimited mass on the pancreatic corpus and tail with peripancreatic effusion and multiple metastases in the liver and spleen . a liver biopsy showed a pancreatic acc . the patient received 9 cycles of gemcitabine plus oxaliplatin ( gemox regimen ) , which had to be stopped because of a persistent grade 2 neuropathy . a ct scan showed complete response after 14 years . at the age of 61 years , a localized prostatic cancer was diagnosed , treated by prostatectomy . the patient carried a brca2 mutation . none of the precedent case reports describe a chemosensibility to the gemox regimen . in spite of the lack of study in these patients , chemotherapy with oxaliplatin seems to be the most effective . long survival can be expected .

the migration of an encircling silicone band through a rectus muscle is a rare postoperative complication associated with the scleral buckling procedure . however , both kreis et al . and tan et al . have reported cases of this rare complication . here , we describe what we believe to be the first reported case of a scleral buckling - associated encircling silicone band that migrated through the rectus muscles , despite the band being securely surgically fixed to the sclera in the scleral tunnel ; i.e. , a review of the video recording of the surgery confirmed that the encircling band had been placed under the medial rectus muscles during the scleral buckling surgery . a 58-year - old man presented with a rhegmatogenous retinal detachment in his left eye . pars plana vitrectomy with the placement of an encircling silicone band ( # 240 ; mira , inc . , waltham , ma . , usa ) the solid silicone band was placed under the four rectus muscles and surgically fixed in a scleral tunnel at all four quadrants ( fig 1 ) . the scleral tunnels were prepared by making two vertical incisions of one - half of the scleral thickness , and then performing a lamellar dissection with a surgical blade . the anterior extent of each tunnel was 2.02.5 mm posterior to the rectus muscle insertions . one year later , the patient complained of a mild foreign body sensation in his left eye . upon examination , the encircling band was found to be exposed on the nasal side of conjunctiva next to the limbus ( fig 2 ) . the patient did not report any diplopia , and the movement of the associated eye in the field of the medial rectus muscle was found to be normal . two weeks later , the encircling silicone band was surgically removed without any complications . during that surgery , it was noted that the encircling band had cheese - wired through the medial rectus muscle , which had become reattached close to its original insertion with no thinning . at the patient 's most recent follow - up , although it has been reported that a solid silicone band can cheese - wire forward through the rectus muscle postoperatively [ 3 , 4 ] , the underlying mechanism of that migration has yet to be elucidated . it has been suggested that a migration of an encircling band may occur if the band is embedded too deeply , placed anterior to the equator , and not securely anchored to the sclera . in the case shown in this present study , the encircling band was fixed in the scleral tunnel at all four quadrants . a previous study has shown that as with the use of sutures , the use of a scleral tunnel is an alternative and secure method of anchoring an encircling band . however , in the case shown in this present study , we theorize that the band migration possibly resulted from the scleral flaps being thin . an alternative explanation as to why the band migrated is that it might have been placed over the rectus muscle . however , a review of the video recording of the surgery confirmed that the encircling band had actually been placed beneath the muscle . it has been reported that migrating bands can induce ocular motility disturbances [ 3 , 4 ] . tan et al . reported a case with ocular motility disturbances in whom reattachment of the cheese - wired lateral rectus muscle was not observed . in the case shown in this present study , the cheese - wired medial rectus muscle was found to have become reattached close to its original attachment site . we theorize that this was the reason why the ocular motility in our patient was preserved . reportedly , the slow process of cheese - wiring migration through rectus muscle allows sufficient time for the muscle to reattach spontaneously behind the migrating band . in conclusion , the findings of this study show that migration of an encircling element can occur within 1-year after placement , and with subtle symptoms such as foreign body sensation . to prevent cheese - wiring migration , a scleral tunnel that is both deep and long enough to securely fix the silicone band even when an encircling silicone band is surgically secured around the eye , periodic and detailed postoperative follow - up examinations and careful surgical procedures should be performed to ensure no migration of the band .

the migration of an encircling silicone band through a rectus muscle is a rare postoperative complication associated with scleral buckling surgery for retinal detachment . in this present study , we describe what we believe to be the first reported case of a patient who experienced postoperative migration of an encircling silicone band through the rectus muscle , despite the band being surgically secured to the sclera in a scleral tunnel . a 58-year - old man presented with a rhegmatogenous retinal detachment in his left eye . pars plana vitrectomy was performed with the placement of an encircling silicone band , and the patient 's retina was successfully reattached . one year postoperatively , the encircling band became exposed on the nasal side of the conjunctiva next to the limbus without any symptoms . two weeks later , the exposed encircling band was surgically removed without any complications . the findings of this study show that even when an encircling silicone band is surgically secured around the eye , periodic and careful postoperative follow - up examinations should be performed to ensure no migration of the band .

certain colorectal conditions , such as ulcerative colitis and diffuse polyposis of the colon and rectum , and congenital intestinal abnormalities , such as total colonic aganglionosis , require a proctocolectomy and some type of intestinal restoration 1 . although these procedures are technically demanding for surgeons , they are more challenging for the patients following the restoration of intestinal continuity . frequency of bowel evacuations typically increases dramatically postsurgery , contributing to induced metabolic derangements 2,3 . we present a case of juvenile polyposis coli , initially treated with a subtotal colectomy and ileostomy , followed by a proctectomy , ileal - j - pouch and serial transverse enteroplasty ( step ) of the distal ileum 4 . an eight - year - old female patient was referred to our department with a chronic history of unremitting bloody diarrhea , weight loss , anemia and intractable rectal prolapse . there was no other significant past medical history or family history and her vaccinations were up to date . the initial clinical examination revealed a pale child who was underweight for age with mild clubbing of the fingernails . the abdominal examination was unremarkable , and the rectal examination was unnecessary because the cause of the prolapsed rectum was obvious , with a presentation of multiple polyps ( fig1 ) . blood tests confirmed clinical evident anemia with a hemoglobin of 7 g / dl , and her albumin level was 19 rectal polyposis was ratified by sight ( fig1 ) , and total colonic involvement was suspected on the contrast enema ( fig2 ) and was substantiated by colonoscopy ( fig3 ) and by the subtotal colectomy specimen ( fig4 ) . the histological diagnosis of hamartomatous juvenile polyposis was confirmed from the colonoscopic biopsies and the operative specimen . despite the initial temporizing approach of subtotal colectomy and ileostomy , anemia and protein - losing enteropathy persisted . the patient was discharged with dietary supplementation , and she had regular outpatient follow - up . the child s nutritional status improved over time , and she had albumin levels consistently over 36 a proctectomy was performed to remove the residual rectal polyps , and the ileostomy was taken down and fashioned as a j - pouch ( fig5 ) . multiple serial enteroplasties ( fig6 ) starting just above the pouch over a length of approximately 80 cm was performed , followed by a hand sewn ileo - anal anastomosis . the postoperative course was uneventful , and the patient was discharged 2 weeks later . at that stage , she was passing two soft stools per day without any medication or dietary restrictions . the follow - up at 3 months postsurgery confirmed a well - child who had gained 1.5 kg . she had returned to school and continued to pass one to two soft stools per day . restorative proctocolectomy has replaced disparate surgical options such as a hartmann s procedure and permanent ileostomy or ileo - rectal anastomoses because of their technical inadequacy , inability to improve quality of life and persistent cancer risk 5,6 . it removes the diseased intestine , thereby improving quality of life and reducing cancer risk 3 . intestinal leaks and pouchitis are major morbidities that might result in a long term or permanent ileostomy . an increased frequency and volume of enzyme - rich liquid stools although dietary restrictions and opioids might temporarily reduce the symptoms , they fail to cure these patients 7,8 . in these cases , the postoperative quality of life is indisputably not better than the pre - operative status . it is well reported that patients affected by severe short bowel syndrome benefit from the step procedure , because such an intervention can significantly reduce the frequency of stooling 913 . such large volumes must therefore have time and an appropriate intestinal length for water , electrolytes and other nutrients to be absorbed . this process is expected to occur within the j or other pouch configurations following restoration 14,15 . these pouches are quickly filled , and they easily surge ; the anal sphincter does not effectively counteract and contain the volume , which is usually controlled by the ileocaecal valve . large amounts of nutrients , electrolytes and water are wasted because evacuation is inevitable and continence training is not possible . step is a technique for lengthening the intestine in short bowel syndrome by interrupting the intestinal lumen thereby also slowing peristalsis . this procedure allows time for the nutrients , water and electrolytes contained within the intestinal lumen ( and distant from the distal anal anastomosis ) to be absorbed 4,11 . the outcome of our technical option was rewarding because the stool frequency and consistency were immediately in an acceptable range of two to three times per day and a soft consistency , respectively . this outcome was achieved without the use of opioids or the need for dietary restrictions . the child tolerated an unrestricted hospital diet and was discharged after 2 weeks when we were certain of her bowel habits and that she had maintained her physiological parameters . at 3 months follow - up , she was growing well and maintained a stool frequency of one to two soft stools per day . stool frequency can be crippling following proctocolectomy , and although opioids might provide temporary relief of the symptoms , they can not prevent relapses . we believe that step alone , performed in an adequate length of bowel , can control the stooling of such patients .

key clinical messagewe present a pediatric case of medically unmanageable juvenile colonic polyposis , initially treated with subtotal colectomy and an ileostomy followed by a proctectomy , ileal - j - pouch and serial transverse enteroplasties ( step ) of the distal ileum . the step procedure in an adequate length was able to control stooling of our patient .

pilar cysts are common , and benign lesions and malignant transformation in them is extremely rare . trichilemmal cyst or pilar cyst is defined as a cyst containing keratin and its breakdown products . it is usually situated on the scalp with a wall resembling external hair root sheath . it is seen mainly in the at middle age and is inherited in an autosomal dominant fashion . in this case report , we present a 55-year - old man with multiple giant pilar cysts that were present since childhood . one of the cyst on the chest was transformed to squamous cell carcinoma ( scc ) . a 55-year - old man was referred to us because of multiple cystic lesions located on the body , some of them measuring several centimeters [ figures 16 ] . these lesions were present since childhood , and during this period the patient had no complaint except cosmetic concerns . patient had ignored these lesions for many years until one of the lesions on the chest became ulcerated and then infected [ figure 5 ] . pathology of the lesion located on the chest showed neoplastic proliferation of the epithelial cells . pathology of the five other lesions showed a cystic lesion covered with stratified squamous epithelium without granular layer containing homogenous laminated keratin . mohs surgery was performed to excise large scc of the chest , and the site of operation was repaired by split - thickness skin graft technique . the patient visited every month for 1 year and during this period he showed no sign of recurrence in the operation site . also , no change was observed in the remaining pilar cyst of the patient and he is now followed every 6 months . one of the lesions on the chest was transformed to scc a closer view of the figure 1 close up of lesions on the trunk pilar lesions on the back of patient a close view of the scc showing large infected ulcer pilar cyst of the scalp pathology of the lesion on the chest showing squamous nests in well - differentiated scc pathology of pilar cyst showing trichilemmal pattern of keratinization ( 400 ) pilar or trichilemmal cysts contain keratin and its breakdown products , lined by a wall resembling the external ( outer ) hair root sheath . the patient had these lesions since childhood , although most patients with this lesions are affected in middle age . although pilar cyst are usually observed on the scalp , the giant cystic lesions of our lesions were scattered over the whole body . one of these lesions on the chest was transformed to squamous cell carcinoma that was excised using mohs surgery . according to our literature review , up to now the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . pilar cysts may rarely transform to scc and also may occur on many parts of the body . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . pilar cysts may rarely transform to scc and also may occur on many parts of the body . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . pilar cysts may rarely transform to scc and also may occur on many parts of the body .

trichilemmal cyst or pilar cyst is defined as a cyst containing keratin and its breakdown products . it is usually situated on the scalp with a wall resembling external hair root sheath . in this case report we present a 55-year - old man with multiple giant pilar cysts that were distributed over the whole body since childhood . one of the cyst on the chest was transformed to squamous cell carcinoma ( scc ) .

this is an interesting case of a child who , following a minor head injury a few weeks earlier , was initially found to have a thin chronic subdural haematoma on plain computed tomography ( ct ) imaging . following burr hole surgery for evacuation of the subdural haematoma his post - operative scan revealed an underlying arachnoid cyst . it is established that intra - cystic bleeding can ensue following minor head injury in patients with this benign developmental malformation . often the haemorrhage is obvious on initial imaging but this is not always the case . a 13-year - old boy presented with a 1-month history of worsening global headache with associated nausea and vomiting . the symptoms developed following an injury sustained by falling and hitting his head on a chair . he had no significant past medical history , met all his developmental milestones and had no significant family history . on examination he had normal higher mental and cranial nerve function and no focal motor deficits . a plain ct head [ figure 1 ] revealed a thin isodense subdural collection overlying the left cerebral convexity with 4 mm of midline shift . sulcal effacement and distortion of the frontal horn of the left lateral ventricle could also be seen . minor head trauma often precedes complications causing rupture of small bridging vessels between the dura and outer membrane of the cyst . , intra- and extra - cystic bleeding had occurred simultaneously causing an isodense arachnoid cyst with an accompanying subdural collection . the child was managed surgically first with a left - sided burr hole evacuation of the chronic subdural hematoma . a second ct head scan was , therefore , performed [ figure 2 ] , revealing the left middle cranial fossa arachnoid cyst causing the residual midline shift . this was also managed surgically with a temporal craniotomy and marsupialization of the high - pressure cyst [ figures 3 and 4 ] . axial computed tomography brain : postburr hole drainage : left temporal arachnoid cyst revealed photograph : craniotomy to reveal membrane of arachnoid cyst photograph : evacuation of high - pressure cystic hematoma the majority of arachnoid cysts are incidental findings on brain imaging and do not require treatment treatment options vary and include microscopic or endoscopic cystocisternostomy , cyst wall resection , and shunt insertion . in this case , the cyst was located in the cerebral convexity and had displaced deeper brain structures ; therefore , craniotomy for cyst wall resection / marsupialization was indicated . there is increasing evidence to suggest that fenestrations made endoscopically are as safe and effective as open surgical techniques . . magnetic resonance imaging should show the cyst as a well - defined nonenhancing mass that is isointense to cerebrospinal fluid and should , therefore , be considered in any child presenting with a subdural collection .

we present an unusual case of a 13-year - old child who following minor head injury presented with what appeared to be a thin chronic subdural hematoma on plain computed tomography imaging . the child was found to have an underlying arachnoid cyst . intra- and extra - cystic bleeding had occurred simultaneously causing an isodense cyst with an accompanying subdural collection . this radiographically occult pathology should be excluded using magnetic resonance imaging in any child presenting with a subdural collection .

metastases to organs such as the liver , bones or central nervous system appear to be a frequent complication of lung cancer , whereas metastases to the suprarenal glands are found less frequently . metastases of lung cancer to the spleen are a great rarity and they are described sporadically . an adrenal gland tumor detected incidentally during imaging tests is described as an incidentaloma [ 37 ] . primary cysts acquiring enormous proportions and hemangiomas are classified as benign tumors [ 9 , 10 ] . metastatic lesions and inflammatory pseudotumors may also be seen , but only very rarely and usually as casuistry [ 11 , 12 ] . splenic lesions may be observed in the course of malignant lymphoma [ 13 , 14 ] . lesions characteristic of sclerosing angiomatoid nodular transformation ( sant ) have also been described . in most cases , the typical characteristics of splenic tumors are established on the basis of histopathological findings , which are obtained by the surgical removal of the tumor or by post - mortem examination [ 8 , 10 , 1618 ] . this work presents a case of simultaneous dissemination of lung cancer to the adrenal gland and to the spleen . a female patient ( age 74 ) was sent from a hospital in zawiercie for further investigations and management of a left lung tumor lesion discovered during x - ray examination . chest surgeons had rejected her from an invasive therapy . however , bronchoscopy was performed and revealed no evidence of pathological bronchial lesions . in this situation the patient was sent to our hospital for the purpose of making the histopathological diagnosis ( history no . 16735/877/09 ) . computed tomography ( ct ) scan showed chest infiltration situated peripherally in the left lung . after establishing the distance , place and depth of the puncture by using ct ( fig . 1 ) , the parietal tumor was visualized by ultrasound and a biopsy was performed . we performed an ultrasound - guided ( free hand technique ) fine - needle biopsy of the lesion using a hitachi eus 515 sonographic machine ( fig . computed tomography imaging scan establishing the distance , place and depth of the puncture ultrasound imaging scan wall located left lung tumor biopsy needle the ultrasound examination of the abdomen revealed a pathological mass in the spleen and in the left adrenal gland ( fig . we also performed in local anesthesia an ultrasound - guided ( free hand technique ) fine - needle biopsy of these lesions . in our case small cell lung cancer was detected in the percutaneous biopsy of the left lung . the same type of cancer as in the left lung was observed in both the adrenal gland and in the spleen ( metastases of small cell cancer ) . imaging methods available to us showed no evidence of cancer metastases in other organs . in the existing literature , we found only a few cases of lung cancer metastases to the spleen [ 2 , 17 , 18 ] . simultaneous metastases of lung cancer to the adrenal gland and the spleen have never been described . the case presented above shows that the metastatic lesion can sometimes be an accessible place to collect tissue for diagnosing the cancer pattern of the primary cancer site . the case is exceptional because the spleen is an organ where lung cancer metastases are not frequently found , while metastases to the adrenal gland alone are common . the case is also unique because the adrenal gland and the spleen are organs where finding concurrent metastases of lung cancer is very rare .

metastases of lung cancer to such organs as the liver , bones or to the central nervous system appear to be a frequent complication of this disease . at the same time , metastases to the adrenal gland are found less frequently . metastases of lung cancer to the spleen are a great rarity and they are described sporadically . our report presents a unique case of left lung cancer with simultaneous metastases to the adrenal gland and to the spleen . all the presented lesions were diagnosed by ultrasound guided biopsy and confirmed by histopathological examination.the patient received combined chemoradiotherapy . she was closely monitored over an 18-month observation period following treatment . no new metastases were reported .

alexandrite laser ( 755 nm ) is one of the most common methods of hair removal . however , the disuse of protective goggles during the application of this laser is a serious problem . alexandrite laser light is visible , and it is known that ocular injuries may occur during the use of alexandrite laser when protective goggles are not worn [ 1 , 2 ] . still , foveal injuries by alexandrite laser are rare . herein , we reported a case with accidental foveal injury and subsequent macular edema secondary to alexandrite laser contact , treated with an intravitreal ozurdex implant . a 35-year - old male patient presented to our clinic complaining of a scotoma in the right eye , which had begun 2 h after looking at a laser probe for a few seconds in an esthetics clinic . fundus examination showed a pale , white and sharply circumscribed laser spot lesion localized at the inferior nasal foveal region , involving the central foveola . a visual field test ( humphrey , zeiss , germany ) displayed a scotoma consistent with the affected region . a hypofluorescent lesion , which was in accordance with the round laser spot , was detected in the early and late phases of fundus fluorescein angiography ( canon inc . , an intense increase in reflectance in the region of the spot was also identified on optic coherence tomography ( oct ; slo , optos , dunfermline , uk ) . there was no change in the findings at the first control visit after 1 week . however , at the second week , the bcva of the patient had decreased to 3/10 , and edema and hemorrhage had occurred around the lesion . oct demonstrated intra- and subretinal edema as well as tissue disruption within the injured area ( fig . ozurdex was implanted via the pars plana region with its specific cartridge in the operating room . an intraocular pressure elevation ( 35 mm hg ) which was successfully controlled by a topical antiglaucomatous agent occurred . one month after the injection , bcva was 20/30 despite a hemorrhage around the injured area on fundus examination ( fig . oct showed decreased macular edema and a hyperreflectance scar in the affected area ( fig . 4 ) . a visual field test demonstrated no change in the scotoma of the patient . it is known that alexandrite laser causes a temperature increase and subsequent coagulation necrosis in melanin - containing tissues , which is termed selective photothermolysis . this temperature rise might cause burns , pigmentation changes , and erythema in surrounding tissues . the first type are anterior segment complications such as uveitis , pupillary distortion , iris atrophy , and cataract , which can occur due to laser applications to the face with closed eyes . the second type are posterior segment complications such as macular hole and macular scar caused by the direct effect of the laser beam on the retina while the eyes are open . the ozurdex intravitreal implant is composed of a biodegradable copolymer of lactic and glycolic acids containing micronized dexamethasone . the drug - copolymer complex gradually releases dexamethasone that transforms into glycolic and lactic acids after the injection and eventually into water and carbon dioxide . after intravitreal injection , the ozurdex implant reduces the vitreous levels of several mediators which cause disruption of the blood - retinal barrier ( i.e. , vascular endothelial growth factor ) and accordingly diminishes the vascular permeability . ozurdex is frequently used in the treatment of macular edema due to diabetes mellitus , retinal vein occlusion , and noninfectious uveitis . in our case , we first used topical steroid drops to prevent complications after alexandrite laser injury ; however , this medication was ineffective . therefore , the inflammatory process secondary to the foveal injury and the subsequent macular edema were treated with ozurdex because of its rapid and potent antiedematous effect . in a case report by anaya - alaminos bcva was reported as 20/200 initially and 20/100 after a 1-year follow - up period in a patient with accidental foveal injury by alexandrite laser . there was no information about the treatment or whether macular edema had occurred during the follow - up period . we believe that the better final visual acuity in our case was achieved by swift resolution of macular edema by ozurdex . retrobulbar and oral steroid treatments have been used in the treatment of foveal injuries caused by femtosecond laser and green laser pointer [ 9 , 10 ] . bcva improved from 20/30 to 20/20 after treatment in a case with femtosecond laser injury , while it increased from 20/100 to 20/60 in a case with green laser pointer injury . the difference between the initial visual acuities and their increases might have been caused by the different energy levels , the durations of laser contact , and the distances of the lesions from the central foveola . the response to treatment was often greater among eyes with a shorter duration of macular edema . we preferred an ozurdex implant to systemic steroids due to its rapid and target - oriented effect and the lower systemic side effects for reducing macular edema . in summary , alexandrite laser injuries might cause macular injury and edema , and the ozurdex implant can be an effective treatment in these cases . the patient was informed about the risks of the treatment , and informed consent was obtained .

alexandrite laser is one of the most common methods of hair removal . its utilization is gradually increasing due to easy accessibility and high effectiveness . however , the disuse of protective goggles during the application of this laser is a serious problem . in this case report , we presented a 35-year - old male patient who had foveal injury by alexandrite laser . the inflammatory process secondary to the foveal injury and subsequent macular edema were treated with ozurdex because of its potent antiedematous effect .

understanding of the daily rhythmicity in nociception is important for the standardization of studies of analgesic drugs . yet , few studies have investigated the daily rhythmicity in nociception . although studies on rats and golden hamsters have indicated the occurrence of greater pain sensitivity during the dark phase of the light - dark cycle , another study on rats indicated the occurrence of greater sensitivity during the light phase , and a study on mice indicated the occurrence of two daily peaks in sensitivity , one during the light phase and one during the dark phase . they were housed in microlon cages maintained at 25 1c under an l12:d12 light - dark cycle . nociception was evaluated by eddy 's hot plate method , tail immersion method , and tail clip method . the latency between the noxious stimulus and the animal 's response was recorded as reaction time . rats previously adapted to an l12:d12 light - dark cycle were divided into 7 groups of 6 animals and tested at one of 7 times of day 4 hours apart . the same groups of animals were retested a week later with the same protocol , except that the animals initially tested first during the light phase of the light - dark cycle were tested first during the dark phase , and vice versa . reaction time ( average of the three methods ) was longest at the transition from light to darkness and shortest a few hours before the transition from darkness to light . this suggests greater pain sensitivity late in the dark phase , which is in agreement with previous studies in rats and golden hamsters . another study in rats suggested the occurrence of greater sensitivity during the light phase , but this was probably an artifact of the experimental procedure , as only two time points during the day were reported . a study on mice suggested the occurrence of two daily peaks in sensitivity , one during the light phase and one during the dark phase . the amplitude of the daily variation in latencies was much smaller in that study than in ours , and it is possible that random oscillations were interpreted as a daily rhythm . daily rhythmicity of nociception in rats the figure shows the daily variation in reaction time to nociceptive stimulation . the horizontal bar at the top indicates the timing of the light - dark cycle . sensitivity to pain is highest late in the dark phase of the light - dark cycle and lowest at the light - dark transition . we , the authors declare that we have not received funds from any agency or organization for carrying out this work . carried out the study cv carried out the replicate study nm and sjp evaluated the data statistically

backgroundmany behavioral and physiological variables exhibit daily rhythmicity . few investigations of the daily rhythmicity in nociception have been conducted , and conflicting results have been obtained . the present study evaluated the daily rhythmicity in nociception in wistar rats.methodsnociception was investigated by eddy 's hot plate method , tail immersion method , and tail clip method . the latency between the noxious stimulus and the animal 's response was recorded as reaction time . separate groups of rats were tested in 4-hour intervals for 24 hours.resultsthere was clear daily variation in response latency . reaction time was shortest a few hours before lights - on and longest at the light - dark transition.conclusionnociception exhibits robust daily rhythmicity in rats . sensitivity to pain is highest late in the dark phase of the light - dark cycle and lowest at the light - dark transition .

a 30-year - old male presented in the emergency department with a history of bilateral blurring of vision of one - day duration . since his systemic hypertension was not well controlled with this drug his physician had switched over to a combination of atenelol 50 mg with chlorthalidone 12.5 mg ( tenoric 50 , ipca pharmaceuticals ) . refraction was done and his visual acuity was improving to 20/20 in both eyes with a spherical correction of -5 diopter sphere ( ds ) . a cycloplegic refraction revealed a value of -3 ds/-0.5 diopter cylinder ( dc ) x 80 in the right eye and -3.75 ds/-1.0 dc x 130 in the left eye . b - scan ultrasonography showed shallow peripheral serous choroidal detachment [ fig . 2 ] . an optical coherence tomogram ( oct - stratus version 4.0 , zeiss ophthalmics ) was also done since retinal striae were present , which showed a mild thickening of the macula . hematological evaluation including renal parameters and computerized tomogram of the head were within normal limits . he was reviewed after five days and his visual acuity had improved to 20/20 without any refractive error . fundus examination revealed disappearance of retinal striae at the macula and peripheral choroidal effusion had also resolved [ figs . a change analysis was done using the software in stratus oct version 4.0 which revealed definite reduction in the thickness of the macula . we report a case of acute myopia induced by oral consumption of chlorthalidone for systemic hypertension . acute transient myopia may be induced by several drugs and different mechanisms have been described . the exact mechanism producing acute myopia is not very clear even though it has been postulated to be due to ciliary body effusion,1 peripheral uveal effusion2 and ciliary spasm3 and lens swelling . oral sulphonamides cause transient myopia as a result of forward displacement of the lens due to allergic ciliary body edema and rotation.3,4 intravenous administration of equine anti lymphocytic globulins causes transient myopia due to ciliary spasm.7 some of the other drugs that have been described to cause transient myopia include indapamide,5 hydrochlorothiazide,2 triamterene2 and topiramate.8,9 in this case a combination of ciliary spasm and peripheral choroidal effusion explain the induced myopia . since the cycloplegic refraction showed a decrease in myopic shift by about 2 diopter it is evident that there was an element of ciliary spasm . retinal striae have been reported to occur with acute myopia after drug intake.10 ciliary body rotation and edema resulting in forward movement of iris lens diaphragm has been reported as a possible cause for induced myopia by some authors.1,2,3,11 in this case there was no definite shallowing of the anterior chamber . we feel that this mechanism may not explain the pathogenesis of myopia in this case . to our knowledge since this is a common diuretic used in the treatment of systemic hypertension , we feel that it is important to be aware of this possible idiosyncratic adverse effect .

we report a case of sudden loss of vision due to the development of acute myopia after the intake of chlorthalidone used for treating systemic hypertension . clinically this was associated with ciliary spasm , shallow peripheral choroidal effusion and retinal striae at the macula with increase in macular thickness seen on optical coherence tomography . all these findings were reversed completely once the drug was discontinued . development of acute myopia should be kept in mind as an adverse effect of a commonly used antihypertensive drug , namely chlorthalidone .

a 16-year - old female patient came to department of oral medicine and radiology with chief complaints of bleeding from gums . on examination , she was thin built . extraoral examination of face and skull revealed hypoplasia of the malar eminences and zygomata , hypoplasia of maxilla and underdevelopment of the mandible [ fig . 1 ] . ears appeared low - set and rotated with presence of skin tags [ fig . 2 ] . intraoral examination revealed inflamed gingiva due to poor oral hygiene . radiological examination supported the clinical findings . patient was then referred to ophthalmology department where ocular examination showed downward slant of palpebral fissures , ptosis of upper lids and limbal dermoid in right eye measuring around 1 1 cm in diameter [ fig . the patient was uncooperative and refused any other treatment except for oral prophylaxis which we performed to relieve her from bleeding gums . extraoral examination of face and skull showing hypoplasia of the malar eminences and zygomata , hypoplasia of maxilla and underdevelopment of the mandible low - set ears along with presence of skin tags ocular examination showing limbal dermoid in right eye the most ordinary type of acrofacial dysostosis is the nager type ( nafd ) of the so - called pre - axial acrofacial dysostosis . this disorder is categorized by the office of rare diseases in the united states as a rare illness ; thus , it has only affected less than 200,000 of the country 's population . some studies have shown that the gene which causes the illness might be located around 9q32 and it is possible that the zfp-37 gene could also be involved . the major symptoms of nager syndrome often include the underdevelopment of the jaw and cheek areas , a jaw that may appear smaller than the usual or normal jaw , hearing problems ( and some defects on the external or internal ear area ) , cleft palate ( which is a congenital defect of the mouth or the lip ) , the absence or underdevelopment of the thumb or thumbs , forearms that appear short ( the forearms could altogether be absent of the radius bone ) , fusing of the bones of the forearms , poor elbow movements ( or limited extension of this body part ) , ears that appear to be backward rotated , the ears manifesting some skin tags ( or they could be low - set ) , and feeding and breathing problems ( especially among infants who are afflicted with this syndrome ) . our patient showed underdevelopment of jaws , hypoplasia of the malar eminences , zygomata and hypoplasia of maxilla . ocular manifestations of this syndrome are rare and include downward slant of palpebral fissures , ptosis of upper lids , coloboma of lower lids , and deficiency of eyelashes of the medial one - third of the lower eyelids . our patient showed downward slant of palpebral fissures , ptosis of upper lids and limbal dermoid in right eye . traditionally , the differential diagnosis of nager syndrome is done with treacher - collins , pierre - robin and goldenhar syndrome . treacher - collins syndrome is characterized by maxilla and mandible hypoplasia , downslanting palpebral fissures , colobomas of inferior eyelid and auricular malformation . pierre - robin syndrome presents with micrognathia , cleft palate and glossoptosis . in goldenhar syndrome , we find hemifacial microsomy , flattening of maxilla , temporal and zygomatic bone , microtia , ocular and vertebral alterations , and in 50% of cases , fallot tetrad . the echographic findings of these malformations become evident usually in the third trimester , therefore a systematic ultrasound exploration including visualization of fetal face line is essential for its detection . if the patient suffers from speech retardation , then a speech therapist could help the patient . they may not totally cure the condition , but they can help in managing the symptoms . treatment of limbal dermoids may consist of periodic removal of irritating cilia , topical lubrication to prevent foreign body sensation , or excision of the lesion if it is causing significant cosmetic disfigurement or interfering with vision . our patient refused to take any ocular treatment and insisted on taking only the dental treatment for which she visited our hospital . correct understanding of the underlying pathophysiologic pattern of this disease is necessary for optimal diagnosis and treatment planning . ophthalmological examination of these patients is a must as ocular manifestations are not uncommon in these patients .

nager syndrome , also called preaxial acrofacial dysostosis , comprises two groups of defects involving the limbs and craniofacial region , respectively . this syndrome is rare and only 70 cases have been reported in the literature . the exact cause of this syndrome is unknown , but there is indication that it is genetically based . ocular manifestations of this syndrome include widely separated downward slanting eyes , absence of eyelashes , ptosis of upper eyelids and colobomas on the inner aspect of lower eyelids . we report limbal dermoid in a patient with nager syndrome . we did not find such an association of limbal dermoid in nager acrofacial dysostosis syndrome on pubmed using nager acrofacial dysostosis , limbal dermoid and ocular manifestations as the keywords .

data synthesized from existing randomised controlled trials show that pyloric drainage procedures reduce the occurrence of early post - operative gastric outlet obstruction after oesophagectomy with gastric reconstruction ( 1,2 ) . pyloromyotomy and pyloroplasty are generally considered simple procedures , however , they may be complicated by stricture , leak , and even death ( 3 ) . because of these complications some advocate not performing an emptying procedure during oesophagectomy ( 3 ) . a 61-year - old gentleman presented in january 2010 with dysphagia for solids and regurgitation . this arose from a long segment of multifocal dysplastic barrett s metaplasia ( 22 - 39 cm ) . full pre - operative staging investigations diagnosed a poorly differentiated mid to distal oesophageal adenocarcinoma ( t3 , n1 , m0 ) . the patient was recruited to the oeo5 trial in april 2010 and underwent neo - adjuvant chemotherapy with 5-fluorouracil and cisplatin . in may 2010 he underwent 3-stage mckeown oesophagectomy with a formal heineke - mikulicz pyloroplasty ( 2-layer , interrupted closure with omental patch ) . final histology of the resected specimen revealed t3 , n1 ( 2/22 ) , m0 , poorly differentiated adenocarcinoma with microscopically involved margins ( circumferential ) with vascular and lymphatic invasion . on post - operative day 7 a routine post - operative contrast swallow was performed . this identified a spill of contrast due to a 5 mm anterior defect at the level of the oesophago - gastric anastomosis suggestive of focal ischaemia . a further contrast swallow revealed contrast aggregating around the drain ( figure 1 ) and that the covered oesophageal stent was appropriately positioned with no evidence of on - going leak ( figure 2 ) . a contrast swallow demonstrating contrast aggregating around the drain & a covered oesophageal stent appropriately positioned with no evidence of leak on post - operative day 11 , at routine dressing change on the ward , a full thickness abdominal wall dehiscence was identified . the patient was managed with washout , drainage and re - suturing of the abdominal wall over a vicryl mesh . the original intra - abdominal tube drain was left undisturbed . on post - operative day 16 the defect at the pyloroplasty suture line was now visible endoscopically and a covered pyloric stent was located appropriately across the defect . due to proximal stent migration this was unsuccessful in controlling the leak ( figure 4 ) . a late pyloroplasty leak diagnosed on contrast swallow & a contrast swallow demonstrating proximal stent migration and evidence of on - going leak the patient was now stable and he requested to eat and drink . a surgical approach to the management of fistula after pyloroplasty was deemed inappropriate because of the recent surgery for intra - abdominal sepsis and the presence of an abdominal wall mesh . a novel approach to the management of a fistula after pyloroplasty was employed . a combined endoscopic and fluoroscopic procedure was performed where a radiologically placed snare was inserted through the abdominal drain and manipulated into the gastric tube through the defect in the pyloroplasty . a gastroscope was used to introduce a t - tube into the gastric tube and into the snare . the abdominal drain and snare were withdrawn and the t - tube placed appropriately , plugging the hole in the pyloroplasty , under direct vision . a further guide wire was left in place along the tract of the tube drain and was used to deploy a pigtail drain alongside the pyloroplasty ( figures 5 and 6 ) . an intra - operative image demonstrating the combined endoscopic and fluoroscopic procedure a pictorial representation of the result of the procedure follow up ct , with contrast both orally and through the t - tube , showed no evidence of ongoing leak from the pyloroplasty . tube satisfactorily plugged the hole through the anterior wall of the pyloroplasty , best demonstrated in the lateral decubitus position ( figures 7 and 8) . the patient was discharged with a capped off t - tube in situ and made a good recovery . a follow - up ct demonstrating no evidence of on - going leak from the pyloroplasty & a follow - up ct demonstrating the flanged t - tube satisfactorily plugging the hole through the anterior wall of the pyloroplasty treatment of leaks by direct suture repair , revision , patching , and application of fibrin glue to leaks have failed to gain widespread acceptance owing to a high failure rate ( 4 ) . alternative methods of controlling leaks such as the placement of covered stents are feasible but can be complicated by stent migration ( 4 ) and inadequate drainage of associated collections . to our knowledge this is the only reported case of management of late pyloroplasty leak following oesophagectomy with a combined endoscopic and radiological placement of a t - tube . the t - tube provided an ideal plug at the site of leak which enabled the patient to consume a normal diet and return home safely .

management of upper gastrointestinal anastomotic leaks is an inter - disciplinary challenge . we present a case of late pyloroplasty leak following 3-stage oesophagectomy . we describe a novel , combined endoscopic and fluroscopic procedure to introduce a t - tube into the anastomotic leak proving an ideal plug at the site of leak which enabled the patient to consume a normal diet and return home safely .

this disorder is relatively frequent on the beard of adult men and on the scalp of children . a 33-year - old man was referred for evaluation of an itchy sensation on his back that had been present for more than 10 years . physical examination revealed a large patch with short and irregular hairs that involved his entire back [ figure 1a ] . dermoscopic evaluation showed that hair follicles contained several short hair shafts with different lengths [ figure 1b ] . histological examination revealed that the follicular structures consisted of different hair shafts , which were enclosed in a common outer root sheath [ figure 2 ] . these findings suggest the diagnosis of pili multigemini . each hair had its own inner root sheath . ( a ) physical examination revealed an extensive and well demarcated patch with short and irregular hairs that involved the entire back ( b ) dermocopic image shows that hair follicles contained several short hair shafts with different lengths histologic image . follicular structures include different hair shafts ( yellow arrows ) , which were enclosed in a common outer root sheath pili multigemini is a hair disorder characterized by clusters of shafts that emerge from a single follicle . a common bulb encloses a dermal papilla that splits into different - sized hair shafts with separate cuticles . normal hair grows from a single hair follicle and exits from one hair canal to the surface of the skin . the cause of pili multigemini is unknown , although there may be a genetic link . it has been suggested that a subdivided papilla produced divided hairs or that multiple hairs may be due to the partial merging of several papillae . an extensive presentation of pili multigemini over a large area has been previously reported in three patients . they showed a wide patch consisting of irregular and coarse thicker hairs over the entire back . the authors propose that this extensive condition could be underdiagnosed because the nevoid multiple hairs have no specific symptoms . trichoscopy enhances the evaluation of hair disorders and facilitates the diagnosis of this rare hair shaft dysplasia . differential diagnosis includes mainly pili bifurcate , which is characterized by bifurcation of the hair shaft . each bifurcation produces two separate parallel branches , which fuse again to form a single shaft , and each branch of the successive bifurcations is covered with its own cuticle . however , in pili gemini , a kinetic papilla splits at the upper end from single to double - tipped during the anagen phase and consequently the same follicular matrix produces two different - sized hair shafts having separate cuticles that emerge through a single pilary canal . pili gemini maintains the double tipped papilla and consequently the hair shaft does not fuse again . in conclusion , we present an extensive case of pili multigemini over the back . this condition has been described previously as the nevoid pili multigemini . despite the rarity of this entity , probably it could be a commonly recognizable condition with an exhaustive exploration . the large areas implicated in the patients described and the normal hair appearance in other locations suggests a genetic link .

pili multigemini is a rare disorder where more than one hair exists in a single hair follicle . papillar tips that divide into several tips will produce several hair shafts , so that characteristically do not fuse again . this disorder is relatively frequent on the beard of adult men and on the scalp of children . however , extensive areas of pili multigemini in other locations have rarely been described .