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pure ligamentous flexion - distraction injury of the lumbar spine is a relatively rare clinical entity comprised of transverse disruption of the disc combined with tension splitting of posterior column . it has been first described by chance in 1948 as a result of flexion over a fulcrum positioned anterior to the spine . it is largely related to the use of lap belt restrains during motor vehicle accidents , when large forces are transmitted through the abdominal cavity often causing concomitant visceral injuries . neurologic deficit is rare as majority of cases involve lumbar levels , below the spinal cord . we report a 19-year - old female backseat passenger wearing a lapbelt who was involved in a head - on collision when the sport utility vehicle ( suv ) she rode in fell into gully . on admission she was paraplegic , with t12 sensory level and no motor and sensory function of s4-s5 ( american spinal injury association ( asia ) a ) . plain x - ray [ figure 1 ] and computerized axial tomography ( cat ) spine showed a flexion - distraction injury at the l3-l4 level , with a small fragment of the l4 vertebral body avulsed [ figure 2 ] . after ruling out intra - abdominal injuries , patient was taken into surgery within 24 h after accident . immediately after skin incision further dissection of the subcutaneous tissue revealed that fascia was disrupted and within the interspinous space a neural structure with tangled nerve fibers was found [ figure 3 ] . on closer inspection it was identified as a conus medullaris , which was wholly severed from the spinal cord . it was removed and laminectomy was performed to attempt dural repair . despite efforts , dura was not found as it probably recoiled into the upper portions of spinal canal as a result of injury . the pedicle screws were placed into l2-l5 vertebral bodies , with subsequent l3-l4 endplate preparation and implantation of an interbody spacer . autologous bone locally procured from facet joints mixed with synthetic bone substitute was used to fill in the interbody space . the postoperative course was complicated by formation of a large subcutaneous hematoma which developed despite placement of a wound drain . the wound was revised 3 days after initial surgery , hematoma was evacuated , and skin was closed in multiple layers . some 7 days later , patient developed a cerebrospinal fluid ( csf ) leak through the incision ; it was treated with a lumbar drainage ( placed at l2 level ) and prolonged bed rest . eventually the patient made a satisfactory recovery and 12 months after surgery was able to walk with a knee - ankle - foot orthosis . she had no motor function below knees , no sensation below l2 , and no voluntary bladder control . x - ray taken in lateral decubitus position ct showing only modest malalignment and the naked facet sign , visible in all planes ( arrows ) . chance type flexion - distraction injuries are rare , the incidence among entire series of thoracolumbar fractures ranges from 5 to 15% . they are a consequence of the hyperflexion of the spinal column around the fulcrum placed in front of the abdomen . typically this occurs as a result of injuries sustained while wearing the seat belt , especially the lap belt . the more anterior position of the axis of rotation , the greater distracting force acting on adjacent segments with a much lesser axial component . flexion - distraction injuries of the spine are often accompanied by serious visceral injuries which should be ruled out in each and every case . although this type of injury is considered highly unstable , neurological deficit is rare , and if present , it is usually caused by direct injury of lower thoracic to upper lumbar levels . in our case the deficit was a result of indirect injury : most likely the avulsion of the conus medullaris was caused by the sudden deceleration with subsequent distraction and eventual failure of ligamentous elements followed by substantial traction of the conus by the nerve roots . recent article by rade et al . , provides an elegant illustration to support this hypothesis . it was found that during the straight leg raise ( slr ) test the conus descends by more than 2 mm . it is plausible to expect far greater dislocation with force acting upon nerve roots during abrupt deceleration in a restrained patient with both hips flexed . to our knowledge there have been only a handful of similar cases reported in literature , all among children and adolescents who sustained injuries in motor vehicle accidents . the authors of relevant articles and reviews emphasize the violent mechanism of injury and a significant risk of intra - abdominal visceral injuries . as the lap belts have been gradually phased out , the incidence of the type of injury described in this article is likely to decline . nevertheless both the practitioners and the motor vehicle users should be aware of potentially dreadful consequences of inadequate or improperly worn seatbelts .

pure ligamentous flexion - distraction injuries of the lumbar spine are relatively rare and even less commonly associated with neurologic compromise . they are largely related to the use of lap belt restrains during motor vehicle accidents . we report a 19-year - old female backseat passenger wearing a lap belt who was involved in a head - on collision . on admission she was paraplegic , with a t12 sensory level and no motor and sensory function of s4-s5 ( american spinal injury association ( asia ) a ) . plain x - ray and computerized axial tomography ( cat ) spine showed a flexion - distraction injury at the l3-l4 level . during surgery in the interspinous space a conus medullaris was identified , which was completely severed from the spinal cord . the patient underwent a fusion procedure and made a good recovery . twelve months after surgery she was able to walk with a knee - ankle - foot orthosis , she has no motor function below knees , no sensation below l2 , and no voluntary bladder control . although described type of injury is very rare , one should always have in mind devastating consequences of inadequate or improperly worn seatbelts .

nevus sebaceous of jadassohn is a congenital hamartoma , which has been reported to be associated with secondary tumors such as syringocystadenoma papilliferum , trichoblastoma and basal cell carcinoma ( bcc ) [ 1 , 2 , 3 , 4 ] . however , more than three multiple tumors arising from a nevus sebaceous of jadassohn is extremely rare . malignant transformation occurs in 1015% of nevus sebaceous in some series , although others suggest that this rate may be lower . studies indicate that the development of bcc or any other malignant neoplasm is very rare . multiple tumors arising from a nevus and/or a phacomatosis provide important clues to understand their histogenesis and their capacity to differentiate and proliferate . here we present a case of multiple tumors of syringocystadenoma papilliferum , sebaceoma , trichoblastoma and bcc that arose from a nevus sebaceous of jadassohn . a 67-year - old female presented with a tumor on the back of her head . the patient had recognized a nodule on the back of her head 37 years earlier . that nodule had gradually enlarged and had started bleeding 10 years earlier . physical examination revealed a 47 26 mm slightly elevated light brown plaque on the back of her head . a 16 13 mm erosive lesion and an 18 17 mm dome - shaped black node were recognized in that plaque ( fig . dermoscopic analysis of the dome - shaped black node showed black homogenous areas , surface scales and arborizing vessels ( fig . dermoscopic analysis of the erosive lesion showed glomerular vessels in pink homogenous areas ( fig . histopathological examination of the black node showed tumor nests with a sheet - like appearance that extended downward into the deep dermis . melanin deposition was scattered irregularly and bleeding was also observed within the tumor nests ( fig . insular tumor aggregations were recognized in the tumor nests and some of those aggregations contained melanin deposits ( fig . immunohistochemistry showed a positive reaction for ber - ep4 in those tumor nests ( fig . 2c ) . at the periphery of the black node , tumor nests also extended into the deep dermis and were composed of several different components ( fig . 2d ) . in the upper portion , tumor nests with interlacing cords were noted . numerous structures resembling follicular germinative cells were recognized and some of those structures extruded from interlacing cords ( fig . a diagnosis of trichoblastoma was made in this upper portion . in the lower portion , the tumor nests consisted of sebocytes , which had a foamy cytoplasm ( fig . 2h ) . the epithelium of the tubules was lined by 23 layers displaying apocrine secretion of decapitation secretion and numerous plasma cells were noted in the stroma of the papillated structures ( fig . a diagnosis of syringocystadenoma papilliferum was made at the erosive lesion . in the slightly elevated light brown plaque , a diagnosis of syringocystadenoma papilliferum , sebaceoma , trichoblastoma and bcc arising from a nevus sebaceous of jadassohn was made . the tumor was resected and there has been no relapse 1 year after the surgery . nevus sebaceous has the potential to generate different lineages of tumors that are not restricted to the sebaceous lineage [ 1 , 4 ] . a secondary tumor from a nevus sebaceous may differentiate into follicular , sebaceous , apocrine and eccrine cells , and rarely , a secondary tumor can differentiate into muscle [ 6 , 7 ] . the simultaneous occurrence of tumors from a nevus sebaceous is not rare , but the number of tumors is usually less than three . a previous study showed that one in 140 cases of nevus sebaceous simultaneously developed three tumors , and another previous study showed that one in 150 cases of nevus sebaceous simultaneously developed four tumors [ 8 , 9 ] . in our case , four distinct tumors ( syringocystadenoma papilliferum , sebaceoma , trichoblastoma and bcc ) arose from a nevus sebaceous . those tumors reflect that nevus sebaceous has a pluripotency for apocrine , sebaceous and follicular differentiation . an intriguing fact is that this patient had a previous history of multiple tumors such as ovarian cystoma , submandibular gland tumor and thyroid cancer . in conclusion , we report a rare case of multiple tumors of syringocystadenoma papilliferum , sebaceoma , trichoblastoma and bcc arising from a nevus sebaceous of jadassohn .

nevus sebaceous is known by its association with one or more secondary tumors , but more than three multiple tumors arising from a nevus sebaceous is extremely rare . a 67-year - old female presented with a light brown plaque on the back of her head that contained a dome - shaped black node and an erosive lesion . histopathological examination showed atypical basaloid cells in the black node . at the periphery of that node , structures resembling follicular germs extruded from interlacing cords in the upper portion and tumor nests with sebocytes were in the lower portion . in the erosive lesion , papillated structures with an apocrine epithelium were observed . in the light brown plaque , enlargement of sebaceous lobules was noted . from those histopathological features , a diagnosis of syringocystadenoma papilliferum , sebaceoma , trichoblastoma and basal cell carcinoma arising from a nevus sebaceous was made . we discuss the rarity of multiple tumors arising from a nevus sebaceous .

malaria is a major health problem in the developing world today , with an estimated 300 - 500 million cases and more than one million deaths each year . artemisinin and its derivatives are rapidly acting antimalarial compounds effective against resistant strains of plasmodium falciparum malaria . this has lead to its increased use in public hospitals in developing countries . we herein report a case of sodium artesunate - induced diuresis in a patient with malaria . a 6-year - old boy presented with fever for five days and vomiting for two days . vomiting was 23 times per day and non - projectile , non - bilious , and contained ingested food particles . there was no dysuria , oliguria , abdominal pain , or bleeding manifestations . on examination , the patient was afebrile with a heart rate of 104/min , respiratory rate of 24/min , and blood pressure of 100/60 mm of hg . liver was soft and non - tender with a span of 6.5 cm in the midclavicular line . investigations revealed : hemoglobin 8.2 gm / dl , total leucocyte count 11,300 /mm ( 60% polymorphs , 40% lymphocytes ) and platelet count 53000 /mm . liver function tests , renal function tests , serum calcium , and phosphorous were normal throughout the hospital stay . the arterial blood gas , urine microscopy , and chest x - ray were normal . widal test , dri - dot test for leptospira and dengue igm antibody test were negative . intravenous sodium artesunate ( 2.4 mg / kg ) , i.e. , 38.4 mg was given at 0 , 12 , 24 , and then once a day for 7 days . the patient did not receive intravenous fluids , diuretics , or vasodilators and continued normal diet and fluid intake . the serum electrolytes , urine output , and urinary electrolytes during the course of hospital admission are shown in table 1 . on day 3 of artesunate treatment , the patient developed increased urine output along with increased excretion of sodium , potassium , and chloride . two days after stopping artesunate the urine output and urinary excretion of electrolytes started decreasing . patient became afebrile on day three and was discharged from the hospital on day 11 of admission . serial values of serum electrolytes , urine output , and urinary electrolytes in patient on artesunate treatment the artemisinin derivatives artemether , artesunate , and dihydroartemisinin are derived from the chinese medicinal herb , qinghao . sodium artesunate is a new antimalarial water soluble drug , which can be administered either by intravenous or intramuscular injection . it is more potent than quinine and rapidly reduces parasitemia and resolves clinical symptoms in resistant parasites strains . this action is due to increased nitric oxide synthesis and inhibitory effect on chloride transport across the cortical thick ascending loop of henle ( talh ) that suppresses tubuloglomerular feedback . nitric oxide causes diuresis and natriuresis by inhibiting the activity of the na , k - atpase and tubular sodium transport . the principal site of action of artesunate in kidneys is supposed to be talh , which has the highest concentration of the enzyme na , k - atpase per tubular length . the increase in the urinary excretion of sodium , chloride and potassium also suggests that this drug has a predominant effect on the proximal segments of the nephron i.e. proximal tubule and the talh . reduction in the reabsorption of sodium and an increase in the distal sodium delivery occurs with a consequent greater sodium reabsorption and potassium secretion in the collecting tubule . the diuretic effect of artesunate is a double - edged weapon that can modify the course of the acute renal failure in malaria . the increase urinary loss of water and electrolytes can worsen the renal failure in hypovolemic patients with pyrexia and tachypnea . however , the administration of artesunate can also be useful in the conversion of an oliguric renal failure to nonoliguric in patients with volume overload . in addition , rapid improvement in pulmonary function has been observed in a patient with respiratory distress syndrome due to the diuretic effect of artesunate . in conclusion , the diuretic effect of artesunate can modify the course of renal failure and respiratory distress syndrome , both of which complicate severe malaria . physicians should be aware of this effect of artesunate , so that it can be used judiciously and to the advantage of patients with severe malaria .

sodium artesunate is used in the treatment of malaria . we report a case of sodium artesunate induced diuresis and natriuresis in a patient with malaria . following artesunate administration there was polyuria accompanied by natriuresis that was reversed after discontinuation of artesunate treatment . the diuretic effect of artesunate can modify the course of renal failure in severe malaria . prescribers should be aware of this effect of artesunate , so that it can be used judiciously and to the advantage of patients with severe malaria .

a 37-year - old korean man was referred to our hospital for evaluation of a solitary nodule on his penile shaft . he had been aware of a slow - growing , asymptomatic nodule for the previous 10 years . more recently , however , he experienced pain , tenderness , increased size , and difficulties during sexual intercourse and erectile dysfunction . there was nothing notable in the patient 's past medical history or family history , and he was generally in good health . a physical examination revealed a 2 cm - sized , ovoid , movable lesion on the dorsum of the penile shaft . the results of a complete blood count , electrolyte battery , liver function , renal function , and urine tests were all within normal limits . magnetic resonance imaging ( mri ) revealed a 2 cm - sized ovoid mass abutting the right corpus cavernosum ( fig . the mass located between the corpus cavernosum and buck 's fascia was removed completely ( fig . the macroscopic appearance of the removed specimen was a 2 cm sized ovoid and bright reddish white mass such as a bean shape . after surgery , he had no pain , a normal sensation of the penile glans , and his erectile dysfunction had improved . at the present time , 26 months after surgery , the patient has experienced no signs of symptoms or recurrence . nf1 is much more uncommon in the genitourinary tract than in other systems , with the bladder being the most commonly involved organ of the genitourinary tract . only one patient with primary neurofibromas associated with penile neurofibromatosis has been described to date in korea . neurofibromatosis typically presents as genital masses or edema in males and clitoral hypertrophy , mimicking intersex condition , in females . although a penile neurofibroma involves continuity of the dorsal neurovascular bundle with the mass , the latter can be excised completely and safely , while preserving the dorsal bundle . although partial excision may be successful , complete resection is necessary not only for cosmetic and functional reasons but also to prevent recurrent problems and to prevent malignant degeneration . therefore , optimal timing of surgery is necessary to allow complete resection without organ impairment . in our patient , the mass did not involve the dorsal neurovascular bundle and could therefore be resected . difficulties in sexual intercourse may include an inability to penetrate due to the mass , despite experiencing normal sensation and erection , as well as erectile dysfunction , due to arterial stealing by the neurofibroma . a bilateral selective pudendal arteriogram showed that both arteries were hypertrophied and supplied the neurofibroma . thus , the erectile dysfunction in our patient was probably caused by a mass effect and psychological influence . the patient gradually became more reluctant to have sexual intercourse and expressed low levels of self - esteem . after surgery , all his symptoms improved and there was no evidence of recurrence . in patients with functional impairment of the penis , resection of the lesion is associated with good outcomes .

neurofibromas of the penis , although very rare , are often associated with neurofibromatosis type 1 . primary solitary neurofibromas of the penis are extremely rare . we describe a 37-year - old man with a solitary neurofibroma in the dorsum of the penis . the patient reported difficulty with sexual intercourse owing to a penile mass and erectile dysfunction . after surgical excision of the neurofibroma , he had no pain and a normal sensation of the penile glans , and his erectile dysfunction improved . at the present time , 26 months after surgery , there has been no evidence of tumor recurrence .

chloroquine has been in use for many years for the treatment of malaria and for the long term prophylaxis of inflammatory diseases such as rheumatoid arthritis , lupus erythematous , and sjogren s syndrome.1 for the latter indications chloroquine was previously prescribed in dosages of 400600 mg / day but has now been reduced to 250 mg / day.23 ocular toxicity associated with chloroquine has been extensively studied since its description in 1957 by cambaiaggi4 and in 1959 by hobbs.5 it can lead to corneal deposits , lens opacities , and maculopathy causing bull s eye lesion.67 chloroquine retinopathy ( cr ) is a severe form of retinal toxicity caused by long - term use of chloroquine with an incidence of 116%.89 retinal photoreceptors and retinal pigment epithelium have been postulated as the primary sites of involvement in chloroquine retinopathy.9 once established , cr is irreversible and may progress even after cessation of the drug.9 in this case report , we present the history and clinical findings of a patient with late onset ocular toxicity , 10 years after stopping chloroquine treatment . a 52-year - old female with sjogren s syndrome presented to us with diminution of vision in both eyes for 1 year . she was diagnosed 19 years prior to presentation and had been taking chloroquine ( 250 mg daily ) for 7 years . the best corrected visual acuities using a snellen chart were 6/9 , n6 and 6/6 , n6 in the right and left eye respectively . she could identify 1 plate correctly in the right eye and 10 plates in the left eye using ishihara s isochromatic plates . indirect ophthalmoscopy of both eyes showed normal looking central fovea with a circular ring shaped area of depigmentation measuring approximately two disk diameters in size akin to a bull s eye appearance . fundus fluorescein angiography showed increased hyperfluorescence due to a window defect in the macular area corresponding to the area of depigmentation and reduced hyperfluorescence due to blockage of choroidal fluorescence in the central fovea [ figures 1 and 2 ] . spectral domain optical coherence tomography of both eyes showed reduced central foveal thickness ( right eye 108 and left eye 132 ) . both eyes showed generalized loss of outer retinal layers in foveal and parafoveal areas while the left eye showed preservation of the inner segment outer segment ( is- os ) junction [ figures 3a and b ] . multifocal electroretinogram showed reduced central and paracentral ring responses and reduced perifoveal ring responses in both eyes [ figures 3c and d ] . based on the history and clinical findings , the patient was diagnosed with late onset chloroquine retinopathy . the patient was informed about the condition and a letter of recommendation addressed to her physician was sent documenting the importance of avoiding chloroquine . fundus fluorescein angiogram showing a ring of increased hyperfluorescence ( window defect ) surrounding a central area of reduced hyperfluorescence ( blocked fluorescence ) ( a ) and ( b ) optical coherence tomography showing generalized loss of outer retinal layers in parafoveal region in both eyes . ( c ) and ( d ) multifocal electroretinogram showing reduced perifoveal ring responses in both eyes in the present case , the patient had placed on chloroquine on a long - term basis over 10 years prior to presentation and had been under the care of an ophthalmologist thereafter . the onset of chloroquine retinopathy more than a decade after stopping chloroquine emphasizes the need for regular follow - up of patients with a history of long - term chloroquine usage . chloroquine is stored in tissues such as the liver and especially the pigmented uveal ocular tissue.10 urinary excretion of chloroquine continues years after cessation of chloroquine.10 these findings buttress the diagnosis of late onset chloroquine retinopathy in our patient . currently there is no definite treatment for chloroquine retinopathy ; hence , it is advisable to discontinue the drug as soon as the signs of toxicity are noted . according to the revised guidelines of the american academy of ophthalmology , each patient who may be placed on long - term chloroquine should undergo a baseline ophthalmic examination which should include both subjective ( 102 visual field testing ) and objective tests ( autofluorescence , multifocal electroretinogram , and spectral domain optical coherence tomography).2 these tests should be repeated annually if the patient falls in the high risk category for developing chloroquine retinopathy.2 the present case report reiterates the need for regular follow - up of patients who have been on chloroquine , even after discontinuation of the treatment . this is important both for the ophthalmologist and the physician to educate the patient about the delayed presentation of this disease entity .

chloroquine retinopathy is a known complication of long - term use of chloroquine . this retinopathy can appear even after usage of chloroquine has stopped . the present case report describes the history and clinical features of chloroquine retinopathy developing a decade after discontinuing the drug .

neonatal facial birth injuries are easily recognizable , but most under - reported form of birth injuries . they are often associated with face presentations , which occur in every 600 - 800 births at term gestation . face presentation usually presents as a deflexed head which becomes extended at pelvic inlet during labor . anything that delays or prevents flexion such as fetal anomalies , contracted pelvis , fetopelvic disproportion or cord around the neck can contribute to face presentation . the presenting part may feel soft and lumpy and the landmarks of the face are felt . the bridge of the nose is an important landmark and often the baby will suck on the examining finger due to rooting reflex . repeated per vaginal examination to assess the presenting part and the progress of labor may lead to bruises in the face as well as damage to the eyes . we describe one such potential case of preventable birth injury involving face , sustained by an unborn child with face presentation , in an institutional delivery . a 22-year - old primigravida was admitted at term with active labor in our tertiary care teaching hospital . per vaginal examination at admission as face presentation was quite uncommon , all our enthusiastic residents as well as the medical students posted in labor room wanted to have a feel of the face during labor . this resulted in repeated unofficial per vaginal examinations in spite of having strict guidelines in place . after delivery , baby was noted to have prominent ulcerations , bleeding and edema of the face [ figure 1 ] . the infant also had injury to right eye with eyelid ulceration , swelling , subconjunctival hemorrhage and difficulty in breathing due to swollen tongue . respiratory distress settled within 2 days and the child was finally discharged after 7 days of hospitalization . facial ulcerations secondary to per vaginal examinations in face presentation iatrogenic neonatal facial injuries are rare , but are one of the most easily preventable neonatal injuries . teaching institutions should have strict guidelines in place in order to avoid iatrogenic birth injuries .

birth injuries involving face are easily recognizable , but are often under - reported . most of these injuries are associated with face presentation . we report an iatrogenic , but potentially preventable facial birth injury sustained by an unborn child in institutional setup .

electro convulsive therapy ( ect ) is an effective treatment modality in the management of depression , mania , bipolar affective disorder ( bad ) , schizophrenia as well as a number of psychiatric disorders since 1930s . complications of ect like subdural hematoma ( sdh ) , intracerebral hemorrhage ( ich ) , have been reported in the literature sparsely . we report a case of chronic sdh in a 38-year - old lady resulting from ect for management of bad . a 38-year - old lady who was a known case of bad for last fifteen years presented with increasingly agitated behaviour along with intense mood fluctuations for last six months . she had a total of 12 sessions over a period of six weeks when she developed frontal headache and frequent vomiting with increasing frequency for last three weeks . there was additional history of altered sensorium , agitation , slurring of speech ( dysphasia ) , and weakness of the right side of the body without loss of consciousness or seizure . on examination by the neurosurgeon and performance of computed tomography ( ct ) scan of the brain , a left temporo - parietal subdural hematoma with midline shift was revealed [ figures 1 and 2 ] . computed tomography scan of the brain showing post electro convulsive therapy subdural hematoma ct scan of the brain showing post ect subdural hematoma owing to the similarity of symptoms with ect , the diagnosis of chronic subdural hematoma following ect is difficult in clinical practice . ect is a well accepted treatment modality for severe mental illness in which a short application of electrical stimulus is used to produce a generalized motor seizure . the generalized seizure lasts several minutes and includes a short 10 - 15 seconds tonic phase followed by a more prolonged clonic phase , lasting for 30 - 60 seconds . most patients scheduled to undergo ect are receiving tricyclic antidepressants ( tcas ) , monoamine , selective serotonine reuptake inhibitors ( ssris ) , lithium carbonate , or a combination of these drugs . the newer drugs such as trazodone and bupropion have lesser complications.[24 ] lithium carbonate prolongs the action of neuromuscular blockade . therefore , pre - ect workup should include a complete medical and neurologic evaluation of the patients . ect can be used safely in elderly patients and in persons with cardiac pacemakers or implantable cardioverter - difibrillators . generalised autonomic nervous system stimulation causes an initial bradycardia and occasional asystole , followed by a more prominent sympathetic response of hypertension and tachycardia . occasionally cardiac dysrrhythmia , myocardial ischaemia , infarction , or neurologic vascular events may be precipitated . first , the medical complications that can be substantially reduced by the use of appropriately trained staffs , best equipments , and best methods of administration of therapy . death due to ect is mostly from cardiovascular and hemodynamic complications and occur most frequently in patients with already compromised cardiovascular profile . the adverse effects of ect includes laryngospasm , circulatory insufficiency , headache , emergence agitation , tooth damage , vertebral compression fractures , status epilepticus , peripheral nerve palsy , skin burns , and prolonged apnea . common adverse effects associated with ect are headache , nausea , fatigue , confusion , and delirium , shortly after the seizure while the patient is recovering from anesthesia . marked confusion may occur in up to 10% of patients within 30 minutes of the seizure and can be treated with barbiturates and benzodiazepines . delirium is usually most pronounced in after the first few treatments and in patients who received bilateral ect or those who have co - existing neurological disorders . high risk group of patients for ect include those with recent myocardial infarction and/or ischemia , uncontrolled blood pressure , intracranial sol , and previous spinal injuries . memory loss is most often reported by patients who have experienced little improvement after ect . few cases of neurological complications such as ich and sdh were not found to increase or deteriorate following ect thereby establishing the safety of this procedure in patients with such neurological complications . although it is difficult to establish the diagnosis of intracranial hemorrhage as iatrogenic , but any change in mental status , worsening of symptoms , or development of focal neurological abnormality in patients undergoing ect should be treated with utmost urgency and investigated with a ct scan of brain to rule out intracranial complications . this case report and other publications suggest that one can not rely only on clinical symptoms when symptoms worsen after ect , although ect is a safe procedure . therefore , it is important to investigate those patients who are having neurological signs and symptoms following ect and a ct or magnetic resonance imaging ( mri ) scan should be performed appropriately . anticipation of complications along with their early diagnosis and treatment are the most important factors in delivering a comprehensive ect .

subdural hematoma is a rare but serious complication following electroconvulsive therapy ( ect ) , a frequently used treatment modality in the management of various psychiatric morbidities including bipolar affective disorder ( bad ) . there are very few reports of intracranial bleeding following ect in the literature . a 38-year - old female , known case of bad for last fifteen years receiving ect , presented with the symptoms of dysphasia , headache , left sided paresis , and sudden deterioration of sensorium . computed tomography ( ct ) scan of the brain was suggestive of left - sided fronto parietal chronic subdural hematoma with midline shift that was drained successfully .

after the operation , he presented with acute urinary retention requiring urinary catheterization , but pulled out the urinary catheter twice because of agitation due to alcoholic stop . for each procedure , a silicon foley catheter sizing 18 french was placed by the nurses without reported difficulty . on postoperative day 5 ( the day following the third urinary catheterization ) , he presented with acute abdominal pain . on physical examination , there was abdominal guarding with hypotension . his temperature was normal , urine output in the foley was 200 ml and the blood parameters showed an acute inflammatory response ( 17000 leukocytes / mm , c - reactive protein : 17.5 mg / l ) , with normal creatinine and elevated urea ( 10 the laparotomy revealed a peritoneal perforation of the urinary bladder , with concomitant urinary peritonitis . the bladder had been perforated by the urinary catheter and there was neither diverticulosis nor tumor ( figure 1 ) . the urinary catheter was left in place for 15 days , and the patient resumed normal urination after its removal . the pathological examination confirmed the absence of tumor and the patient is well after 6 months of follow - up . figure 1intra - peritoneal rupture of the top of the bladder due to the contact with the urinary catheter . intra - peritoneal rupture of the top of the bladder due to the contact with the urinary catheter . intraperitoneal perforation of the urinary bladder may occur in particular situations such as trauma , iatrogenicity ( cystoscopy , prostatectomy ) , cancer , radiotherapy , infections or inflammatory lesions ( cystitis , schistosomiasis , tuberculosis ) and diverticulosis . a few cases of idiopathic rupture that could have been related to the consumption of toxic substances ( alcohol , drugs ) have been reported . few cases of rupture after urinary catheterization without other predisposing factors have been described in the literature . the diagnosis is challenging because the symptoms are not specific ( pain , hematuria , oliguria ) , as well as blood parameters ( inflammatory syndrome , renal insufficiency ) , leading to misdiagnosis and inappropriate or late treatment . the medical history of this patient with several successive urinary catheters may suggest this diagnosis ( even if no traumatic difficulty was reported during the catheterization ) . retrograde urethro - cystography or an abdominal computed comography scan with bladder opacification can establish the diagnosis and localize the site of rupture . the main factor for the therapeutic management the classical treatment for intraperitoneal rupture of the bladder is surgical repair and urinary rest ( with a foley catheter left in place for one week at least ) . laparoscopy is possible in this setting and this approach would have been chosen if the perforation was diagnosed in our case . in selective cases ( particularly in the absence of peritoneal signs ) , idiopathic intra - peritoneal rupture of the bladder is difficult to diagnose due to the non - specific clinical picture . despite it surgical treatment consists of bladder repair and a search for conditions that increase the risk of rupture ( tumor , diverticulosis , infection ) .

intraperitoneal rupture of the bladder is a rare cause of peritonitis . intraperitoneal rupture of the bladder was diagnosed during an emergency laparotomy for suspected mesenteric ischemia . the patient had undergone iterative urinary catheterization after a vascular bypass . the perforation was excised and sutured and the patient was catheterized for urinary rest for 15 days . urinary catheterization is a possible cause of intraperitoneal rupture of the bladder .

the patient was placed in the supine position with the knee in a dangling position at around 70 of flexion . the un - operated left leg was supported with circumferential operative leg holders with sufficient padding at the fibular head to prevent common peroneal nerve palsy caused by the pressure4 ) . first , the patellar apex , patellar tendon and lateral femoral condyle were identified by palpation . the lateral triangle , bounded by the lateral border of the patellar tendon , the medial circumference of the lateral femoral condyle , and distally by the superior rim of the anterior horn of the lateral meniscus , was palpated . once the optimum site for the anterolateral arthroscopic portals are selected , a longitudinal skin incision approximately 4 - 5 mm long was made with a scalpel ( no . subsequently , a vertical incision about 3 - 4 mm was made at the lateral capsule . the sheath was inserted with a careful rotatory movement , avoiding injury to the cartilage and synovial covering of the anterior cruciate ligament . when the sheath penetrated the fibrous capsule and tissue resistance declined , the knee joint was extended and the sheath was carefully advanced into the medial portion of the suprapatellar pouch . the " needle technique " was used to make an anteromedial instrumental portal . by constantly visualizing the needle , the relationship between the skin opening and capsular opening was assessed by monitoring the changes in the relative direction of the scope and the instruments . in an extended knee position ( 20 ) , the capsular opening migrated proximally and the direction from the skin to in a more flexed knee position , the capsular opening migrated distally and the direction from the skin to the capsule moved caudally ( fig . the capsular opening migrated slightly laterally and medially , respectively . the commonly used position in knee arthroscopy is valgus extended knee for the examination of the medial meniscus and varus flexed knee ( figure of " 4 " position ) for the lateral meniscus . at 30 of knee flexion with valgus stress , the direction from the skin to the capsule changed cephalad with a slight lateral tilt . at 90 of knee flexion with varus stress , the direction from the skin to the capsule changed to a caudal with a slight medial tilt . to confirm the mismatch between the skin and the capsule according to various knee positions , capsular suture was carried out using roll wire ( no . 2 , zimmer , warsaw , in , usa ) . the skin was temporarily sutured with staples ( skin stapler , dimensions 6.93.9 mm , johnson & johnson company , cincinnati , oh , usa ) . the relative positions were confirmed by intraoperative real - time c - arm images in various knee position , as described ( figs . 2 , 3 ) . by knowing the direction of portals according to the knee positions , it was possible to access the portals with instruments without changing to the original dangling position or further violating the capsule . even for experienced surgeons , changing the arthroscopic instruments through the knee portals can be a cumbersome process . this is due to the mismatch of the initial relationships between the skin and soft tissue orifices according to the knee position . in these situations , changing the knee position into the original ( index ) position or further spreading the portal opening using a mosquito may lead to prolonged operation time and further violation of the capsule . by understanding the relative skin - capsular mismatch in arthroscopic portals according to knee positions , instrument insertion can be facilitated with the adjustment of the direction of instrument insertion or skin traction . we believe that knowing the direction of portals according to knee position is useful during arthroscopic procedures . the small size of the capsular incision coupled with the elimination of frequent capsular violation limits fluid extravasation , which can complicate a straightforward procedure and lead to increased postoperative swelling . in this series , it was accessible to all portals without any difficulty . various authors have reported new techniques and approaches for easy access of the arthroscopic instruments into the different regions of the knee compartments with their own limitations . kim and kim6 ) reported different locations of portals to visualize the different regions of the compartments and concluded that the locations should be flexible as per the need of the surgeon rather than fixed . ahn and ha7 ) showed that a posterior trans - septal portal further improves the visualization of the posterior compartments . although different orientation of portals are available for better visualization of specific intra - articular regions , most of arthroscopic surgeries can be managed by using anterolateral , anteromedial and posteromedial portals . this note describes the relationships between skin and soft tissue opening according to knee positions . based on our experience of handling more than two thousand arthroscopy surgeries , we have established that correct understanding of the skin - capsular mismatch minimizes the soft tissue injury and indirectly reduces the operative time . the limitation of this study is that the degree of skin - capsular mismatch could not be shown quantitatively . further studies with a large number of patients and an appropriate measuring method could additionally enhance the quality of the paper .

damage to soft tissues , chondral surfaces , and the menisci may result from imprise or overly aggressive establishment of portals in arthroscopic knee surgeries . in this note , we address the relationship between the skin and the capsule at portal sites according to knee positions . understanding the skin - capsular mismatch may facilitate arthroscopic procedures and indirectly reduce the operation time .

bevacizumab is thought to normalize tumor vasculature and restore the blood - brain barrier , decreasing contrast enhancement and peritumoral edema . antiangiogenic treatment targeting the vegf pathway using bevacizumab causes a rapid decrease in t1 contrast - enhancing tumor parts , with high radiographic response rates ranging between 3060% . however , in one third of all patients , glioblastomas are more prone to progress as nonenhancing tumor after bevacizumab treatment . recently defined rano ( response assessment in neuro - oncology ) criteria for glioma progression recommend fluid - attenuated inversion recovery ( flair)/t2 hyperintensity on mri as a surrogate for nonenhancing tumor ; however , nonenhancing tumor can be difficult to differentiate from other causes of flair / t2 hyperintensity ( e.g. , radiation - induced gliosis , peritumoral edema ) . positron emission tomography using the radiolabeled amino acid o-(2-[f]fluoroethyl)-l - tyrosine ( f - fet pet ) may offer an improvement of the detection of true , i.e. , metabolically active tumor extent of high - grade glioma over and above the information provided by contrast enhancement and flair hyperintensity on mri alone . a 58-year - old patient suffered from aphasia and agitation 4 weeks prior to admission . initial mri revealed a contrast - enhancing , tumor - suspicious lesion in the left temporal lobe , and a biopsy was performed stereotactically . molecular analysis of prognostic factors revealed that the o6-methylguanine methyltransferase promoter ( mgmt ) was not methylated and isocitrate dehydrogenases 1 and 2 ( idh1/idh2 ) were not mutated . in order to achieve a macroscopically complete tumor resection , a fluorescence - guided resection using 5-aminolevulinic acid ( 5-ala ) after surgery , fractionated external radiation therapy was initiated . within 2 weeks after tumor resection , after stratification for molecular markers , the patient was treated with concomitant radiotherapy up to a dosage of 60 gy , and afterwards with adjuvant bevacizumab every 2 weeks over 8 months . due to a severe allergic reaction , the combination therapy with irinotecan had to be discontinued after the first application . during bevacizumab treatment , clinical controls and follow - up mris in 8- after 8 months of single bevacizumab therapy , hemiparesis of the right side and deterioration of the aphasia occurred . 1b , encircled within black lines ) is significantly larger , suggesting signs of nonenhancing tumor progression during bevacizumab treatment [ 2 , 5 ] . to further evaluate the mri results , f - fet pet ( fig . metabolically active tumor volume of f - fet pet uptake at a threshold = 1.6 and volume of the hyperintense flair signal were calculated [ 6 , 7 ] ( fig . 1d , encircled within white lines ; e ) show that the volume of the flair hyperintensity is considerably larger than the metabolically active tumor volume . 1f ) , the volume of the flair hyperintensity ( grey ) shows no correspondence in size and spatial configuration to the f - fet pet metabolically active tumor volume . moreover , in comparison to the flair hyperintensity , f - fet uptake at a threshold = 1.6 ( red ) reveals additional subareas of metabolically active tumor ( fig . 1f ) in transaxial , coronal , and sagittal orientation . for treatment of tumor recurrence , chemotherapy with dose - intensified temozolomide clinically , chemotherapy response could not be observed ; during follow - up an exacerbation of the aphasia and hemiparesis occurred . due to a severe impairment of the patient 's clinical condition , palliative care was initiated 3 months after tumor recurrence . the differences in mri and f - fet pet might be explained by a complex morphologic heterogeneity of glioblastoma , leading to difficult interpretation of standard mri sequences . there are highly malignant tumor parts with and without contrast enhancement , and , furthermore , t2-based signal hyperintensity is a combination of infiltrating tumor cells , necrotic areas , tumor edema , and treatment - related leukoencephalopathy , e.g. , due to radiotherapy . in contrast , f - fet pet shows metabolically active tumor independent of any anatomic or pathophysiologic changes and may therefore reflect tumor extension more accurately than mri . in this context , localization of the f - fet pet metabolically active tumor does not always match the pathological findings seen on mris . for example , in a small group of patients with recurrent high - grade glioma treated with bevacizumab and irinotecan , which partially responded according to rano criteria without response based on f - fet pet imaging findings , tumor progression could be detected earlier by f - fet pet . in summary , during bevacizumab treatment , f - fet pet in addition to contrast - enhanced and flair-/t2-weighted mri might add important information for assessment of tumor recurrence , as compared with standard mri alone .

antiangiogenic treatment using bevacizumab may cause difficulties in distinguishing between antivascular and true antitumor effects when using mri response criteria based on changes of contrast enhancement ( i.e. , macdonald criteria ) . furthermore , more precise tumor response assessment criteria ( i.e. , rano criteria ) , which incorporate nonenhancing t2/flair sequences into macdonald criteria , may be influenced by other causes of t2/flair hyperintensity ( e.g. , radiation - induced gliosis ) . the authors present discrepant mr and [ 18f]fluoroethyl - l - tyrosine pet imaging findings in a patient with bevacizumab treatment failure .

ectopic thyroid is a developmental anomaly of the thyroid gland in which thyroid tissue is present at sites other than its normal location in the neck . it is usually uncommon to have multiple locations of ectopic thyroid tissue ( ett ) present simultaneously . though ett can manifest at any age it is mostly noted at adolescence . about 65 - 80% of cases manifesting with ett are females . lingual thyroid is the most common ectopic location for the thyroid accounting for 90% of cases . it is very rare to have two ectopic foci of thyroid tissue simultaneously and only a very few cases of dual ectopy have been reported in the world literature . we report a case of dual thyroid ectopy , wherein 99 m - technetium ( tc ) pertechnatate hybrid single - photon emission computed tomography / computed tomography ( spect / ct ) played an important role in demonstrating multiple ectopia of the thyroid gland . the present case report is about a 10-year - old girl who presented with anterior midline neck swelling since 6 months , which was gradually increasing in size . on evaluation , she was found to have hypothyroidism without evidence of growth retardation . on local examination , a swelling 2 2 cm size , cystic , well - defined with a smooth surface neither compressible nor reducible was palpated just below the hyoid bone , which was moving up with protrusion of tongue and with deglutition , but was not trans - illuminant . serum biochemistry revealed t3 = 86 ng / dl ( normal , 70 - 200 ng / dl ) , t4 = 4.3 g / dl ( normal , 4.5 - 12.5 g / dl ) and thyroid - stimulating hormone level of 27 iu / ml ( normal , 0.5 - 4.7 iu / ml ) . with a provisional diagnosis of sub - hyoid thyroglossal cyst , patient was referred for a technetium thyroid scan to rule out functioning thyroid tissue in the cyst . tc - pertechnatate thyroid scan revealed a midline focus of tracer uptake corresponding to the palpable neck swelling suggestive of ectopic functioning thyroid tissue . in addition , a small linear streak of tracer activity was seen to extend superiorly [ figure 1 ] from the ett in the neck . spect / ct of the neck revealed ett on the right side of the thyroid cartilage , in the sub - hyoid location ; in addition to this another ectopic rest of thyroid tissue was noted in the midline floor of the mouth . tracer was also seen to extend along a tract , which was well - appreciated on sagittal section [ figure 2 ] . 99m - technetium - pertechnatate thyroid scan anterior ( a ) and lateral ( b ) view revealing a midline focus of tracer uptake corresponding to the palpable neck swelling suggestive of ectopic functioning thyroid tissue . in addition , a small linear streak of tracer activity was seen to extend superiorly ( a - arrow ) computed tomography ( ct ) and hybrid single photon emission computed tomography / ct images of the neck revealing ectopic thyroid tissue on the right side of the thyroid cartilage , in the sub - hyoid location ( a and b ; arrow ) and in the midline floor of the mouth ( c and d ; broken arrow ) . furthermore , tracer was seen to extend along a tract which was well appreciated in sagittal images ( e and f ) its original position is marked by the foramen caecum at the junction of anterior two - thirds and posterior one - third of the tongue . an evagination appears between the first and second pharyngeal pouch at 4 weeks of gestational age which lengthens to form a tube and descends inferiorly and anteriorly to pass anterior to the hyoid bone and forms the lateral lobes of the thyroid . the pathway from the pharynx to the anterior neck is marked by the thyroglossal duct . hybrid spect / ct may be useful in accurate localization of ectopic thyroid rests and to differentiate from other causes of midline cervical masses . in our case report , planar thyroid scintigraphy showed single ectopic thyroid at the sub - hyoid location but on hybrid spect / ct , we found dual ectopia along the thyroglossal tract . in evaluating thyroid ectopy , spect / ct may thus have an incremental value over planer scintigraphy for accurate localization of ectopic thyroid rests which may influence patient management .

ectopic thyroid tissue ( ett ) refers to the presence of thyroid tissue in locations other than the normal anterior neck region between the second and fourth tracheal cartilages . multiple ectopia of the thyroid is extremely rare . here we report a case of 10-year - old girl with anterior midline neck swelling and hypothyroidism with dual ectopia of thyroid gland without orthotopic thyroid gland . planar 99 m - technetium pertechnatate scan identified ett corresponding to the palpable neck swelling . single photon emission computed tomography / computed tomography ( spect / ct ) demonstrated ett in two locations , one corresponding to the palpable mass and another in the in the sublingual location . this case thus demonstrates the important role of hybrid spect / ct in the identification of dual ectopia along the thyroglossal tract .

patients with bilateral temporal bone fractures that run through the otic capsule may lose their hearing . the present paper describes a case of cochlear implantation after a bilateral temporal bone fracture causing bilateral profound hearing loss and ossification of one cochlea . however , attempted surgery on the left ear failed due to ossification of the cochlea . a subsequent postoperative ct scan showed patency of the right cochlea and the patient was successfully implanted on that side at a second stage . a 52 year - old male with bilateral deafness and right facial nerve palsy presented to our clinic . the patient who was referred for consideration of cochlear implantation had a history of head injury 8 months before . an initial ct scan 2 months following his injury showed a transverse temporal bone fracture on the right side and a longitudinal one on the left side . the left ear was chosen for implantation as the injury was less severe on that side . right transverse fracture ( black arrow ) and left longitudinal fracture ( white arrow ) preoperative scan demonstrating patency of both cochlea when surgery was attempted , cochleostomy revealed complete obliteration of the scala tympani . efforts to insert the electrode through the scala vestibuli were also unsuccessful . the procedure was abandoned and a subsequent high resolution ct scan showed complete ossification of the left cochlea and but a patent cochlea on the right ( fig . 3 and 4 ) . twenty days following the initial attempt , a right cochlear implantation was successfully performed . following mapping , programming , and aural rehabilitation , the patient was able to understand speech and use the telephone . longitudinal fractures begin in the squamous part of the temporal bone , extend through the external auditory canal , across the middle ear , and terminate near the carotid canal . transverse temporal bone fractures extend across the petrous ridge , with the fracture beginning in the posterior fossa and crossing the otic capsule into the middle ear space . furthermore the fracture might allow communication between the subarachnoid space and the middle ear , increasing the risk of meningitis or involve the internal auditory meatus , causing possible damage to the cochlear nerve . there are few reports in the english literature of successful cochlear implantation in patients with deafness from temporal bone fractures ( 1 - 3 ) . histopathological studies of temporal bone fractures reveal a variety of injuries such as complete destruction of the organ of corti and stria vascularis , loss of hair and ganglion cells , hemorrhage into the cochlear duct , and labyrinthitis ossificans ( 3,4 ) . as reported by nadol et al ( 5 ) a number of ganglion cells may survive in temporal bone fractures . nadol reports an average survival of only a third of the ganglion cell population after temporal bone fractures . however , there is no strong evidence in literature showing a correlation between ganglion cell counts and auditory function after cochlear implantation . ( 6 ) although fractures of the otic capsule usually heal by fibrosis , sometimes they may be complicated by cochlear osteoneogenesis and result in ossification of the cochlea . the most frequent site of ossification is the basal turn of the scala tympani ( 2,3 ) . in such cases successful electrode insertion has been achieved by drilling out the obliterated portion of the basal turn ( 2 ) or by performing a partial labyrinthectomy to gain access to the round window and allow electrode insertion ( 7 ) . postoperative results in these patients were comparable to those with a conventional scala tympani insertion . in addition , the sooner the implantation was performed , the greater the possibilities for a successful insertion , as there is less time for ossification ( 1 ) . the exact time of ossification of the cochlea following a temporal bone fracture is not known . as illustrated in our case , it is best to obtain temporal bone imaging immediately prior to implantation to rule out labyrithitis ossificans or other abnormalities , especially when there has been a long time delay between injury and surgery . in our case the initial ct scan failed to demonstrate ossification of the cochlea , showing patency of both cochleas . according to siedman et al ( 9 ) ct scan may miss up to 22% of cases of cochlear luminal obstruction , subsequently found at surgery . in addition , as reported in literature mri scan ( t2-weighted images ) is becoming an increasingly significant imaging modality in investigating cochlear patency . its role in differentiating between cochlear fluid , fibrosis or bone formation is under evaluation in a number of centers . with t2-weighted image it is possible to assess the cochlea , the semicircular canals , the internal acoustic meatus and the cerebello - pontine angle . as shown by laszig et al the detection of the absence of fluid seems to be a more sensitive method for determining patency of the cochlea ( 10 ) . temporal bone fractures may lead to ossification of the cochlea and implantation must therefore be performed as soon as possible following injury . both hrct and mri scans are required to assess patency of the cochlea , involvement of the internal auditory meatus or possible communication between the subarachnoid space and the middle ear . in cases of bilateral fractures , implantation is best avoided on the site of transverse fracture , due to the possibility of direct intracranial communication and risk of meningitis .

patients with bilateral temporal bone fractures frequently suffer profound hearing loss . these patients may benefit from cochlear implantation . displaced fracture lines and ossification of the cochlea might complicate insertion of the electrode array . in the present paper we present a case of a failed cochlear implantation due to ossification of the cochlea , followed by a successful one on the opposite side in a patient who sustained bilateral temporal bone fractures . preoperative imaging may identify these factors , help guide the appropriate surgical approach and choose which side to implant . we aim to highlight the necessity of imaging and discuss the reliability of computed tomography ( ct ) scanning in predicting cochlear patency in cases of temporal bone fracture . possible management options in the case of complex cochlear implantation are also described .

a 70-year - old japanese man with a 50-year , 10 cigarettes / day smoking history presented with hoarseness . chest computed tomography ( ct ) and brain magnetic resonance imaging ( mri ) showed a tumor in the left lung and multiple metastases to the lung , the bone , and brain . among serum tumor markers , neuron - specific enolase ( 68.9 ng / ml ) and the pathology of transbronchial biopsied specimens demonstrated acidophilic tumor cells showing a cord - like distribution with small necrosis and one mitosis in 10 high - power fields ( fig . 1 ) . combined with immunohistochemical ( ihc ) findings of cd56+/chromogranin a+/synaptophysin+ ( fig . 2a ) and a low ki-67 labeling index ( < 20% ) , biopsied specimens stained with hematoxylin and eosin staining show small necrotic foci ( inset ) and tumor cells with fine nuclear chromatin and moderate eosinophilic cytoplasm growing in a trabecular pattern and organoid nesting arrangement . ( c ) fish analysis of the alk locus using a break - apart probe strategy . strongly positive staining for synaptophysin ( bb ) ( a ) and alk ( 5a4 , nichirei biosciences , tokyo ) ( b ) was observed in tumor cells . ( c ) approximately 74% of the tumor cells showed rearrangement at the alk locus . further analyses revealed that anaplastic lymphoma kinase ( alk ) gene rearrangement was observed by both ihc ( fig . the patient was initially treated with carboplatin ( auc5 , day 1 ) and etoposide ( 100 mg / m , days 1 - 3 ) , however , the tumor size remained unchanged after four cycles of chemotherapy . thereafter , he was treated with an alk inhibitor , crizotinib ( pfizer , tokyo , japan ) 250 mg po twice daily , which reduced the tumors in the lung ( fig . 3a and b ) and the brain ( fig . a chest ct scan and brain mri before and during treatment with crizotinib ( a and c : may 2015 , b and d : aug 2015 ) . crizotinib reduced the primary mass ( arrows ) from 26 mm ( a ) to 15 mm ( b ) , and the multiple lung metastases ( arrowheads ) disappeared . crizotinib also reduced the brain metastasis ( arrows ) from 13.3 mm ( c ) to 6.2 mm ( d ) . although alk rearrangement accounts for approximately 5% of non - small cell lung carcinomas ( nsclcs ) , alk rearrangement in neuroendocrine tumors ( nets ) including small cell lung carcinomas ( sclcs ) , large cell neuroendocrine carcinomas ( lcnecs ) , and carcinoid tumors is quite rare ( 1 ) . to date , only four net cases , two sclc cases , one lcnec case and one atypical carcinoid tumor case have been reported to have alk rearrangement ( 2 - 5 ) . in addition , nakamura and colleagues reported that no alk rearrangement was found among 227 nets including 52 carcinoid tumors ( 6 ) . the biopsied tissues in the present case were optimal specimens without crushing for microscopic observation , and alk rearrangement was confirmed using two methods ( ihc and fish ) . we thus contend that the lung tumor in the present case met the criteria of atypical carcinoid tumor , and that this atypical carcinoid tumor also had alk rearrangement . however , a potential limitation of this case is that the pathology of transbronchial biopsied specimens may not reflect the character of the whole tumor . we first treated our patient with chemotherapy according to a previous report ( 7 ) , however , the regimen was ineffective . we then administered crizotinib , and it reduced the tumors in both the lung and brain . however , it was also reported that nets with alk rearrangement may be resistant to alk inhibitors ( 2,3 ) . further analyses are needed to elucidate whether crizotinib may become a key tool for the treatment of carcinoid tumors with alk rearrangement .

this is the first report in which crizotinib , an anaplastic lymphoma kinase ( alk ) inhibitor , reduced an atypical carcinoid tumor with alk rearrangement . a 70-year - old man developed a tumor in the left lung and multiple metastases to the lung and brain . the pathology of transbronchial biopsied specimens demonstrated an atypical carcinoid pattern . combined with immunohistochemical findings , we diagnosed the tumor as atypical carcinoid . alk gene rearrangement was observed by both immunohistochemical ( ihc ) and fluorescence in situ hybridization . he was treated with chemotherapy as first - line therapy , however , the tumor did not respond to chemotherapy . thereafter , he was treated with crizotinib , which successfully reduced the tumors .

colonic duplications are rare congenital anomalies that usually present during the first decade of life . the clinical picture varies according to the location and extent of the lesion , as well as the type of mucosal lining . treatment of choice is complete resection of the duplication that in case of a long tubular duplication of the colon entails total or subtotal colectomy due to shared blood supply of the native bowel and the duplication . a 3-year - old boy presented with complaints of constipation and bleeding per rectum since birth . on digital rectal examination , two lumina were palpable with an intervening septum . barium enema showed long segment colonic duplication with distal communication just above anal verge [ figure 1 ] . meckel 's scan for ectopic gastric mucosa was negative . on exploratory laparotomy , complete colonic duplication from cecum till 2 cm proximal to anal verge both the lumens were of the same caliber and appearance . on gross examination , it was difficult to identify the normal colon and duplication . the common wall between the two was divided at the distal end using a stapling device inserted through the anus . barium enema showing the rectum ( r ) , the duplication ( d ) and common wall ( com ) intra - operative photograph showing the total colonic duplication colonic duplication is a rare entity , though more common in children as compared to adults . gastrointestinal bleeding in bowel duplications can result from angiodysplasia or ectopic gastric mucosa leading to ulceration . however , the bleeding per rectum could have been caused by fissures due to the passage of hard constipated stool . the constipation in the present case possibly occurred due to a fecaloma in the distal part of the duplication causing compression of the normal lumen , thus obstructing the evacuation of the rectum . since the communication between the duplication and the normal large bowel was just proximal to the anal verge , the contraction of the external sphincter possibly allowed the feces to enter the duplication . and , as fecal matter continued to accumulate in the duplication , the constipation became progressive . by dividing the septum between the normal and duplicated distal large bowel , the rectum acquired a single lumen , and hence the evacuation became easy . a large communication between the duplicated and normal large bowel also ensured emptying of the fecal matter that collected in the duplication . limited division of the common wall in case of a complete colonic duplication is a simple , safe , easy , and quick procedure .

colonic duplications are rare congenital anomalies . treatment of choice is complete resection that in case of a long tubular duplication requires total or subtotal colectomy . a simple surgical technique for treatment of complete colonic duplication is described , which avoids the complications of extensive colonic resection .

a 31-year - old male resident of tbilisi , georgia , sought treatment at the infectious pathology , aids and clinical immunology center in early june 2008 , six days after onset of illness . at that time , he was enrolled in an ongoing laboratory - based surveillance study of acute febrile illness ( afi ) . initial clinical symptoms were fever ( maximum axillary temperature 39c ) , arthralgias and myalgias in the lower extremities , back pain , vomiting , and diffuse abdominal pain . vital signs indicated a pulse rate of 87 bpm and blood pressure of 140/80 mm hg . travel history included regular trips to marneuli district ( a rural district south of tbilisi ) . laboratory findings were normal , with the exception of elevated band neutrophils ( 20% , reference range 1%6% ) and lymphocytosis ( 44% , reference range 19%37% ) . c - reactive protein level was elevated ( 48.9 mg / ml , reference range < 6 mg / ml ) , and decreased total protein ( 62 mol / l ) was noted on day 9 of illness , along with a decrease in urine output . proteinuria ( 3 g/24 h ) , microscopic hematuria , and elevated blood pressure ( 160/90 mm hg ) were also observed . the patient was transferred to the nephrology department of the institute of urology in tbilisi with a diagnosis of acute renal failure . patient s fluid input and output were closely regulated , and his condition improved gradually without dialysis . serum samples were tested for antibodies against a panel of pathogens , including salmonella enterica serovar typhi , s. paratyphi a and b , epstein - barr virus , brucella spp . , coxiella burnetti , hantavirus , rickettsia ( spotted fever and scrub typhus groups ) , west nile virus , tick - borne encephalitis virus , and leptospira spp . all serologic test results were negative except for the test result for hantavirus . to corroborate hospital laboratory diagnostics , serum samples were also tested in a private laboratory in germany by using western blot assay ( recomblot bunyavirus immunoglobulin [ ig]g / igm ; mikrogen , neuried , germany ) and immunofluorescence antibody ( ifa ) assay ( progen , heidelberg , germany ) . results from the western blot assay were negative for puumala igg and positive for hantaan igg and dobrava igm . 3 in egypt conducted a hantavirus igm elisa ( focus diagnostics , cypress , ca , usa ) on paired serum samples ( acute - phase sample positive : negative ratio = 8.42 ; convalescent phase = 7.815 , cut - off 1.10 ) cultures were negative . a renal biospy specimen obtained 10 days after disease onset showed acute tubular necrosis with mild - grade arteriolosclerosis ( figure ) . acute tubular necrosis in a renal biopsy specimen of the patient . the patient s general condition started to improve , with increased urine output , resolution of abdominal pain , and normalization of blood pressure ( 120/70 mm hg ) . the clinical presentation and results of serologic tests for this patient who had renal failure but no apparent hemorrhage were consistent with results previously described for hantavirus infection . although the differential diagnosis included other infectious and noninfectious causes of renal failure including leptospirosis , ongoing surveillance of afi in georgia provided the serologic testing capability that enabled clinicians to identify a hantavirus infection as the cause of this illness characterized by high fever and evidence of renal dysfunction ( increased serum creatinine , proteinuria , and hematuria ) . the pathologic changes observed in hantavirus infection presumably result from increased vascular permeability , characterized clinically by elevated hematocrit and decreased serum protein levels . damage to the vascular endothelium along with cytokine - induced renal tubular and interstitial pathologic changes may partially explain renal failure associated with hantavirus infection . although the most commonly noted renal pathologic finding is acute interstitial nephritis , acute tubular cell necrosis is also described . a comparative study of puumala and dobrava renal pathology noted increased intensity and extent of medullary interstitial capillary injury , hemorrhages , and tubular necrosis among dobrava virus - infected patients ( 9 ) . the strain of the infecting virus is unknown , but serologic results , along with the available information from surrounding countries , implicate dobrava virus as the infecting strain . the relatively mild clinical symptoms of this patient suggest that a variant of dobrava virus , or a closely related hantavirus strain that cross - reacts with dobrava , may be the etiologic agent . information about the circulating virus strains , clinical manifestation , rodent hosts , and disease prevalence are lacking for the caucasus .

we describe a laboratory - confirmed case of hantavirus infection in the republic of georgia . limited information is available about hantavirus infections in the caucasus , although the infection has been reported throughout europe and russia . increasing awareness and active disease surveillance contribute to our improved understanding of the geographic range of this pathogen .

pocket infection of pacemakers and implantable cardioverter defibrillators ( icd ) is usually treated by removing the device and re - implanting it in a new site following treatment . we present a complicated case of pocket infection , treated without the removal of the device . a 78-year - old man , a known case of hypertension , hyperlipidemia , and coronary artery bypass grafting ( cabg ) 8 years previously , presented with complete heart block plus unstable atrial and slow ventricular escape rhythm ( left bundle branch block pattern ) and received a permanent pacemaker ( vvir mode ) . he was subsequently admitted several times due to decompensated heart failure ( left ventricular ejection fraction = 20% and moderate mitral regurgitation ) , which led to his finally receiving a three - chamber cardiac resynchronization therapy ( crt ) device . afterwards , the patient developed a large hematoma around the pocket ; the sutures were , therefore , removed for drainage . the removal of the sutures was followed by the development of extended skin necrosis with massive purulent secretions and cellulitis around the incision site ( figure 1 ) . the patient was afebrile and symptomless , white blood cell count was 10050/mm , blood culture was negative , lab tests were normal , and there was no evidence for endocarditis . surgical tissue debridement was performed and staphylococcus epidermis grew in its culture , suggesting contamination . pocket infection being regarded as the primary diagnosis , the patient was treated with intravenous antibiotics ( vancomycin , gentamicin , and ciprofloxacin ) , which cleared the inflammation and resolved the cellulitis . the existence of a massive soft tissue defect prompted the consultant surgeon to suggest relocating the generator and applying a tissue graft . be that as it may , the patient was device - dependent and the generator could not be transferred to another place as it was impossible to re - implant the left ventricle lead . additionally , further morbidity was reduced by eradicating the infection and curing the wound conservatively through keeping the generator outside the body ( figure 2 ) . the wound was thereafter irrigated with zinc sulfate lotion ( 1% ) daily and dressed with silver sulfadiazine ointment and alginate comfeel dressing . the generator remained out of the pocket for 5 weeks in a sterilized condition ; and during the last 2 weeks , the edges of the wound were approximated gradually by sutures . after the wound had completely healed , the generator was successfully re - implanted in its prior location after sterilization with decocept. the surgical wound healed rapidly , the patient had no problem during a 10-month follow - up , and the crt worked properly ( figure 3 ) . infection of a medically inserted device is usually treated with appropriate antibiotics ; and if there is not a rapid response , the removal of the foreign body is advocated . otherwise , pocket infection carries a high risk of mortality and morbidity.1 , 2 however , some reports have suggested that conservative treatment can be successful too . in such instances , leads are preserved and the icd is replaced following debridement , irrigation , and antibiotic therapy.3 in the case of our patient , the removal of the entire pacing system could probably predispose the patient to unexpected events . he suffered from heart failure ( new york heart association functional class iii ) and the coronary sinus lead was surgically implanted with difficulty in the first attempt ; the patient was , as a result , considered a high - risk case for another re - implantation . on the other hand , skin graft could pose a risk of anesthesia and surgery , a risk which seemed to be too difficult for the patient to tolerate . consequently , normal wound healing rather than skin graft application was opted for with a view to providing the patient with the opportunity to benefit from the crt function . we would posit that those who are susceptible to cardiac complications , especially the elderly population , can be treated with a conservative method that includes broad - spectrum antibiotics , debridement , and modern dressing . we would suggest that a conservative treatment of pacemaker pocket infection in those who have no signs of other infections such as endocarditis or septicemia could be a proper treatment , although it is needs more time and medical care . opting for such treatment must be at the doctor s discretion based on the patient s condition .

pocket infection of a cardiac device is usually treated by removing the device and re - implanting it in a new site after complete treatment of the infection . this report illustrates a complicated case of pocket infection in the wake of the implantation of a permanent pacemaker ( cardiac resynchronization therapy ) . the patient was treated conservatively through daily irrigation and dressing , broad - spectrum antibiotics , and debridement without the device being removed ; the generator was kept out of the pocket for 5 weeks and then re - implanted in the same location successfully.the method of treatment presented herein can be of value , not least in the elderly population who might experience life - threatening events following the replacement of their cardiac devices .

a coronary av fistula consists of a communication between a coronary artery and a cardiac chamber , a great artery or the vena cava . in 1865 , krause1 ) described a congenital av fistula , and this type of fistula makes up a part of the congenital anomalies of the coronary arteries , which occur in 1% to 2% of the population2 ) . multiple fistulas occur in 10.7% to 16% of all coronary av fistulas , whereas both coronary arteries are involved in about 5%2 - 4 ) . this report describes the bilateral involvement of a coronary av fistula , and this was coexistent with variant angina . a 64-year - old woman presented with complains of progressive dyspnea on effort and also chest discomfort she 'd had for 10 days . she has had no particular prior medical / surgical history . on the physical examination , the pulse rate was 70 beats / min and the blood pressure was 130/70 mmhg . the electrocardiogram displayed normal sinus rhythm ; the chest x - ray showed no signs of pulmonary hyperemia or mass lesions , and the central shadow was normal ( cardiothoracic ratio=0.49 ) . the transthoracic echocardiography demonstrated a normal finding with good lv systolic function and the stress transthoracic echocardiography also showed no regional wall motion in the left ventricle . 24-hour holter monitoring showed no specific findings except for rare ventricular premature complexes ( vpcs ) . there were no atheroma and stenotic lesions on the coronary angiogram , but it did reveal bilateral coronary av fistula coexistent with vasospatic angina . one fistula originated from the distal septal branch of the left anterior descending artery and the other originated from a distal right ventricular branch of the right coronary artery and the latter drained into the right ventricle ( figure 1 ) . the spasm study found that the middle to distal left anterior descending artery had diffuse 90% vasoconstriction with chest pain after a left intracoronary ergonovine injection . for coronary av fistula , the site of origin of the fistula is from the right coronary artery in 50% to 58% of patients , from the left anterior descending artery in 25% of patients , from the circumflex artery in 18.3% of patients , from the diagonal branch in 1.9% of patients and from the left main coronary artery ( lmca ) or the circumflex - marginal branch in 0.7% of patients , respectively2 , 3 , 5 , 6 ) . in the above mentioned studies , the coronary av fistula drainage site was the pulmonary artery in 29.8% to 43% of patients , the right ventricle in 14% to 40% of patients , the right atrium in 19% to 20.2% of patients , the left ventricle in 5.8% to 19% of patients and the left atrium in 5% of patients . twenty percent of the patients with congenital coronary av fistula have other congenital or acquired heart disease2 - 4 , 7 - 9 ) . for the adults with av fistula angina pectoris and myocardial infarction that are due to coronary artery steal occur in 3% to 7% of av fistula patients . congestive heart failure develops in 19% of these patients , and endocarditis in the fistula occurs in 20% . only one case has been reported with the coronary av fistula coexistent with variant angina10 ) . the treatment of a coronary av fistula depends on its presentation and the magnitude of the pulmonary - to - systemic flow . yet it has been suggested that surgical ligation4 ) , with or without the use of extracorporeal support , could be a form of treatment due to the natural history of the disease , which is not always benign , and also due to the low incidence of spontaneous fistula closure in patients with a large coronary av fistula . in conclusion , we report here on a case that showed the very rare occurrence of bilateral coronary av fistula that was coexistent with variant angina .

a coronary arteriovenous ( av ) fistula consists of a communication between a coronary artery and a cardiac chamber , a great artery or the vena cava . it is the most common anomaly that can affect coronary perfusion . yet bilateral involvement of a coronary fistula , constitutes an uncommon subgroup of coronary av fistulas . we herein report on a case of bilateral coronary av fistula that was coexistent with variant angina originating from the distal right ventricular branch of the right coronary artery and the distal septal branch of the left anterior descending artery , and the latter drained into the right ventricle .

this valvular defect is often detected in infancy and tends to be incompatible with life . in asymptomatic patients , the presence of a unileaflet mitral valve is most commonly due to a severely hypoplastic posterior mitral leaflet . a 22-year - old otherwise healthy man presented to the clinic complaining of recurrent episodes of " heart racing " . prior laboratory testing was not significant for anemia , infection , thyroid or adrenal disease , vitamin deficiencies or electrolyte derangements . a transthoracic echocardiogram revealed a preserved left ventricular ejection fraction of 55% , normal left ventricle size and thickness , normal diastolic filling and an unusual incidental finding of a unileaflet mitral valve . the anterior mitral valve leaflet was markedly elongated , mildly thickened , and occupied the entire closure line of the mitral annulus with no contribution of the hypoplastic posterior leaflet . the coaption zone was intact with no mr ( fig . 1 , 2 , and 3 , supplementary movie 1 and 2 ) . as to his palpitations , he was diagnosed with an underlying anxiety disorder and successfully treated with anxiolytic therapy . congenital malformations of the mitral valve are extremely rare and include congenital mitral stenosis , parachute mitral valve , double orifice of the mitral valve , cleft mitral valve , atresia , and unileaflet mitral valve.1 ) unileaflet mitral valve is the rarest of these congenital anomalies . aplasia of the posterior mitral valve leaflet is responsible for unileaflet mitral valve in the neonatal period and associated with severe mr that is usually incompatible with life without surgical intervention.2 ) existing literature in the field document cases of unileaflet mitral valve in asymptomatic patients of all age groups and mostly result from a severely hypoplastic posterior mitral valve.1)2)3)4)5)6)7 ) the prevalence of this anomaly is estimated to be 1:8800 in german population;2 ) however , the true prevalence is expected to be much lower in the general population . to the best of our knowledge , there have only been 11 reported cases of post - neonatal unileaflet mitral valve in the literature , of which 70% of cases were found to be in asymptomatic patients . the majority of cases were found after detection of a systolic murmur during routine physical examination.1)2)3)4)5)6)7 ) in the aforementioned cases , transthoracic echocardiography ( tte ) revealed a thickened and elongated anterior mitral valve with significant prolapse into the left atrium.2 ) in two of the cases , patients developed severe mr and underwent surgical correction and were found to have hypoplasia of the posterior mitral valve with absence of the chordae tendinae.4)6 ) it was also suggested that 3d echocardiography with transesophageal echocardiography ( tee ) outlines the congenital abnormalities of mitral valve better than tte or only tee.1 ) the prognosis in asymptomatic individuals is unclear ; however , there is a potential for worsening mr with age due to annular dilation . there are no guidelines or recommendations currently for screening or follow - up monitoring of these patients . in our opinion , it may be appropriate to follow - up such patients with annual physical examinations and perform echocardiography if symptoms develop or mr murmur develops / worsens . in conclusion , hereby we represent a rare case of unileaflet mitral valve and review not only the importance of echocardiography in accurate diagnosis , but also highlight the importance of periodic surveillance with echocardiography in early detection of mr in such cases . transthoracic echocardiogram color doppler of the parasternal long axis : no evidence of mitral regurgitation .

unileaflet mitral valve is the rarest of the congenital mitral valve anomalies and is usually life threatening in infancy due to severe mitral regurgitation ( mr ) . in most asymptomatic individuals , it is mostly due to hypoplastic posterior mitral leaflet . we present a 22-year - old male with palpitations , who was found to have an echocardiogram revealing an elongated anterior mitral valve leaflet with severely hypoplastic posterior mitral valve leaflet appearing as a unileaflet mitral valve without mr . our case is one of the 11 reported cases in the literature so far . we hereby review those cases and conclude that these patients are likely to be at risk of developing worsening mr later in their lives .

he described the case of a pregnant woman who suffered intraperitoneal haemorrhage in the absence of trauma ; the source of bleeding was not identified . it occurs more frequently in males than in females at a ratio of 3 : 2 , and most cases occur in later middle age between 55 and 64 years . we report the case of a young male presenting with abdominal apoplexy because of rupture of the short gastric arteries . a 23-year - old male , of polish origin , was brought in by the ambulance to the emergency department . the duration from pain onset to presentation was 15 h. on that morning , he complained of feeling light headed and had experienced an episode of syncope . there was no history of trauma , nausea or vomiting . on examination , he was tender predominantly in the epigastric and right iliac fossa sections of the abdomen . the remainder of the physical examination was unremarkable on presentation , his vital signs were : heart rate of 78/min ; blood pressure of 99/50 ; respiratory rate of 20/min ; temperature of 35.7c ; saturating 100% on air with a gcs of 15/15 . he reported a pain score of 12 on a 4-point scale . in terms of laboratory tests , he had an hb of 89 g / l , an mcv of 92 fl , a platelet count of 146 10/l , a white cell count of 14.5 10/l and a crp of < 1 mg / l . he was placed nil by mouth and started on intravenous fluid resuscitation . however , while awaiting a chest x - ray , he experienced two further episodes of syncope . the operation was converted to an open laparotomy as the source of bleeding could not be identified . rupture of the short gastric vessels is a rare case of abdominal apoplexy . in their review of 85 patients with abdominal apoplexy , carter and gosney the left gastric artery , superior mesenteric artery and splenic artery were listed as the most common sites of rupture in order of decreasing frequency . we have summarized the key features of 13 cases of short gastric vessel rupture identified in the literature ( table 1 ) . table 1:selected cases of short gastric vessel rupture from the literatureauthoragesexchief symptompredispositionoperationresultrege and bhat 14 yearsmaleabdominal pain ligation and splenectomyrecoveryhight and philippart 21 yearsmaleabdominal painvomitingligation and splenectomyrecoverywilliams et al . 21 yearsmaleabdominal painvomitingligationrecoveryho 21 yearsmaleabdominal fullness and diarrhoeavomitingligationrecoverykaplan and hausmann 26 yearsmalechest and abdominal painvomitingligationrecoveryrodero et al . 25 yearsmaleabdominal painvomitingligationrecoveryhsien - pin sun 20 yearsmaleabdominal pain ligationrecovery selected cases of short gastric vessel rupture from the literature most cases of abdominal apoplexy are believed to occur because of a predisposing vascular lesion . in older patients , arteriosclerosis with or without hypertension is believed to be the most common cause . with younger patients , congenital defects in the medial coat of the visceral arteries eight of the 13 cases , we identified occurred following bouts of vomiting ( table 1 ) . hayes et al . proposed that retching may cause a partial , gastric volvulus . the severity of the pain is related to the volume and rapidity of expansion of the blood volume . this is followed by a latent period , devoid of symptoms , lasting from hours to days . in the terminal phase , there is an increasing severity of symptoms and the manifestations of shock become evident . our patient appears to have presented in the early terminal phase . in terms of investigations , computed tomography of the abdomen has been reported as the most useful . in haemodynamically unstable patients , fast ( focused assessment by sonography in trauma ) examination may be useful to detect intra - abdominal haemorrhage . however , it was necessary to convert to laparotomy via an upper midline incision to identify the bleeding point . the choice of intervention is determined by the age and condition of the patient , as well as the site of bleeding . prognosis for the patient is dependent on early diagnosis and intervention . in patients who do not undergo surgery , mortality has historically approached 100% . however , prompt surgical intervention has been described to reduce mortality to 8.6% , especially where a definite bleeding point can be located and ligated . diagnosis of abdominal apoplexy is dependent on maintaining an awareness of the condition in one 's differential diagnoses . unfortunately , lack of suspicion continues to delay diagnosis , worsening the prognosis for the patient . one should suspect abdominal apoplexy in individual presenting with atypical abdominal pain and signs of shock . vomiting , hypertension and arteriosclerosis should further raise suspicion of possible vessel rupture . in cases where abdominal apoplexy is suspected

abdominal apoplexy or idiopathic spontaneous intraperitoneal haemorrhage is defined as the presence of free blood within the peritoneal cavity . non - traumatic and non - iatrogenic causes may cause abdominal apoplexy . it has a variable clinical presentation , with abdominal pain being an early and non - specific symptom . we report a rare case of a 23-year - old male with abdominal apoplexy because of rupture of the short gastric artery . he presented to our department with abdominal pain . later , he developed signs of shock , and was found to have haemoperitoneum on laparotomy . we ligated the short gastric artery , which was the bleeding source , and he had an uneventful postoperative course . we also review the literature on existing cases of short gastric vessel rupture .

foreign bodies that could lead to subsequent perforation or obstruction of the gastrointestinal tract are swallowed , usually accidentally , by children or adults . for the vast majority of patients , treatment is conservative , allowing safe passage of these objects through the intestinal tract . morbidity associated with ingestion is rare , and depends on the type of foreign body ingested . the authors report the case of a rare intestinal complication caused by mischievous ingestion of magnets . a 19-year - old mentally challenged woman presented with abdominal pain of 3 days duration . laboratory investigation results were unremarkable , including normal white blood cell and differential cell counts . a plain abdominal radiograph showed the shadow of a string of small beads in lower abdomen ( fig . abdominal computed tomography scan revealed foreign bodies of probable metallic origin within the small bowel . as there was no evidence of serious complications , initially , expectant treatment was planned awaiting spontaneous passage . however , after 2 days , the shadow of the foreign bodies on serial radiographs remained unchanged , and then , explorative laparotomy was performed . at laparotomy , an adherent jejunal loop containing the foreign bodies was found 20 cm distal to the ligament of treitz ( fig . further exploration revealed that these objects were 19 beads of a magnetic necklace which had come in contact with each other , compressing the interposed bowel walls , resulting in necrosis and the formation of a jejuno - jejunal fistula ( fig . 3 ) . segmental resection of the fistula formed a jejunal loop and an incidental appendectomy was performed . on inquiring of the patient 's mother of any possible inciting event , she recalled that the patient had played with a magnet necklace during a visit to her cousin 's home 2 weeks prior . her postoperative recovery was uneventful , and she was discharged from the hospital 9 days after surgery . in korea , japan , and china , magnets are employed as a traditional remedy to relieve stiffness in the shoulders and neck , as well as to improve peripheral circulation . unfortunately , in these countries , magnets are easily accessible in local pharmacies , and are produced in a small enough size to swallow , passing through the alimentary tract after accidental ingestion . in many of the toys , the magnets are embedded in plastic parts , though they become easily detached . psychiatric illness in older children and adults , as our case , may also be a risk factor for ingestion of foreign bodies . possible predisposing psychological conditions include autism , developmental delay , mental retardation , attention deficit hyperactivity disorder , angelman syndrome , neurosis , reactive attachment , and anxiety . so parents and doctors should be alerted to the dangers of these toys . although the ingestion of a single magnet may not be problematic , multiple magnets can pass into the lumen of the intestine separately or in groups , and then attract each other , holding the intestinal walls between them . the affected areas of the walls then become compressed and necrotic , resulting in intestinal perforation or fistula . moreover , if the mesenteric vessels are involved between the walls , intraperitoneal hemorrhage may also occur . in an experimental analysis of the actual magnets removed from a patient , the force produced by any two magnets is inversely proportional to the square of the distance between them . this translates to a dramatic drop in force with increasing distances between any two magnets . one can appreciate how these magnetic forces generate such powerful effects and injuries within the intestines . in our case , the magnetic attraction between the components of the necklace resulted in the unusual appearance of ingested foreign bodies on plain radiograph . therefore , the causal relationship between the foreign body and intestinal fistula formation was not recognized preoperatively . in conclusion , psychiatric illness in older children and adults , as in our case , may also be a risk factor for ingestion of foreign bodies . although ingested nonmagnetic foreign bodies are likely to be passed spontaneously without consequence , ingested magnets may attract each other through the intestinal wall and cause severe gastrointestinal complications . thus , close observation and early surgical intervention should be considered after ingestion of magnets .

we describe the case of a 19-year - old mentally challenged woman who developed jejuno - jejunal fistula following ingestion of a magnetic necklace . this case report demonstrates the necessity of prompt treatment when the ingested intestinal foreign body is suspected to be multiple magnets , even if there are no sharp edges ; and even when it seems the object could be evacuated spontaneously . ingested magnets are capable of attracting each other across the bowel wall , leading to serious intestinal complications such as pressure necrosis , perforation , fistula formation , or intestinal obstruction .

the cardiofaciocutaneous ( cfc ) syndrome ( omim 115150 ) is a syndrome where patients have multiple congenital anomalies or mental retardation , failure to thrive , psychomotor delay , a characteristic face , congenital heart defects , and abnormalities of the skin , eyes , gastrointestinal tract and central nervous system . the syndrome was first described 20 years ago by reynolds et al in eight children . additional reports soon followed and , according to a recent review , about 59 patients have been reported , providing the basis for an accurate delineation of the phenotypic spectrum of the syndrome . nevertheless , a question has lingered for many years whether cfc is a unique and separate condition , or a variant of the noonan syndrome ( omim 163950)[39 ] or of the costello syndrome ( omim 218040 ) . a useful diagnostic approach was provided with the creation of a cfc index based on 82 clinical traits and their frequencies in the population with the cfc syndrome . however , matters changed radically only with the discovery of different genes whose mutations cause each one of these syndromes : the protein tyrosine phosphatase shp-2 gene ptpn11 for noonan syndrome , hras for costello syndrome , and kras , braf , mitogen - activated protein / extracellular signal - regulated kinase mek1 and mek2 for cfc . one - year - old indian boy was referred to our center at 8 months of age with some dysmorphic features and developmental delay for evaluation . he was a product of a full - term normal delivery with a birth weight of 3.6 kg ( more than the 95 percentile ) . developmental history showed a delay from the beginning with the child unable to crawl , sit or hold objects in hand or produce any sounds like the normal children . family history revealed the child being the third sibling after two normal children and two early abortions . clinically his weight was 10 kg ( 90 percentile ) , height 73 cm ( 75 percentile ) , and head circumference 44.5 cm ( 25 percentile ) . dermatological evaluation showed a dysmorphic child with asymmetrical face , bitemporal narrowing and prominant metopic sutures , epicanthic fold , wide mouth , sparse hair , and absent eye brows [ figures 1a c ] . his skin was dry and mildly hyperkeratotic ; eczematous lesions were found on his neck and extremities , keratosis pilaris on his arms [ figure 2 ] , seborrheic dermatitis on his scalp , and miliaria on his back . dysmorphic face with asymmetry , bitemporal narrowing and prominant metopic sutures , epicanthic fold , wide mouth , sparse hair and absent eye brows close - up view of facial features dry and mildly hyperkeratotic skin . eczematous lesions found on his neck and extremities , keratosis pilaris on his arms there was an evidence of a vascular hemangiomatous plaque measuring 5 cm3.5 cm involving the medial aspect of right lower limb [ figure 3 ] . a vascular hemangiomatous plaque involving the right lower limb the results of routine hematological examination , blood biochemistry analysis , urinalysis , thyroid function tests , and serum zinc level assessment were within the normal ranges . mri scanning of the brain showed bifrontal subdural t2 hyperintense areas plus prominence of the frontotemporal subarachnoid space and corpus callosum hypoplasia . cfc syndrome is a sporadic developmental disorder involving characteristic craniofacial features , cardiac defects , ectodermal abnormalities , and developmental delay . the syndrome is caused by gain - of - function mutations in four different genes braf , kras , mitogen - activated protein / extracellular signal - regulated kinase mek1 and mek2 , all belonging to the same ras extracellular signal - regulated kinase ( erk ) pathway that regulates cell differentiation , proliferation and apoptosis . it is characterised by failure to thrive , relative macrocephaly , a distinctive face with prominent forehead , bitemporal constriction , absence of eyebrows , hypertelorism , downward - slanting palpebral fissures often with epicanthic folds , depressed nasal root and a bulbous tip of the nose . the cutaneous involvement consists of dry , hyperkeratotic , scaly skin , sparse and curly hair , and cavernous hemangiomata . most patients have a congenital heart defect , most commonly pulmonic stenosis and hypertrophic cardiomyopathy . neurological involvement in the cfc syndrome is extensive , and can involve functions of the cortex , brain stem and ventricular system . we established the diagnosis in our patient based on typical dysmorphic features concordant with cfc syndrome . noonan syndrome and costello syndrome , especially the former , can be phenotypically similar to cfc syndrome and should be excluded . noonan syndrome differs by less severe psychomotor delay bordering to normality , low posterior hairline with thick hair , cubitus valgus , neck abnormalities , ectodermal involvement characterized by nevi , caf - au - lait spots , familial occurrence . coarse face , nasal and/or anal papillomata and a predisposition to child tumors such as neuroblastoma , redundant skin of hands and feet with deep palmar and plantar creases , elbow joint limitation .

the cardiofaciocutaneous ( cfc ) syndrome is a condition of sporadic occurrence , with patients showing multiple congenital anomalies and mental retardation and characteristic dysmorphic features . we , thus , report a rare case of this syndrome in a 1-year - old child who presented with typical features of cfc syndrome .

gombocz and colleagues performed a prospective , randomized , double blind study in 40 patients undergoing cardiopulmonary bypass ( cpb ) . they investigated the anti - inflammatory potential of dextran-70 to modulate systemic inflammatory response syndrome ( sirs ) and myocardial ischemia / reperfusion ( i / r ) injury following cardiac operations . interestingly , they could demonstrate that the infusion of dextran-70 before and after cpb reduces inflammation and cardiac troponin i release . the potential deleterious effects of coronary artery bypass grafting ( cabg ) are well investigated under various conditions including cpb and off - pump coronary artery bypass ( opcab ) . this includes the systemic inflammation response induced by contact between immune competent cells and the extracorporal circuit , the ischemia - reperfusion injury of several organs , and the potential endotoxemia after splanchnic hypoperfusion and consecutive damage of the mucosal barrier . it is well known that in low - risk patients the inflammatory response after cpb is less pronounced . avoiding cpb might improve the outcome even in elderly patients with higher morbidity and might lead to good long - term results . nevertheless , the use of cpb is an essential requirement in certain cardiac surgery patients and routinely performed in cardiac surgery . the inflammatory response to cpb is accompanied by an increase in body temperature , leucocytosis and tissue oedema as well as an increased release of cytokines such as interleukin-6 ( il-6 ) and il-10 . this was the rationale for investigations of immune modulation by corticosteroids , cyclooxygenase inhibitors , complement directed therapies , and adhesion molecule blockade . the need for further studies was demonstrated by new insights regarding the therapy with aprotinin . recently , it was shown that this widely used drug in cardiac surgery is associated with an increased risk of death even in long - term follow up after five years . gene array analysis revealed that leukocytes overexpress adhesion and signalling proteins after cpb which may lead to succeeding tissue inflammation . previously , an anti - inflammatory effect of dextran could be demonstrated in experimental settings . steinbauer and colleagues showed in ischemia - reperfusion injury in striated muscle , using intravital microscopy , that dextran attenuates postischemic leukocyte rolling in a molecular weight dependent manner . in this context , the study by gombocz and colleagues yields interesting aspects on the immune modulation by dextran-70 in patients undergoing cabg . using dextran-70 infusion in the early post - cpb phase after 24 hours procalcitonin as well as cardiac troponin i and soluble adhesion molecules were found to be lower using dextran-70 . thus , this study suggests that compared to gelatine , dextran-70 reduces the inflammatory response in patients after cpb . some limitations of the study by gombocz and colleagues need to be addressed : the single centre design including a small number of patients and a short observation period of approximately two days . nevertheless , the authors succeeded to further the exciting area of peri - operative inflammation in cardiac surgery . as so often , further investigations are warranted to evaluate the effects of dextran-70 treatment in cardiac surgery . these trials need to be limited to high - risk patients most likely to experience benefit by anti - inflammatory therapies . additionally , a combination of plasma inflammatory mediators and gene array analysis may lead to the identification of patients being more susceptible to harmful effects of cpb . cabg = coronary artery bypass grafting ; cpb = cardiopulmonary bypass ; il = interleukin ; i / r = ischemia / reperfusion ; opcab = off pump coronary artery bypass ; sirs = systemic inflammatory response syndrome . gm has done paid consultation and verbal presentations for b braun melsungen ag , germany . gm has performed research projects in collaboration with b braun melsungen ag and has thereby received other funding in the past . gm has also received fees for presentations and funds for performing research projects from serumwerke bernburg , germany .

potential deleterious effects of cardiopulmonary bypass ( cpb ) and cardioplegic cardiac arrest are known to influence outcome . the inflammatory response after cpb may have unfavourable effects especially in high - risk patients , for example , the very elderly . thus , to blunt the release of pro - inflammatory mediators seems to be a promising approach . so far , numerous attempts at immune modulation have been performed . however , the management of cardiac surgery patients needs further improvement . in this context , gombocz and colleagues investigated the potential anti - inflammatory effect of dextran-70 . their results suggest that compared to gelatine , dextran-70 reduces the inflammatory response in patients after cpb .

despite of extreme diversity in the deep sea environment , only a few kinds of microorganisms have been isolated and identified . most of them are reported to belong to phylum of bacteroidetes , , . there are only four recognized species of the genus sunxiuqinia , a member of the phylum bacteroidetes ; sunxiuqinia elliptica , sunxiuqinia rutila , sunxiuqinia faeciviva and sunxiuqinia dokdonensis . among them , s. elliptica and s. rutile were isolated from shallow sea sediment in a sea cucumber farm and at nagasuka fishery harbor , respectively . in contrast , s. dokdonensis and s. faeciviva were isolated from deep sub - seafloor sediment at a depth of 900 m below the seafloor off the west part of dokdo island and at a depth of 247.1 m below the seafloor off the shimokita peninsula , respectively , , , . based on difference of sampling depth this is the first report of the whole genome sequence of the genus , sunxiuqinia . this report would be helpful to extend our understanding about the genome and general characteristics of sunxiuqinia for future studies of marine environment . dna was extracted using bead - beating technique and sequenced on hiseq 2000 ( illumina ) . de novo genome assembly was performed using clcbio clc genomics workbench v7.5 and annotation was conducted on rast . seed viewer was used for subsystem functional categorization of the predicted open reading frames ( orfs ) and for visualization . whole genome sequencing has generated and assembled into 225 contigs and the estimated genome size is 4,962,831 base pairs . the n50 of the genome is 43,403 bp and the g + c content of 45.4% . the genome was predicted to contain 4,550 coding sequence , 47 trnas and 3 rrnas . most of the annotated genes were involved in carbohydrate metabolism ( 378 ) , amino acids and derivatives ( 336 ) , protein metabolism ( 228 ) , cofactors , vitamins , prosthetic groups and pigments formations ( 184 ) , rna metabolism ( 129 ) ( fig . 1 ) , which is thought to be necessary to maintain the bacterial activity in the harsh environment like seafloor sediment . these genetic traits of marine isolate of sunxiuqinia will provide a clue to the subsequent study of bacteria in the unique ocean ecosystem . this whole genome shotgun project has been deposited at ddbj / embl / genbank under accession no . lgia00000000 .

sunxiuqinia dokdonensis dh1 t was isolated from deep sub - seafloor sediment at a depth of 900 m below the seafloor off seo - do ( the west part of dokdo island ) in the east sea of the republic of korea and subjected to whole genome sequencing on hiseq platform and annotated on rast . the nucleotide sequence of this genome was deposited into ddbj / embl / genbank under the accession lgia00000000 .

serotonin toxicity ( st ) is a rare clinical state , which may be serious and life - threatening . the most common reasons are usage of multiple serotonergic drugs together or combination of one serotonergic agent and one monoamine oxidase ( mao ) inhibitor . isoniazid ( inh ) is a weak mao inhibitor and also may affect cytocrome enzyme system . st due to combination of isoniazidand essitalopram was rarely reported before . to suspect from the state and history of serotonergic drug usage are main factors for diagnosis . although clinical signs of st on nervous system are well - known , no specific electrophysiological finding was demonstrated before . here , we report a case of serotonin toxicity due to the combination of essitalopram and isoniazid . we also observed transient electromyographic findings , which may probably be due to serotonergic activity . a forty - three - years - old female developed acute onset fever and confusion . she was receiving corticosteroids with the diagnosis of takayasu arteritis for the recent three years . she was also taking isoniazid cause of a suspicious positive result in tuberculin skin test ( ppd ) for the last three months . neurological examination revealed fever , rigidity , clonus , bilateral positive babinsky and hoffman signs , and hyperactive deep tendon reflexes . cranial and spinal magnetic resonance imaging ( mri ) showed no abnormalities nor contrast enhancement . nerve conduction studies were normal , but needle electromyography ( emg ) demonstrated spontaneous neurogenic changes ( fibrillation and positive waves ) in her extensor digitorum brevis ( edb ) , tibialis anterior ( ta ) , and gastrocnemius muscles . we learned that she had been taking essitalopram 20 mg / day for last month from her relatives . with the help of this medical history , she was diagnosed as serotonergic toxicity , essitalopram was stopped , and the patient was hydrated . in her follow - up , she started to get better on the third day of drug - free period . nowadays , the term serotonin toxicity ( st ) is often referred to serotonin syndrome ( ss ) . there is no specific laboratory or diagnostic test , so the diagnosis is mostly based on the clinical aspects . a history of serotonergic agent or increment in the dosage , four major or three major + two minor symptoms , exclusion of psychiatric , toxic , metabolic and endocrine diseases , and neuroleptic usage were needed for diagnosis . our case fulfilled all criteria and revealed six major symptoms like myoclonus , tremor , rigidity , hyperreflexia , fever , and confusion . although citalopram is widely used because of its favorable safety profile and lack of cytochrome p450 inhibition , it has been reported to cause serotonin toxicity , usually in the setting of an overdose . citalopram is a substrate for cyp2c19 and 3a4 , and isoniazide may inhibit cytochrome p450 ( cyp450 ) isoforms , potent inhibition of cyp2c19 and cyp3a . inh is also a mao inhibitor , so may affect the metabolism of anti - depressant drugs . general management measures include removal of the precipitating serotonergic agent and supportive strategies to control the state . in our case , general supportive treatment to stop essitalopram improved the clinical symptoms and signs of the patient . ss is a generalized toxic syndrome ; therefore , both cognitive and neuromuscular findings may be observed . but , in most of the cases , clinical symptoms are more prominent in lower and distal part of extremities , which also may be a clue for a spinal process . as to our best knowledge the spontaneous denervation potentials that we observed during the etiological investigations of the patient may be due to a coincidental disease . nerve conduction studies were normal while there were acute denervation potentials , which lead to the idea of a pathology located more proximal to spinal ganglia . moreover , disappearance of spontaneous denervation potentials after treatment of serotonergic syndrome would be an evidence for toxic effects of serotonin on spinal cord cells . as a result , usage of inh inhibited the metabolism of serotonin and induced serotonergic toxicity in our case . interaction of inh and essitalopram was rarely reported before ; however , our case is the first to report probable electrophysiological findings due to serotonergic toxicity .

here , we report a case of serotonergic toxicity due to combination of essitalopram and isoniazid , which was rarely reported before . moreover , we observed transient neurogenic denervation potentials in needle electromyography , which disappeared with the treatment of serotonergic toxicity . as to our best knowledge , this is the first case , reporting transient electromyographic changes probably due to serotonergic toxicity .

emc was first described by enzinger and shiraki in 1972.1 emc accounts for approximately 2.5% of all soft tissue sarcomas.2 to our knowledge , bras et al . in 1985 , we represent a case of emc arising from the orbit in a 34-year - old male . we report the presentation , diagnosis , treatment , histopathology , immunocytochemistry , and molecular genetic studies of this case . a 34-year - old male without any known medical illnesses was referred to our clinic with 3 years history of the left orbital mass . ophthalmic examination indicated a palpable firm inferotemporal orbital mass of the left globe with no skin changes . the patient 's visual acuity , pupil , anterior segment , posterior segment , and optic nerve examinations were all within normal limits . a brain and orbit computed tomography ( ct ) scan , both with and without contrast , demonstrated a well - circumscribed left inferotemporal orbital mass measuring 14 mm 9.6 mm 15.3 mm , with mild globe displacement but no involvement , bony invasion or remodeling and no attachment to the extraocular muscles and no optic nerve compression [ figure 1 ] . coronal ( a ) and sagittal ( b ) computed tomography scan with contrast showing a left inferotemporal heterogeneous orbital mass measuring 14 mm 9.6 mm 15.3 mm . the bony orbit appeared to be intact a complete resection of the mass was undertaken for both diagnostic and therapeutic purposes . microscopically , the tumor comprised of spindle - shaped cells embedded within the myxoid matrix . the tumor cells were arranged in a reticular growth pattern , nests , and perivascular pseudo - rosette . there was no mitotic activity , there were small cells showing eosinophilic to vacuolated cytoplasm , oval to round nuclei with inconspicuous nucleoli , revealing perivascular arrangement in some regions , and nests and cords in a myxoid background [ figure 2 ] . molecular genetic study utilizing fluorescence in situ hybridization was positive for ewing sarcoma breakpoint region 1 ( ewsr1 ) chromosome . ( a and b ) histopathology study , ( a ) small cells showing eosinophilic to vacuolated cytoplasm , oval to round nuclei with inconspicuous nucleoli , revealing perivascular arrangement in some areas ( h and e , 200 ) . ( b ) nests and cords in myxoid background ( h and e , 200 ) . ( c ) immunocytochemistry study showing vimentin reveals strong and intense diffuse expression ( avidin - biotin complex technique , 400 ) on the basis of immunohistopathology and molecular genetic studies , the patient was diagnosed with emc of the orbit . the patient was sent for additional systemic investigations to the oncology center , which reported no evidence of metastasis . throughout the 6 months follow - up after resection , there were no additional signs of local recurrence both clinically and with radiology studies . first described by enzinger and shiraki in 1972,1 emc accounts for approximately 2.5% of all soft tissue sarcomas.2 emc most commonly affects the lower extremities.3 differentiation of emc from other sarcomas is possible due to cytological and molecular genetic studies45 in conjunction with clinicopathological features . several cases of emc have been reported in different anatomical locations of the body,6 with only a single previously reported case of emc within the orbit.7 chondrosarcoma is a misnomer used to describe the emc at the histopathological level , and only minority of cases show evidence of well - formed hyaline cartilage . in addition , only a few cases of emc have been found to be s100 positive , whereas skeletal chondrosarcomas nearly always stain positive.8 gross examination of emc usually indicates neoplasms as soft tissue multinodular with smooth surface that is fairly circumscribed . microscopically , the tumors present mostly as multinodular comprising of uniform round to oval or spindle - shaped cells embedded within the myxoid matrix and round to oval hyperchromatic nuclei and eosinophilic cytoplasm . the tumor cells are arranged in a reticular growth pattern , nests , cords , or strands . immunohistochemistry9 indicates the tumor cells are usually intense diffuse positive staining for both vimentin and synaptophysin with a variable percentage of staining to s-100 protein , desmin , and epithelial membrane antigen ( ema ) . poor prognosis and an aggressive tumor is based on size larger than 10 cm with high cellularity ; anaplasia and rhabdoid phenotype , with focal regions of ki-67 staining > 25%.10 localized emc is usually treated with a complete excision of the tumor with a wide surgical margin . currently , there is no evidence of the effectiveness of adjunctive therapy such as radiation or chemotherapy if there are no signs of metastasis . the only emc case previously reported in the orbit was treated with exenteration due to the aggressive tumor presentation cause very severe proptosis of the eye.7 in our case , the microscopic features were identical to previous reports with no mitotic activity or necrosis . cyclin d1 decorated more than 75% of the nuclei , and p53 and ki-67 were present in 15% and < 1% of nuclei , respectively . ema ; s100 ; glial fibrillary acidic protein ; pan - cytokeratin ; actin ms all failed to express any immune - positivity . the patient was treated with complete resection of the mass with a wide surgical margin without adjunctive therapy . to our knowledge

extraskeletal myxoid chondrosarcoma ( emc ) is a rare soft tissue tumor . numerous cases of emc have been reported in different anatomical locations . there is currently only a single case of emc of the orbit and that was reported in 1985 . we report a second case of orbital emc in a 34-year - old healthy male .

a 45-year - old female patient was referred to the vitreoretina unit of our tertiary eye care hospital with a history of diminished vision in the left eye of one week duration following blunt trauma to the left eye . on examination , the vision in the left eye was perception of light with good projection in all quadrants . slit lamp biomicroscopy showed a clear cornea with few descemet folds , a deep anterior chamber , iridodonesis , and aphakia . the vitreous face was broken , and tobacco dusting was present in the anterior vitreous . indirect ophthalmoscopy showed presence of a dislocated crystalline lens in the posterior vitreous with intact capsule and early cataractous changes . there was a total retinal detachment with a large peripheral retinal tear just short of 90 degrees extending from 12 to 2:30 o clock . the patient underwent a routine 3 port 20 g pars plana vitrectomy with peribulbar anesthesia after being explained the guarded prognosis . the procedure was done using the accurus vitrectomy system and visualization through a biom coupled to visu microscope . lensectomy was performed using a phacofragmatome in 3d mode , and all visible nuclear and cortical material was emulsified and/or aspirated in the mid vitreous cavity . iridotomy was performed at 6o clock . at the end of fge intra - operatively , the surgeon noticed a fluffy piece of hydrated lens cortical material lying sub - retinal , approximately 1.5 mm in diameter , just within the inferotemporal arcade . removal of the sub - retinal cortex would have involved turning over the flap and searching for the cortex . the risk to benefit of successfully locating and removing the cortex , without causing surgical trauma , was weighed . the surgeon decided to go ahead with the remaining steps of surgery as planned without attempting to remove the cortex [ fig . 1 ] . immediate post - op day 3 fundus photo showing sub - retinal lens cortex just within inferotemporal arcade the patient had an uneventful post - operative course with no undue inflammation or raised intraocular pressure . there was complete absorption of the lens cortex over a period of 3 months [ fig . 2 ] . reflexes from oil over foveal area subsequently , silicone oil removal was performed , and the patient had a best corrected visual acuity of 20/40 with an attached retina [ fig . rpe changes in the form of stippling were visible over the underlying rpe [ fig . 20 months after surgery , the patient had a flat retina with best corrected visual acuity of 20/40 and presence of rpe changes at the site of the cortex . fundus photo silicone oil removal showing flat retina and presence of rpe stippling in affect area ffa early phase confirms presence of changes in underling rpe in affected area it is not common to encounter sub - retinal lens cortical material under the retina . the presence of a large retinal tear coupled with a posterior dislocated lens requiring fragmentation resulted in this unique condition . during phacofragmentation , it is not uncommon to have cortical and nuclear pieces of the lens to be dispersed in the vitreous cavity . in this clinical setting with a detached retina and almost giant retinal tear , use of perfluorocarbon heavy liquid after vitrectomy and prior to starting phacofragmentation could have minimized the chances of the same , as prevention is the only viable option . the mechanism of spontaneous absorption is similar to the removal of sub - retinal blood . this is usually associated with permanent changes in the underlying rpe , as was confirmed in this case on ffa . it is important to note that the decision to leave behind the cortex near or under the fovea would have resulted in poor visual recovery due to underlying rpe changes and attendant overlying photo receptor damage . while this presentation was unique and visual recovery good , the possibility of this complication should be borne in mind in such clinical situations and suitable intra - operative preventive measures taken .

we report a rare case of retained sub - retinal cortical material , which underwent spontaneous resorption . patient presented with a left eye traumatic retinal detachment with a large retinal tear and posteriorly dislocated cataractous lens . vitrectomy , lensectomy , silicone oil injection , and endolaser were performed . a good visual result was achieved . the report draws attention to this condition and highlights possible technique for minimizing risk of this complication in similar cases .

answer : intracranial calcification associated with isolated hypoparathyroidism . a 75yearold man with no history of systemic disease presented with an inability to walk . computed tomography ( ct ) of the brain revealed bilateral calcification of the lenticular nuclei and cerebellum ( fig . biochemical test results indicated low serum total calcium ( 5.6 mg / dl ) , elevated serum phosphorus ( 5.9 mg / dl ) , inappropriately low levels of intact parathyroid hormone ( 13 pg / ml ) , and normal serum creatinine ( 0.94 mg / dl ) . although chronic hypocalcemia is often asymptomatic , longlasting chronic hypocalcemia may have accounted for the patient 's difficulty in walking . untreated cases may affect function in multiple systems 1 , rendering early diagnosis utilizing ct imaging essential . computed tomography ( ct ) of the brain demonstrates dense calcification of the bilateral basal ganglia and dentate nuclei . written informed consent was obtained from the patient for the publication of this case and the accompanying images . a copy of the written informed consent is available for review by the editorinchief of clinical case reports .

key clinical messagehypocalcemia due to chronic hypoparathyroidism presents with nonspecific symptoms . however , if untreated , hypocalcemia may affect neurological , cognitive , muscular , and cardiac function . computed tomography ( ct ) findings may confirm a diagnosis of chronic hypoparathyroidism . although autoimmune acquired hypoparathyroidism is a rare disease , early diagnosis and treatment are critical for avoiding severe complications .

to report a case of recurrent ocular inflammation after optimal therapy of bilateral syphilitic panuveitis responding to oral celecoxib . ocular examination disclosed bilateral panuveitis . serological testing confirmed blood and cerebrospinal fluid syphilitic involvement . ocular inflammation after healing of infectious uveitis is a rare ophthalmic sequela . in an immunocompetent patient non - steroidal therapeutic options , as celecoxib , could be a good option of treatment in such immune cases . syphilis is experiencing an extreme increase of its incidence in europe and the usa since the early 2000s . although penicillin is considered an excellent effective therapy against treponema pallidum , patients hiv positive with cerebrospinal fluid involvement can fail treatment . in immunocompetent patients , either immune uveitis or syphilitic re - infection should be evoked if ocular inflammation is observed after optimal therapy . we report a syphilitic panuveitis presenting recurrent ocular inflammation after correct treatment with penicillin , treated with celecoxib , a nonsteroidal anti - inflammatory drug selective for cyclooxygenase-2 . a 76-year - old immunocompetent man presented with discomfort and blurry vision in both eyes for the last week . slit - lamp examination revealed 1 + cells and rare flare with fine keratic precipitates and 4 + cells with hypopyon in the right and left eye , respectively ( fig . dilated fundus exam showed dense vitritis and hyperemic optic nerve with blurred margins in both eyes . extensive workup undertaken revealed a serum t. pallidum rpr titer of 1:256 and positive tpha . the patient completed a 2-week cycle of intravenous penicillin g and topical steroids , experiencing a complete resolution of panuveitis and bcva of 20/20 in both eyes . after 3 weeks of the end of treatment , the patient presented up to three anterior sclerouveitis episodes , responding to topical steroids . a re - infection of treponema was microbiologically and clinically ruled out , so an immune ocular response was evoked , starting with celecoxib 200 mg a day , with progressive improvement and no recurrences observed for 1 year of follow - up . b slit - lamp photograph showing a cellular reaction and keratic precipitates in the right eye . e and f bilateral episodic anterior sclerouveitis after penicillin treatment a slit - lamp photograph demonstrating anterior uveitis with hypopyon in the left eye . b slit - lamp photograph showing a cellular reaction and keratic precipitates in the right eye . interaction of t. pallidum with the host 's immune system stimulates humoral and cellular immunity . t. pallidum produces lipoproteins on the outer membrane that interact with lipopolysaccharide - binding proteins , inducing the expression of inflammatory mediators via cd14 and toll - like receptor 2 recognition . these glycolipids induce cellular activation by releasing pro - inflammatory cytokines , mainly tumoral necrosis factor alpha , even after successful penicillin treatment . recurrent ocular inflammation after infectious uveitis treatment is a rare ocular sequela . in the immunocompetent host , such as our patient , it can occur due to a syphilitic re - infection or to an immune process , which may be caused by antigenically inert treponema cell surface . to our knowledge , this is the first report of an immune response after optimal treatment of ocular syphilis at an immunocompetent host . systemic nsaids such celecoxib are known to control ocular inflammatory processes such chronic iridocyclitis or recurrent acute anterior uveitis [ 4 , 5 ] , being suitable and sparing - steroids option of therapy in these cases . in conclusion , the outer treponema surface may play a role in immune responses observed after syphilis treatment . we present the first case reported in literature of immune sclerouveitis after neurosyphilis treatment treated successfully with celecoxib .

purposeto report a case of recurrent ocular inflammation after optimal therapy of bilateral syphilitic panuveitis responding to oral celecoxib.methodsa case report was conducted.resultsa 76-year - old man presented with painful blurry vision in both eyes . ocular examination disclosed bilateral panuveitis . serological testing confirmed blood and cerebrospinal fluid syphilitic involvement . after 2 weeks of intravenous penicillin therapy , recurrent episodic sclerouveitis was observed.conclusionocular inflammation after healing of infectious uveitis is a rare ophthalmic sequela . in an immunocompetent patient , either re - infection or immune uveitis should be evoked . non - steroidal therapeutic options , as celecoxib , could be a good option of treatment in such immune cases .

marijuana is found to have many physiological and pathophysiological functions , including mood alteration , motor and co - ordination activities . there is no literature on molecular adsorbent recirculation system ( mars ) therapy for hepatotoxicity following marijuana drug abuse . we report a case of fulminant hepatic failure following marijuana drug abuse successfully managed with mars therapy . a 23-year - old male college student presented to the emergency department with a history of malaise , fatigue and vomiting of 1 week duration . he also had a history of yellowish discoloration of the sclera and breathlessness of 3 days and developed altered sensorium a day prior to presentation . his father gave a history of marijuana abuse for the past few months and denied history of smoking and alcohol . on examination , he was irritable pale and icteric , blood pressure was 130/80 mmhg . sequential laboratory parameters of patient tests for hepatitis a , hepatitis b , hepatitis c and e , human immunodeficiency virus , cytomegalovirus , parvovirus , wilson s disease , autoimmune hepatitis and acetaminophen induced hepatitis were all negative . peripheral smear for malaria parasite , anti - dengue antibodies and leptospira serology were all negative . the diagnosis of acute fulminant hepatitis secondary to marijuana abuse was considered as other causes of fulminant hepatic failure were all ruled out and blood and urine were positive for thc . his condition worsened over the next 2 days and developed respiratory distress requiring intubation and ventilator support . on the 3 day , he was initiated on extra - corporal liver - purification system ; mars . after three sessions of mars , each lasting 8 h , there was a remarkable improvement in level of sensorium and biochemical parameters . he was extubated on the 6 day and by 10 day his liver parameters came down remarkably . it was found to have many physiological and pathophysiological functions , including mood alteration , control of feeding and appetite , motor and co - ordination activities , analgesia , immune modulation and gut motility . marijuana toxicity usually produces drowsiness , disorientation , confusion , cognitive disorganization , impaired judgment , hallucinations , paranoia and rarely toxic psychosis . until 1969 the prevalence of hepatotoxicity , ranging from 17% to 66.7% is higher when consumed in combination with alcohol . in a recent study by borini et al . , among users of only marijuana , hepatomegaly was observed in 57.7% and splenomegaly in 73.1% and slightly elevated aspartate transaminase , alanine aminotransaminase and alkaline phosphatase in 42.3% , 34.6% and 53.8% respectively . we report an unusual case of fulminant hepatic failure following marijuana drug abuse as other causes of fulminant hepatic failure were all ruled out and blood and urine for active metabolite of marijuana , thc was positive . activated charcoal may prevent further absorption from the gut if ingestion is within a couple of hours , but clinical data is lacking on efficacy . the effect of cannabinoid antagonist , 41716a [ n-(piperidin-1-yl)-5-(4-chlorophenyl)-1-(2,4-dichlorophenyl)-4-methyl-1h - pyrazole-3-carboxamide hydrochloride ] in animal models of rat and rhesus monkey produced reversible , dose - dependent antagonism of the discriminative stimulus properties of delta 9-thc . however , the effect of sr141716a on blocking or reversing the cannabis intoxication in humans is lacking . the most important aspect of treatment of fulminant hepatic failure is to provide good intensive care support . monitoring of metabolic parameters , surveillance for infection , maintenance of nutrition , and prompt recognition of gastrointestinal bleeding are crucial . though artificial support systems for kidney failure have been widely available for the past several decades , it is only recently that they have become a promising treatment modality for fulminant hepatic failure . they are mainly used temporarily as a bridge to liver transplantation or until the liver regenerates and to date , have no impact on survival . mars is performed with an albumin - containing dialysate , which is recycled in a closed loop that contains a charcoal cartridge , an anion exchanger resin adsorber and a conventional hemodialyzer . it was 1 applied in the treatment of acute liver failure in 1993 by stange et al . it has also been used to treat various poisoning with or without liver failure such as acetaminophen , amanita phalloides , phenytoin , lamotrigine , theophylline and calcium channel blockers . to date there is no published literature on the use of mars therapy for hepatotoxicity following marijuana drug abuse . however , it is unclear whether thc is also removed by mars , which needs further investigation . mars is an effective and safe means of treating fulminant hepatic failure of any etiology . however , studies are required to focus on the optimal timing of initiation and intensity of mars albumin liver dialysis .

marijuana is used for psychoactive and recreational purpose . we report a case of fulminant hepatic failure following marijuana drug abuse who recovered following artificial support systems for acute liver failure . there is no published literature of management of marijuana intoxication with molecular adsorbent recirculation system ( mars ) . mars is effective and safe in patients with fulminant hepatic failure following marijuana intoxication .

hernia of morgagni was first described by giovanni battista morgagni , an italian anatomist and pathologist in 1769 . it is a rare cause of diaphragmatic hernia usually presenting in children . the defect involves failure of fusion of the septum transversus , the diaphragm and the costal arches . even though the resulting space is often congenital , conditions such as pregnancy , trauma , chronic cough , obesity and constipation , all of which may increase intraabdominal pressure predisposes the condition [ 3 , 4 ] . usually asymptomatic patients may have symptoms such as abdominal discomfort , bloating , vomiting and bowel obstruction . morgagni hernia is an uncommon cause of gastric outlet obstruction with very few case reports in the literature . an 80-year - old man presented to our emergency room with multiple episodes of non - bilious vomiting for 4 days and not - passing stool or flatus for 2 days . he also complained of intermittent pain in the upper abdomen , which was not associated with fever , chest pain , melena or diarrhea in the recent past . however , he complained of increased cough and shortness of breath for the last 4 days on the background of a diagnosed chronic obstructive pulmonary disease . his vital parameters were normal , other than an oxygen saturation of 82% for which oxygen had to be given via face mask . laboratory investigations revealed hypokalemia ( 3.1 meq / l ) with other parameters being normal . on the chest x - ray 1 ) . computed tomography ( ct ) scan of the abdomen performed demonstrated a right anterior diaphragmatic hernia with stomach as content . ( figs 2 and 3 ) the patient was taken to the operating room , and an upper midline laparotomy was performed . findings included an ~4 3 cm defect to the right of the sternum in the anterior diaphragm ( fig . 4 ) , with incarcerated pylorus / antrum of stomach as contents and a grossly distended stomach . the contents were reduced , and the defect closed primarily with 2 - 0 non - absorbable interrupted mattress sutures ( fig . 5 ) . postoperative period was uneventful , and chest x - ray revealed a corrected defect with reduced contents ( fig . the patient was started orally on the first postoperative day and discharged on the sixth postoperative day . morgagni hernia is the rarest of all congenital diaphragmatic hernias with an incidence of ~3% . it occurs through the foramina of morgagni lying between the sternal and the costal margins of the thoracic diaphragm and was first described in 1769 . although the hernias are congenital , they rarely present in children and usually are incidentally found on routine radiological imaging in adults . symptoms if present are often nonspecific , including abdominal discomfort , bloating , vomiting and bowel obstruction . emergency presentation in such patients are commonly acute respiratory distress , gastric outlet obstruction or strangulation / ischemia of entrapped bowel [ 5 , 6 ] , with an incidence of 1214% . diagnosis is usually suggested by chest x - rays indicating an air fluid level in the chest with barium studies revealing contrast in the stomach / bowel herniating through the diaphragm . ct scan of the chest and diaphragm are highly accurate and help proceed with surgical management . surgical management via laparotomy is the most common surgical approach with advantages of easier reduction of hernial contents , inspection of the contralateral diaphragm for other hernia defects and easier evaluation and surgical treatment of the acute complications , such as strangulation and ischemia of luminal abdominal organs . the laparoscopic approach has the benefits of less postoperative pain and earlier return to activities and work but is less suited to emergency presentations where surgical resection or treatment of necrotic or ischemic contents may be required [ 3 , 9 ] . morgagni hernia is an exceedingly rare hernia in adults and may present with gastric outlet obstruction in the emergency room . this clinical entity should be kept in mind while evaluating the patient , and early surgical intervention should be initiated .

morgagni hernia is the rarest of all congenital diaphragmatic hernias , first described in 1769 . it is rarely symptomatic and found on routine radiological examinations for other conditions . gastric outlet obstruction in adults with morgagni hernia is exceedingly rare . an 80-year - old man was taken to the operating room with a diagnosis of morgagni hernia with gastric outlet obstruction . an upper midline laparotomy was performed , and the incarcerated pylorus and antrum of the stomach reduced with primary closure of the defect . postoperative period was uneventful , and the patient was discharged on the sixth postoperative day . morgagni hernia is exceedingly rare in adults and may present with gastric outlet obstruction in the emergency room . this clinical entity should be kept in mind while evaluating the patient , and early surgical intervention should be initiated .

labial adhesions are common in young girls and occasionally occur in elderly women . it is hypothesized that the relative hypoestrogenic states of these age groups predispose them to labial adhesions . although surgical dissection is sometimes required , topical oestrogen creams and gentle massage usually lead to successful breakdown of adhesions in these groups within a few weeks . labial adhesions have also been described in reproductive age women secondary to female circumcision , lichen sclerosis , herpes simplex , diabetes , pemphigoid , and caustic vaginitis [ 36 ] . to our knowledge , a 25-year - old woman , nulligravida , was divorced 5 years ago and was nondiabetic patient . because of some upper egypt traditions she refused to go to doctors ( as approximately all surgeons in our localities are male ) . she had a severe itching with severe monilia infection , and she consulted only dermatologic doctors , the conditions worsened till complete adhesion occured leading to urine retention with difficulty in micturition , then urine retention with overflow . on taking the history , she was circumcised as a routine procedure done for young female in villages and rural area in upper egypt . on examination she had a complete vulval adhesion with only pinhole opening for micturation with complete adhesion of the labia and overflow of the urine with some leucodermic area from chronic irritations ( figures 1(a ) and 1(b ) ) . the area was infiltrated with injection of adrenalin in concentration of 1 : 10000 . separation of the adhesion and fusions was done , and bilateral vulvar flaps to reconstruct the defect of raw surface resulted . randomized mucocutaneous flaps were dissected posteriorly to be advanced to reconstruct the raw surface after separation of adhesion . urethral catheter was applied and lastly suturing of flaps using polyglactin ( 4/0 ) ( ethicon ) . she was instructed for standard perineal care , which included spreading the labia periodically and washing with water and antiseptic . the patient was discharged 48 hours after surgery with no complications and instructed to continue routine perineal hygiene at home . examination revealed that her labia were completely healed ( see figures 1(a ) , 1(b ) and 2(a ) , 2(b ) ) . labial fusion is most commonly noted between 3 months and 4 years of age and has a peak incidence of 3.3% between 13 months and 23 months of age . labial fusion is considered to be an acquired condition secondary to hypoestrogenism and vulvovaginitis [ 8 , 9 ] . several previous authors hypothesized that the relative hypoestrogenic state of the immediate postpartum period contributes to the formation of labial adhesions [ 1 , 10 ] . this middle age lady has no history of recent delivery and did not have any pattern of hypoestrogenic state . severe and recurrent infection with bad hygiene can also predispose to labial adhesion , especially monilia infection . the local cultural problems led to the late presentation of the case till complete urine retention occured . the fusion of distal based randomized mucocutaneous flap to reconstruct the defect has not been described in the literature before and the healing of the flap was excellent .

case . a 25-year - old woman presented with acute urine retention with overflow 6 months after an inadequate treatment of severe monilia infections . examination revealed complete adhesion between both labia majora . division of adhesion was done with reconstruction by labial mucocutaneous flap . complete recovery was achieved with good cosmetic outcome . conclusion . labial adhesions whatever their severity is can be surgically divided with complete correction by locally designed flap to reconstruct the introuitus with rapid recovery , good healing , and good cosmetic outcome .

rhabdomyoma is the most common cardiac tumor in fetal life , though a rare condition . cardiac rhabdomyomas ( cr ) are closely associated with tuberous sclerosis complex ( tsc ) . tuberous sclerosus ( ts ) is an autosomal dominant condition involving multiple organs such as heart , brain , eye , kidney and skin . crs may precede the skin , neurological and radiological signs of tsc by months or even years . however , association of craniofacial malformation such as pierre robin sequence ( prs ) , clubfoot and intestinal obstruction with ts has not been described earlier . this is probably the first case where ts and prs have been described in the same patient and that too in a neonate . the present case report is about a 3-day - old late preterm ( 36 weeks of gestational age at birth ) male neonate , who was referred to us from an outside institution with complaints of bilious vomiting and antenatally detected multiple cardiac tumors . the baby was born out of a non - consanguineous marriage to a 29-year - old primigravida . two homogenous echogenic tumors of size 7 mm and 4.5 mm were noted in the ventricular septum . other locations were medial papillary muscle of mitral valve and right ventricular free wall . the tumors were non - obstructive . however , feeds could not be started as he developed abdominal distension and bilious vomiting on day 1 . physical examination was remarkable for the presence of prs ( micrognathia , glossoptosis and cleft palate ) . clinically , bradycardia and irregularly irregular heart rate was present ( heart rate varying between 80 and 110/min ) . x - ray erect abdomen showed multiple air - fluid levels suggestive of lower small bowel obstruction and a barium enema study revealed microcolon probably indicating an atretic lesion in the distal small bowel . nature of convulsions became mixed on day 6 with predominantly myoclonic type and was controlled with phenobarbitone and levetiracetam . computed tomography scan - brain showed subependymal nodules and cortical tubers [ figure 2b ] . a diagnosis of ts was made in view of cr and cortical tubers in brain . the parents did not give consent for explorative laparotomy and took discharge against medical advice on day 10 of life . lack of facilities and logistic reasons precluded the genetic work - up of our patient . bilateral club foot and bilious gastric aspirates apical four chamber view of two - dimensional echocardiogram showing multiple rhabdomyomas of heart in the mid and apical interventricular septum ( a ) . multiple subependymal nodules and cortical tubers ( shown by black arrows ) in computed tomography scan of brain ( b ) fetal cr is a rare condition , but the most common cardiac tumor in fetal life accounting for 60 - 86% of fetal cardiac tumors . though crs have a natural history of spontaneous regression , prognosis is guarded as it is very frequently associated with ts which is inherited as an autosomal dominant trait with variable penetrance and prevalence of 1/6,000 people . clinical manifestations include seizures , skin lesions and hamartomas affecting multiple organs such as heart , brain , eye and kidneys . the first signs of ts can be picked up on routine prenatal ultrasound screening where lesions are found in heart ( rhabdomyomas ) and brain ( cortical tubers , subependymal nodules and subependymal giant cell astrocytoma ) . with advancement in fetal echo and magnetic resonance imaging , prenatal diagnosis a recent meta - analysis showed that the incidence of ts was 64% in fetuses with crs . furthermore , a positive family history of ts and multiple cardiac tumors were more likely to be associated with ts . a 42 year retrospective review of 70 patients consisting of 43 fetuses and 27 neonates with ts showed that crs comprised 33% of pathological findings , those of central nervous system origin 47% and renal cystic disease 13% . skeletal anomalies like clubfoot and cleft palate are not usually associated with tsc and have been reported in isolated cases . our case is unusual in having bilateral congenital talipes equino varus along with prs and small bowel obstruction . prs has been associated with nearly 40 varieties of syndromes and chromosomal anomalies , but the underlying pathogenesis of this disease has still remained enigmatic . recently a genetic association has been identified between a neurocutaneous syndrome ( neurofibromatosis type 2 ) and prs . the present case is also a neurocutaneous syndrome with prs as an additional finding , but genetic work - up could not be done . when ts is diagnosed in the perinatal period , it is associated with high incidence of morbidity and mortality . however , it is observed that survival rates of patients diagnosed antenatally was practically the same as for those after birth it has been reported that more than 80% of crs have complete resolution within infancy and early childhood . however unlike crs , cerebral lesions do not regress , but instead progressively increase in size and number . our knowledge of natural history of crs in utero is limited and meta analytical approach to the prenatal management of ts is lacking . the age - dependent nature of the characteristic features of tsc has presented challenges for the diagnosis in the first year of life . hypomelanotic macules may be present in infancy , but facial angiofibromas and shagreen patches usually do not occur until puberty and renal angiomyolipomas occur in adults . crs may be the earliest finding of tsc in utero and may precede the detection of the brain or kidney lesions and can be symptomatic in the fetus and newborn .

we report a case of a neonate who presented to us with multiple rhabdomyomas of heart , cortical tubers in the brain and skeletal anomalies such as pierre robin sequence , bilateral clubfoot and lower small bowel obstruction . though a diagnosis of neonatal tuberous sclerosis was made , the association of skeletal anomalies and intestinal obstruction was a rare and unusual finding .

meningococcal disease is an acute bacterial disease caused by neisseria meningitides which is a gram negative capsulated coffee - bean - shaped bacteria seen in pairs . thirteen serogroups of n. meningitidis have been identified based on capsular polysaccharide antigen and of these ; four sera groups namely a , b , c , and w135 are known to cause epidemics . multiple outbreaks have occurred in 1966 , 198586 , 2005 , 2007 , and 2009 . an outbreak of meningococcal disease was reported in three districts of andhra pradesh namely , vishakhapatnam , vijayanagram , and srikakulam in 20052006 . the etiological agent was again found to be of the sera group a. there are no other cases reported from other parts of south india . we are reporting two cases of meningococcal meningitis from an urban health centre from vellore , tamil nadu . two male children , 12 years and 7 years , presented to the casualty of the urban health centre within 2-week interval . they came from the same community and lived half a kilometre of each other . the first child presented with history of fever , headache , neck pain , and vomiting for 4 days . the second child had these symptoms for one day and approached the urban health centre within 1 day . the past history , immunization history , and developmental history were normal for both children . on examination both children were drowsy , febrile with a glasgow coma scale ( gcs ) of 13/15 for first child and 15/15 for the second child . they were both admitted and underwent lumbar puncture for cerebrospinal fluid ( csf ) analysis . at lumbar puncture both the csf was turbid . the investigation results [ table 1 ] were as follows : results of cerebrospinal fluid analysis for both children both children had predominant leucocytosis and increased neutrophil predominance in the blood investigation . the children were referred to the tertiary centre for further treatment and management due to deterioration in the sensorium . the polymerase chain reaction ( pcr ) test revealed the causative strain to be neisseria meningitis serotype c. both children were administered injection cefotaxime by parenteral route for two weeks and were well at discharge . once the diagnosis was established , we initiated preventive measures to the health - care providers , immediate family , and the community . health education about the signs and symptoms of this disease was given to the community and the health volunteers after the admission of the first child . the second child was brought to the health centre within a day of the symptoms because of the intervention of local health volunteers . after the diagnosis of the second child , further health education was planned and done to the surrounding urban community . as schools were closed for the summer vacation , chemoprophylaxis was not given to the school children . volunteers and health workers did intensive monitoring for 3 weeks consecutively after the diagnosis of the first case . limited data available on the epidemiology of meningococcal infection in india reveal a low background incidence of disease . the disease is characterized by fever , headache , and vomiting , and neck stiffness . if not detected early it can run a fulminating course leading to septicaemia and death . the age group mostly affected are children and young adults as observed in our case report . human beings are reservoirs for this bacterium and 1015% of healthy subjects harbour it in the upper respiratory tract . the transmission of disease is by droplet infection from nose and throat of patients and carriers . the incubation period is 210 days and outbreaks are more common in spring and winter months . the second child was brought to the centre early due to increased awareness initiated by the volunteers and health care workers after diagnosis of the first child . the management of suspected meningococcal disease includes admission to a health centre for lumbar puncture and csf examination . primary care physicians are also responsible for the health of the community , the management of cases also involves prevention of secondary cases . it should be initiated within 48 h of diagnosis of cases to all identified persons who had close contact with the case within 7 days of onset of the disease . drugs with dosage recommended for chemoprophylaxis in meningococcal disease two different types of meningococcal conjugate vaccine ( mcv ) are available in india . one is a quadrivalent ( a , c , w135 , y ) mcv and the other is a monovalent serogroup a vaccine . the indian academy of paediatrics ( iap ) state that routine use of mcvs in india is not justified because of low incidence of disease . mass vaccination is recommended during outbreaks ( defined as more than 10 cases per 100,000 populations ) .

meningococcal meningitis has rarely been reported in tamil nadu . we report here two children diagnosed with meningococcal meningitis in vellore , tamil nadu , on may 2014 . the causative strain was neisseria meningitidis serotype c. the role of the primary care physician in early diagnosis , appropriate referral , and preventive measures of this disease to the immediate family and community is stressed .

continuous venovenous haemofiltration ( cvvh ) is an established treatment for patients with acute kidney injury . during cvvh , the rate at which this happens depends on the difference in their concentrations between serum and replacement fluid , and on the rate of treatment . patients presenting with acute kidney injury may have concomitant severe hyponatraemia or hypernatraemia . over - rapid correction of the serum naconcentration if cvvh is needed , the naconcentration in the replacement fluid ( usually 140 mmol / l ) needs to be adjusted in order to avoid rapid changes of the serum naconcentration . in the present paper if cvvh is necessary , the naconcentration of the replacement fluid should be increased by adding concentrated nacl solution ( table 1 ) . effect of adding different volumes of 30% nacl to replacement fluid effect of adding different volumes of 30% nacl ( 5 mmol / ml ) to a 5 l bag of replacement fluid containing a naconcentration of 140 mmol / l . generally , it is not considered safe to lower the serum naconcentration by more than 8 to 10 mmol / l over 24 hours , especially in the setting of chronic hypernatraemia . usually , a stepwise correction of the patient 's serum naconcentration is planned using replacement fluid made up to successively lower naconcentrations . if the serum nadecreases by > 2 mmol / l in 6 hours , either the rate of filtration should be decreased or the fluid bags should be changed to bags with a higher naconcentration . the volumes of 30% nacl added are small and will not affect the concentration of other electrolytes in the solution significantly . if cvvh is needed , the naconcentration of the replacement fluid should be reduced by adding sterile water ( table 2 ) . generally , it is not considered safe to increase the serum naconcentration by more than 8 to 10 usually , a stepwise correction of the patient 's serum naconcentration is planned using replacement fluid made up to successively higher naconcentrations . effect of adding different volumes of water to replacement fluid effect of adding different volumes of water to a 5 l bag of replacement fluid with a naconcentration of 140 mmol / l . [ na ] , sodium concentration ; [ hco3 ] , bicarbonate concentration ; [ k ] , potassium concentration . if the serum naconcentration has increased by > 2 mmol / l in 6 hours , either the rate of filtration should be decreased or the fluid bags should be changed to bags with a lower naconcentration . the concentration of bicarbonate and potassium in the final solution will also be reduced , and the patient may need additional supplementation . the authors would like to thank the icu pharmacists at guy 's & st thomas ' hospital for their contribution .

in patients with acute kidney injury and concomitant severe hyponatraemia or hypernatraemia , rapid correction of the serum na+ concentration needs to be avoided . the present paper outlines the principles of how to adjust the na+ concentration in the replacement fluid during continuous renal replacement therapy to prevent rapid changes of the serum na+ concentration .

denovo deletions may affect the terminal part of a chromosome or an interstitial region , and usually affect a fairly large number of genes and produce recognizable syndromes . microscopically visible deletions generally involve multiple genes , and the consequences of losing this much genetic material from even one of the chromosome pair can be severe . if the missing part contains important instructions , it can lead to congenital malformations , development delay , or mental retardation . a one and a half year old girl child born of non - consanguineous marriage to a 27-year - old primi mother was referred to the genetic clinic . the child was born at term by elective cesarean section because of symmetric iugr and oligohydramnios . birth weight was 2.0 kg and length was 48 cm with head circumference 30 cm . there was no significant family history , no evidence of teratogenecity , and no other pregnancy associated complications . clinical examination of the new born revealed grade 3 pansystolic murmurs in the left lower sternal border . echo was suggestive of congenital acyanotic heart disease vsd , asd , pda with moderately severe pulmonary hypertension . the first genetic consultation was at the age of 1 year 6 months for dysmorphic features and congenital heart disease . facial dysmorphism included microcephaly , brachycephaly , hypertelorism , upslanting palpebral fissures , strabismus , broad nasal bridge , wide anterior fontanalle , anteverted nares , high arched palate , microretrognathia , low set dysmorphic ears , and short neck . pallor was seen along with cyanosis and nail bed clubbing of the finger and toes . mri ( brain ) showed partial agenesis of corpus callosum , bilateral mildly enlarged subarchanoid space , and mildly dilated third and lateral ventricles . cytogenetic analysis from the proband was carried out by peripheral blood lymphocyte culture and gtg banding . karyotype of the proband showed deletion in the terminal region of the long arm of chromosome 6 in all the metaphase analyzed . the karyotype of the child was 46,xx , del(6)(q24qter ) [ figure 1a and b ] . ( a ) karyotype 46,xx , del(6)(q24 to qter ) showing deletion , ( b ) partial karyotype showing 6q 24 to terminal deletion cytogenetic analysis from the proband was carried out by peripheral blood lymphocyte culture and gtg banding . karyotype of the proband showed deletion in the terminal region of the long arm of chromosome 6 in all the metaphase analyzed . the karyotype of the child was 46,xx , del(6)(q24qter ) [ figure 1a and b ] . ( a ) karyotype 46,xx , del(6)(q24 to qter ) showing deletion , ( b ) partial karyotype showing 6q 24 to terminal deletion cytogenetic analysis from the proband was carried out by peripheral blood lymphocyte culture and gtg banding . karyotype of the proband showed deletion in the terminal region of the long arm of chromosome 6 in all the metaphase analyzed . the karyotype of the child was 46,xx , del(6)(q24qter ) [ figure 1a and b ] . ( a ) karyotype 46,xx , del(6)(q24 to qter ) showing deletion , ( b ) partial karyotype showing 6q 24 to terminal deletion here , we describe a case with deletion in the terminal region of long arm of chromosome 6 with break point at q 24 region . chromosome 6q deletion is associated with dysmorphic features and multiple anomalies , which were seen in the index case also . the more frequently observed clinical features in 6q deletion are congenital heart defects , which were also seen in our case . other features like brachydactyly , clinodactyly along with peripheral clubbing and cyanosis were also seen [ figure 2a , 2b and figure 3 ] . literature search with chromosome 6q24 deletion showed few reports with varying other anomalies like nuchal cyst , diaphragmatic hernia , multicystic kidney , etc , which was not seen in our case . caselli et al . , have reported a similar case with deletion of 6q24.3 - 25.1 in an 8-year - old with growth failure , cardiac septal defects , mental retardation , and dysmorphic features . ( a and b ) front and side profile showing dysmorphic facies child with chromosome 6q deletion the region involved in our case is q24 to terminal and therefore explains the neurodevelopmental delay and peculiar facial features . more than twenty two genes are present in this deleted region , which might be important in the normal development of cardiovascular and central nervous system . the karyotype of parents were normal , which further suggests that this must be a denovo event and not due to unbalanced product of familial translocation . as the parental karyotype was normal and the child had a denovo deletion , the chance of recurrence is very low . nevertheless , prenatal diagnosis by amniocentesis and fetal karyotyping has been offered to the couple , in case of future pregnancies . hence , chromosome analysis should be offered to every child with mental retardation and dysmorphism as in this case . fish studies and array - cgh analysis may be useful to delineate the size of the deletion and the genes involved , which in our case was not affordable by the patient .

in this report , we describe a one and a half year old girl showing terminal deletion of long arm of chromosome 6q . the associated abnormalities such as congenital heart disease , mental retardation , and dysmorphic features are described . cytogenetic studies with gtg banding showed 46,xx , del(6)(q24qter ) . karyotype of the parents was normal suggesting a denovo event .

a 25-year - old primiparous lady presented to the antenatal clinic for a routine checkup at 36 weeks gestation . during her visit , she demonstrated a large pedunculated tumor on the dorsal surface of the interphalangeal joint of her right thumb ( figure 1 ) . this initially started as a small pimple which she first noticed at approximately 24 weeks gestation . she decided to burst and drain the pimple then , and again 4 weeks later , which resulted in a small bleed from the lesion on both occasions . however , the pimple continued to grow in size , and at 32 weeks gestation she presented to her general practitioner who commenced her on oral co - amoxiclav . this had no effect on the lesion , and it continued to grow , reaching a size of 2.52.51 cm on presentation to her antenatal visit at 36 weeks gestation . it had at that stage become oozy , foul - smelling and was causing her significant local pain . at that point , we referred her to the plastic surgery team at our affiliated general hospital . the lesion was a polypoid lobulated mass measuring 242310 mm , with slicing showing a fatty tan surface admixed with blood . histopathological examination of the lesion showed evidence of a hemangioma with overlying infarction , ulceration and inflammation with no atypical features ( figure 2 ) . she was admitted to the rotunda hospital a week after with pregnancy - induced hypertension , which was controlled with labetalol 200 mg twice daily . they are common in infants , and commonly present on the face and scalp , usually spontaneously regressing . in the adult research has suggested a possible role for estrogen as a mediator for vascular proliferation and hemangioma formation . this could potentially explain the rapid growth of this lady s hemangioma during her pregnancy . to the best of our knowledge

this is a case of a rapidly enlarging cutaneous pedunculated tumor on a patient s thumb during her pregnancy . this was excised and identified as a hemangioma . a literature search identified a possible hormonal factor in causing an accelerated growth of this tumor .

choroid plexus papillomas are considered as who grade i tumors and are among the most common tumors in children less than 2 years . however the present case illustrates a case where the tumor was detected in - utero and had to be operated upon relatively early because of obstructive hydrocephalus and raised intracranial pressure with excellent outcome . this male infant was the first baby born to parents with non - consanguineous marriage , residing in a remote village . the prenatal period was uneventful except in 8 month of gestational age when a routine ultrasonographic ( usg ) examination revealed large head size , exact details were not available . although the pregnancy was declared high risk , the baby was born at full term through caeserian section . at birth , although the baby cried immediately after the birth he did not take feeds . in the next few days the baby had progressive increase in head size with excessive crying along with multiple episodes of vomiting . he was brought to us at the age of 3 weeks with complaints of progressive increase in head size , episodes of excessive inconsolable cries associated with multiple vomiting as well as failure to gain weight . on examination , he was subjected immediately to non - contrast computerized tomography ( ncct ) scan of the brain [ figure 1 ] that showed a large hyperdense mass lesion in the left lateral ventricle arising from the atrium causing biventricular obstructive hydrocephalus with enlargement of lateral ventricles . on three dimensional reconstruction it was clear that the lesion was arising from the left lateral ventricle choroid plexus wit h four well - defined stalks of attachments medially to the atrium . these radiological findings pointed towards choroid plexus papilloma ( cpp ) and he was planned for surgical excision of the lesion . in view of the complaints and features suggesting raised intracranial pressure since birth it was highly likely that the pathology was developed in fetal life and only when the problem became unbearable , parents sought medical advice . pre - operative non - contrast computerized tomographic scan - axial view patient was given general anesthesia and positioned prone and left sided parieto - occipital osteoplastic craniotomy was performed using midas rex drill . brain was tense on removing the bone flap , prophylactic cerebro spinal fluid ( csf ) drainage from left frontal horn was performed to relax the brain . after this , duramater was opened and transcortical - transventricular approach was taken to the occipital horn of left lateral ventricle . large , soft - firm , white - yellow colored , cauliflower - like lesion were seen that was could be partly sucked out . under high - magnification microscope the feeders from the choroidal arteries were identified , coagulated and severed with minimal retraction of the lesion , utilizing the buoyancy provided by the csf . sequentially the other remaining stalks of attachments were identified microscopically and coagulated with bipolar electrocautery and cut . the complete lesion was excised in - toto [ figure 2 ] and the choroid plexus visible in the left lateral ventricle was coagulated . hemostasis was achieved and closed with a ventricular drain placed in left lateral ventricle that was kept for 3 days . post - operative ncct scan of the brain showed complete excision of the lesion with resolved hydrocephalus and accompanying interstitial edema [ figure 3 ] . the baby was discharged on post - operative day 10 and he was taking oral feeds with no further episodes of vomiting . post - operative non - contrast computerized tomographic scan the infant was doing well at the last follow - up at 6 months post - operation . a post - operative magnetic resonance imaging ( mri ) brain revealed no evidence of residual lesion , resolution of hydrocephalus and evidence of corticectomy . when these lesions are located in the lateral ventricle , the median age at diagnosis is 18 months , and there is no predilection to occur in either sex . they are well - circumscribed lobulated intraventricular masses with cysts , necrosis , and hemorrhage . on immunohistochemistry they are positive for cytokeratin , vimentin and s-100 protein which is expressed in 90% of cpps . in around 20% of pediatric cases , these tumors show malignant transformation ; approximately half of them are located within the atrium . they are similar in aspect to the glomus of the choroid plexus , being formed by a single cell layer of cuboid or cylindrical cells surrounded by a thin fibrovascular structure . in the latest world health organization ( who ) classification , cpps are classified as grade i. on ncct of brain these tumors frequently contain calcifications , hemorrhages in various stages , or cysts . in some cases , cpps may fill the ventricular cavity , whereas in others , the ventricle may be markedly dilated ipsilaterally . the surgical approach should aim to expose and interrupt the proximal arterial supply from the choroidal artery to devascularize the tumor at an early stage of the procedure followed by in - toto excision of the tumor . in the present case , authors illustrate an unusual presentation of cpp with raised intracranial pressure ( icp ) since birth . after the complete surgical excision post - operative period was uneventful and the baby is doing well till last follow - up , 6 months post surgery . this case illustrates the rare fetal cpp and the need for proper pre - operative planning , meticulous surgical technique , and intensive intra - operative monitoring for normothermia , fluid - electrolyte balance , and blood replacement for achieving excellent results .

choroid plexus papillomas ( cpps ) are rare tumors having bimodal distribution . pediatric cpps are commonly present in supratentorial compartment and most commonly located in lateral ventricle and usually present at 16 - 18 months . authors could find only one case report of fetal choroid plexus papilloma in the literature . in the present case , authors illustrate an unusual presentation of cpp with raised intracranial pressure ( icp ) since birth , the need for proper preoperative planning , meticulous surgical technique , and intensive intra operative monitoring for normothermia , fluid - electrolyte balance , and blood replacement for achieving excellent results .

in this issue of critical care , robinson and colleagues investigate the effect of increasing doses of the low molecular weight ( lmw ) heparin enoxaparin ( commonly used as prophylaxis against venous thromboembolism ( vte ) ) on systemic heparin concentrations , expressed as anti - factor xa levels . reported rates for deep venous thrombosis in patients admitted to the icu range from 22 to 80% depending on patient characteristics . thromboprophylaxis with unfractionated or lmw heparin lowers the risk for deep venous thrombosis by more than 50% . nevertheless , the risk of vte in critically ill patients receiving lmw heparin prophylaxis is still much higher than in other patient groups . amongst several factors that may explain the higher incidence of vte in critically ill patients , such as full immobilisation or withholding anticoagulant prophylaxis is still much higher than in other patient groups . amongst several factors that may explain the higher incidence of vte in critically ill patients , such as full immobilisation or withholding anticoagulant prophylaxis because of a high bleeding risk , it was hypothesized that limited bioavailability ( that is , lower plasma anti - factor xa activity ) of subcutaneously administered heparin in those patients with impaired peripheral circulation , due to vasopressor medication to maintain central blood pressure , might be important . indeed , in a first comparative trial it was shown that critically ill patients on high dose vasopressor medication had much lower anti - factor xa concentrations after the subcutaneous administration of lmw heparin in comparison with intensive care patients that had lower doses of vasopressor or in comparison with patients in the surgical ward . a subsequent study also found consistently lower anti - factor xa levels after subcutaneous heparin in critically ill patients . in another similar study , critically ill patients with excessive subcutaneous oedema had lower anti - factor xa concentrations compared to a control group without oedema . this observation was confirmed in a group of critically ill multiple trauma patients , who showed variable and low heparin concentrations after subcutaneous injections . robinson and colleagues compared plasma anti - factor xa levels after the subcutaneous administration of the lmw heparin enoxaparin at the conventional dose ( 40 mg ) and at increasingly higher doses ( up to 70 mg ) in intensive care patients . they found a dose - dependent increase in peak anti - factor xa levels ( 4 hours after the injection ) ranging from 0.13 iu / ml at the conventional 40 mg dose to 0.29 iu / ml at the 70 mg dose . considering that optimal efficacy and safety of lmw heparin for thromboprophylaxis in orthopaedic and abdominal surgery was achieved with dosages of heparin resulting in peak plasma anti - factor xa activities ranging between 0.25 and 0.30 iu / ml , it may be concluded that critically ill patients need much higher doses of lmw heparin than other patients . based on the findings of robinson and colleagues , the subcutaneous dose of lmw heparin should be increased to 60 mg daily . alternatively , direct intravenous administration of ( lmw ) heparin may be considered ; however , experience with this type of thromboprophylaxis is limited . the mechanism by which critically ill patients have lower anti - factor xa levels after subcutaneous administration of heparin is not completely understood . the initial hypothesis was that patients on high dose vasopressor medication had impaired subcutaneous blood flow and thereby limited ability to adsorb the subcutaneous heparin . an alternative explanation is that the presence of oedema hinders the absorption of heparin , although that hypothesis was not proven . in addition , it has been suggested that systemic inflammation and associated multiple organ dysfunction may have an impact on heparin binding to plasma proteins and drug metabolism . the clinical relevance of lower anti - factor xa levels after conventional doses of ( lmw ) heparin in critically ill patients is also not totally clear . theoretically , the low anti - factor xa levels may lead to suboptimal prophylaxis and could indeed be a contributory factor to the higher incidence of thromboembolic complications in critically ill patients despite routine thromboprophylaxis based on the observations of robinson and colleagues and others , a randomized controlled trial with conventional versus higher doses of thrombosis prophylaxis in critically ill patients aiming at the reduction of the incidence of vte and other clinically relevant outcomes is justified . such a study would also enable the evaluation of bleeding complications related to the administration of prophylactic heparin , as intensive care patients are at higher risk for hemorrhage as well . in summary , there is ample evidence that conventional thromboprophylaxis leads to lower systemic heparin levels in critically ill patients . it is not clear whether this contributes to the relatively high incidence of vte in intensive care patients . a clinical trial evaluating higher doses of heparin for prevention of vte and assessing the bleeding risk of such an approach is justified .

venous thromboembolism is a relatively frequently occurring complication in critically ill patients admitted to the icu despite prophylactic treatment with subcutaneous low molecular weight heparin . several studies show that critically ill patients have significantly lower plasma anti - factor - xa activity levels compared to control patients after administration of subcutaneous heparin . robinson and colleagues show in this issue of critical care dose - dependent but relatively low levels of anti - factor xa activity at increasing doses of enoxaparin . anti - factor xa levels thought to be required for adequate thromboprophylaxis are observed only at doses of enoxaparin that are one and a half times higher than the conventional dose ( 40 mg ) .

a 34-year - old woman presented acute subarachnoid hemorrhage ( hunt and hess grade 2 ) with severe headache . left vertebral artery angiography demonstrated a 2.5 mm sized left side sca aneurysm with a narrow neck ( fig . 1a ) . under general anesthesia , we coiled this aneurysm by using a 2 mm4 cm microplex hypersoft coil v - trak system ( microvention , california , usa ) ( fig . the most proximal part of the detachment zone protruded minimally into the basilar artery ( ba ) after detachment ( fig . we performed follow - up fluoroscopy of coil mass to check out coil stability for 2 postoperative days ( pods ) , and there was no significant change of coil mesh ( fig . 1d ) . in the ninth pod , she complained newly developed headache and dizziness . magnetic resonance imaging revealed a small size acute infarction in the left side sca territory , but there is no evidence of recanalization of the aneurysm or recurred subarachnoid hemorrhage . left side sca was intact on mr angiography ( mra ) , however , fluoroscopy revealed unraveling of proximal coil loop and herniation into the ba ( fig . 1e ) . herniated coil loop and remaining coil mesh were pulsating together with arterial pulsation . we initially considered stent placement to trap the coil loop , but decided to wait because she was stable and ba and its branches were intact on mra . oral aspirin 100 mg per day was given . during following three days , we repeated fluoroscopy and ascertained no further herniation of the coil loop . the shape of the herniated coil loop was changed and the herniated coil was spontaneously repositioned into the probable left side sca on fluoroscopy ( fig . we confirmed no change in configuration of the herniated coil loop on follow - up fluoroscopy during 3 months , and we performed mra in the 90th pod . the previous infarction was resolved and there was no evidence of recanalization or sca occlusion . coil loop herniation into the parent artery is an uncommon complication of aneurysm embolization , and the incidence was 2.4 - 4.2% in previous reports ( 1 , 2 ) . the mechanism of coil herniation appeared to be coil instability after detachment and excessive embolization ( 2 ) . contrarily , minimal protrusion of the coil 's detachment zone into the parent artery may occur not infrequently during embolization , and this situation can be managed uneventfully with or without anti - platelet treatment . in our institution , we usually check follow - up fluoroscopy during two pods in this situation . in our case , we could not indicate exactly the day when unraveling of coil developed , but probably it might occur near to ninth pod when the patient complained new headache and dizziness . delayed unraveling and herniation of coil loop was reported previously ( 3 ) . in our case , it was difficult to insert last coil loop into aneurysm sac and we remained short detachment zone within the lumen of parent artery . this configuration is usually stable , but it may cause adverse consequence like our case . so - called ' stored tension ' ( 3 ) in last coil loop might cause delayed unraveling . in our opinion , if minimal protrusion of the detach - zone into the parent artery is present , we should consider the possibility of delayed herniation of coil loop , and follow - up fluoroscopy for the evaluation of coil mesh may be necessary during minimum two postoperative weeks . managements of coil herniation after detachment are controversial and include removal by using a snare ( 4 ) , repositioning by using balloon ( 5 ) , or trapping of coil by using stent placement ( 2 , 6 - 8 ) . occasionally , coil herniation is left in the lumen of parent artery with or without anti - platelet treatment if herniated coil aligns along the parent arterial wall or only a small closed loop is herniated ( 4 ) . there was no systematic analysis about the fate and risky configuration of herniated coil loops in the parent arterial lumen . only a suggestion was presented that herniated coil with a free end pulsating in the direction of blood flow may be a risky configuration ( 4 ) . in our case first , we might be able to remove the coil mesh by using a snare , but we should consider the state of the coil mesh with abundant thrombus and possible thromboembolism . we can stabilize the herniated coil loop along the wall of ba by the stent deployment ; however , we should consider the procedure related complication and delayed in - stent stenosis ( 8) . because she was stable and relatively young , we deferred stent placement . in our opinion , the herniated coil loop may be affected by hemodynamics of the parent artery and coil 's tendency toward its natural loop shape . in our case , this restoration tendency made the last stable configuration of the herniated loop . in our opinion , follow - up fluoroscopy with anti - platelet medication may be another option and guide proper decision for management of this situation . in conclusion , minimal coil protrusion into the parent artery may undergo delayed further unraveling and herniation of a coil loop , and the shape of the herniated coil can be changed to favorable configuration . when we consider management of this condition , follow - up fluoroscopy may be helpful , and conservative management with anti - platelet medication may be one of the options of various treatments .

herniation of coil loops into the parent artery is one of the complications of endovascular embolization with detachable coils . in this clinical setting , we can not predict the consequence of the herniated coil loop . we report an unusual case of a superior cerebellar artery ( sca ) aneurysm with delayed coil herniation into the basilar artery and spontaneous reposition into the sca .

herpes simplex virus ( hsv ) mastitis in a nonlactating female is extremely rare . to the best of our knowledge , we describe the first case of herpes simplex mastitis diagnosed on scrape cytology smears prepared from the ulcer over the nipple of the left breast . possible modes of infection and the importance of distinguishing these cases from other causes of mastitis are also discussed . a 36-year - old lady presented to the outpatient department with pain and swelling in the left breast since two days . she had been given oral ciprofloxacillin and topical mupirocin by the local physician , but there was no improvement in her condition . , there was 10 cm area of indurated erythema on the medial aspect of the left breast along with an ulcer measuring five mm in diameter on the medial aspect of the nipple . air - dried smears prepared from the scrapings of the ulcer were sent to us for cytological examination . microscopic examination of scrape cytology smears revealed isolated as well as aggregates of keratinocytes showing marked nuclear pleomorphism and nuclear enlargement . these keratinocytes revealed homogenous opaque nucleus and dense basophilic cytoplasm with well - defined cell boundaries [ figure 1 ] . numerous multinucleated giant cells with ground glass nucleus and nuclear molding were also seen [ figure 1 ] . keratinocytes showing marked nuclear pleomorphism and nuclear enlargement ( giemsa , 200 ) ; inset shows multinucleated giant cells with ground - glass nucleus and nuclear molding ( giemsa , 400 ) . however , herpes simplex can affect any skin or mucous membrane surface , in addition to the eyes , central nervous system and viscera . few cases of maternal infant transmission of the virus during breastfeeding resulting in nipple lesions have been documented[46 ] and only rare cases have been reported in nonlactating women . furthermore , development of breast lesions as the first and the sole manifestation of clinically apparent hsv infection without oral or genital herpes is uncommon . there is little information regarding transmission of hsv to the breast except in cases associated with neonatal breast feeding . in the present case , further enquiry failed to reveal the source of hsv infection and we are left to speculate the mode of infection . the first possibility is that the virus was transmitted to the breast through autoinoculation from an asymptomatic oral or genital lesion . autoinoculation to other body sites such as face , fingers and eyes has been documented . the second possibility is that the present lesion may be a primary infection of the breast from recent sexual contact with an asymptomatic hsv carrier . the infected asymptomatic carrier can carry the virus in the saliva and can transmit the virus through close personal contact . kobayashi et al . have described the cytologic changes in the smears from nipple discharge of two cases with hsv infections . the cytology of nipple discharge smears of their cases revealed the ground - glass appearance of the nuclei with multinucleated syncytial cells . in addition , positive hybridization was found with intense staining for the hsv dna in the nuclei of cells having a ground - glass appearance . they concluded that cytologic observation together with an in situ hybridization procedure may be a rapid and valuable tool for the detection and final demonstration of hsv infections . in contrast to impression cytology , the scrape cytology is a traumatic procedure . scraping the lesion collects in situ cells . a certain degree of expertise is required for scraping and making the smear . in scrape cytology smear , cell - to - cell relation is not maintained . to compensate for all these drawbacks , scrape cytology offers a better cell yield even in keratinising lesions and small focal lesions . results of scrape cytology are likely to be more specific and sensitive . in our case , air - dried scrape cytology smears stained with giemsa stain revealed characteristic cytological features of hsv infection . it is important to establish the correct clinical diagnosis and confirm it with cytology smears . it is also important to distinguish herpes mastitis from bacterial mastitis since herpes mastitis heals spontaneously in 710 days without scarring , whereas bacterial abscess requires surgical drainage with residual scarring . in addition , it is important to distinguish herpes simplex mastitis from varicella zoster mastitis , because zoster infection can become confluent , hemorrhagic , heal slowly with scarring and can be associated with residual neuralgia . to conclude , in suspected cases , scrape smears can be of great value in the early diagnosis of herpes simplex mastitis .

herpes simplex virus ( hsv ) is a rare cause of breast infection . few cases of maternal - infant transmission of the virus during breastfeeding resulting in nipple lesions have been documented . only three cases have been reported in nonlactating women . we report an additional case of hsv mastitis in a 36-year - old nonlactating female who was diagnosed on scrape cytology smears .

in the previous issue of critical care , toufekoula and colleagues investigated the serum and tissue concentrations of malondialdehyde ( mda ) as readout for lipid peroxidation during sepsis . they submitted rats to sepsis by multidrug - resistant ( mdr ) isolates of pseudomonas aeruginosa , which showed significantly higher mda concentrations in liver , spleen and aorta compared to control animals . the rat data were compared to an historic cohort of patients ( n = 93 ) with mdr gram - negative bacteria - induced ventilator - associated pneumonia , in which mda serum concentrations during the first 7 days were elevated and correlated with survival during sepsis - induced hepatic or cardiopulmonary failure . sepsis induced by mdr gram - negative bacteria is a growing concern in intensive care units . a vulnerable patient population , long - term use of invasive medical equipment and increasing use of broad - spectrum antibiotics considerably affect overall morbidity , mortality and health care costs . despite the amount of research in the field of sepsis , a link between specific toxic mediators and patient outcome , which would enable biomarkers of organ failure to be defined and therapy adjusted accordingly , has not been established . the study by toufekoula and colleagues features a dual approach to the organ distribution pattern of lipid peroxidation during sepsis . in vivo data from rats this combined approach greatly enhances the value of the study since transferring results from rodent models to the clinical situation in septic patients is challenging . thus , comparing experimental and clinical data as presented by toufekoula and colleagues is an excellent example of translational research . in the rat sepsis model , however , in ventilator - associated pneumonia different strains of p. aeruginosa may differ considerably with regard to onset and mortality . moreover , the pattern in which organs are affected differs according to the infectious agent . thus , the patterns of mda production as reported by toufekoula and colleagues might not be extrapolated to sepsis induced by other types of bacteria , which emphasizes the need to stratify both rodent and human sepsis for the underlying pathogen . additionally , toufekoula and colleagues induced sepsis in the rats by intraperitoneal injection , while sepsis in patients was caused by bacterial migration through the lung . this might explain why patients with pulmonary failure ( that is , acute respiratory distress syndrome ) showed high circulating mda levels whereas pulmonary mda concentrations in septic rats remained rather unchanged . this highlights the fact that the way organs are affected in humans might differ markedly from animal models and may be directly dependent on the route of infection . recently , circulating lipid peroxidation products ( other than mda ) during sepsis were shown to correlate with increased hospital mortality rates . however , the present study shows that the concentrations of circulating mda have to be interpreted with utmost caution in septic patients . survivors among septic patients with hepatic dysfunction or with acute respiratory distress syndrome and concomitant cardiovascular failure showed higher circulating mda concentrations than non - survivors . in contrast , survivors with acute renal dysfunction displayed lower circulating mda levels than non - survivors . questions concerning the underlying mechanism or the hypothesis that a toxic end product may predict survival under certain circumstances can not be answered from this descriptive study . microdialysis is an established method to monitor both metabolic and inflammatory markers in clinical settings . several methods for mda detection in microdialysis fluid have been described and it would be of great interest to evaluate mda production directly in different organs throughout the course of sepsis . nevertheless , as an additional parameter , circulating mda may potentially become a valuable tool in evaluating septic patients concordant with the recently proposed model of ' bioscore ' combining different biomarkers in one model . understanding the pathophysiology during mdr p. aeruginosa - induced ventilator - associated pneumonia and sepsis is desperately needed as infection rates are likely to increase in the future and novel therapeutic options will not be readily at hand . the study by toufekoula and colleagues provides valuable information about mdr p. aeruginosa - induced sepsis and , thus , a data foundation for mda quantification in vital organs . although the data are entirely descriptive , the study sheds light on mda as a promising biomarker , which may potentially facilitate the evaluation of intensive care patients during the course of severe sepsis .

multidrug - resistant gram - negative induced sepsis poses an increasing threat to the vulnerable intensive care patient . the study by toufekoula and colleagues reports the serum and tissue concentration of malondialdehyde ( mda ) , the toxic end product of lipid peroxidation , during the course of experimental and human gram - negative sepsis . the complementary results from this dual experimental and clinical approach argue for highly compartmentalized lipid peroxidation during sepsis . establishing a correlation between mda concentration and survival provides valuable insights into the pathophysiology of gram - negative sepsis . yet , further studies are needed to understand and establish mda as a biomarker during sepsis aggravated by organ failure .

nmr spectroscopic data were recorded on a bruker avance 500 spectrometer equipped with cryogenic probe for h and c measurements ( bruker ) . gc - ms analyses were carried out using a varian 3900 gc with saturn 2100 ion trap mass spectrometer equipped with hp-5 ms capillary column ( 30 m 0.25 mm 0.25 m ) in electron ionization ( 70 ev ) mode ( agilent / varian ) . column chromatography was performed with 80 200 mesh silica gel ( fisher scientific ) . hplc was carried out with a zorbax eclipse xdb - c8 column ( 4.6 mm 150 mm , 5 m ) on an agilent 1100 series system equipped with fraction collector and diode array detector . the genes from s. arenicola described herein were cloned from the fosmids used in its genome sequencing project . these were cloned into the gateway vector system ( invitrogen ) to enable their use in the metabolic engineering system , including co - expression with previously characterized enzymes ( i.e. , to investigate configurations or increase yield ) . enzymatic products were analyzed by gc - ms of organic extracts from the relevant recombinant cultures . where necessary ( i.e. , for 3 and 4 ) , these cultures were scaled up to enable production and purification of larger quantities for structural characterization by nmr . previously described as a colorless solid;h and c nmr , as well as ms , data largely match literature values , with the few significant differences supported here by hmbc correlations ( si , table s1 ) . previously described as a colorless solid;h and c nmr data largely match literature values , again with the few significant differences supported here by hmbc correlations ( si , table s2 ) .

while more commonly associated with plants than microbes , diterpenoid natural products have been reported to have profound effects in marine microbe microbe interactions . intriguingly , the genome of the marine bacterium salinispora arenicola cns-205 contains a putative diterpenoid biosynthetic operon , terp1 . here recombinant expression studies are reported , indicating that this three - gene operon leads to the production of isopimara-8,15-dien-19-ol ( 4 ) . although 4 is not observed in pure cultures of s. arenicola , it is plausible that the terp1 operon is only expressed under certain physiologically relevant conditions such as in the presence of other marine organisms .

a new technological world of electrochemical sensorimotors mimicking biological muscles is emerging , requiring thick films of conducting polymers for their construction . here , we present how the best conditions to generate thick films of the polypyrrole / polyvynivlsuphonate blend are determined , how the selfsupported electrodes are constructed , and how their electrochemical and sensing properties are determined . the material can be applied to create sensing artificial muscles . an unexplored and unparalleled technological world is emerging , in which electrochemical tools and robots replicate the intracellular matrix content of functional cells ( ions water and reactive macromolecules ) , and their functions . haptic muscles , dual and simultaneous sensing motors , can now be replicated by sensing artificial muscles . the electrochemical reactions of the constitutive material drive , simultaneously , the movement rate of the motor ( through the flowing current ) and the mechanical , thermal , or chemical sensing ( through the muscle potential evolution ) of the working conditions . if each of the film constitutive chains is a reacting molecular motor : where is the electrical double layer ? ; how can electrochemical models include reactiondriven volume variations and conformational energetic changes of the reacting polymeric chains ? ; if we can mimic haptic muscles , can artificial proprioception and artificial proprioceptive robots be developed ? , could the attained results help to describe parallel biochemical functional reactions or biological functions and predict malfunctions ? development of structural and biomimetic electrochemical models and multifunctional electrochemical devices ( sensing motors , sensing motor batteries , sensing smart windows , artificial chemical synapse for neuron computer interfaces ) as well as electrochemoconformational memories that can actuate as multipotent memories for biomimetic computers , like possible brain memory models .

abstractinvited for this month 's cover picture is the group of professor toribio f. otero at the centre for electrochemistry , intelligent materials and devices at the polytechnic university of cartagena ( spain ) . the cover picture shows an electrochemical cell as well as three representative cyclic voltammetric responses , displaying the electrolyte potential window , the monomer oxidation polymerization potential range , and the polymer oxidation reduction potential window . for more details , read the full text of the full paper at 10.1002/open.201600139 .

clinical seizures as well as photoparoxysmal responses can be triggered by an intermittent photic stimulation ( ips ) , most commonly in generalized epilepsies.1 while some focal seizures are caused by an ips , most are caused by problems of the occipital lobe or visual cortex.2 now we are reporting the case of a 12-year - old female patient with a partial seizure triggered by an ips , proved to be a left temporal lobe seizure , which is supported by continuous video - eeg monitoring . a 12-year - old girl with a history of three convulsions was hospitalized for diagnostic evaluation including continuous video - eeg monitoring . her mother heard a crashing sound followed by regular gasping and sounds of her striking the floor . however , she did not see the convulsive movement because of the locked door . after the convulsions , the patient was confused for several minutes , and complained of nausea and generalized myalgia . she did not have any history of perinatal problems , developmental delay or febrile convulsions . she did not have any relatives with epilepsy in her family . in a neurological examination her mri scan with epilepsy protocol was also normal . during the continuous video - eeg monitoring , interestingly , this epileptic seizure was induced by photic stimulation . immediately after a 15-hz ips this was followed by a forced turning of the head to the right and then a secondary generalized tonic - clonic seizure . after a seizure of about 3 minutes 1 ) showed a photoparoxysmal response ( generalized spike and waves around 3 hz during 3 seconds ) during the 15-hz ips followed by ictal discharge with an onset of rhythmic alpha activity over the left temporal area ( t5 maximum ) . she did not complain or remember any visual problems , such as loss of sight , flickering lights , or visual hallucinations during the seizure or postictal period . the patient was given levetiracetam , 1,000 mg / d and had no seizures for 6 months . the follow - up eeg revealed photoparoxysmal response , however , no seizure was elicited by ips . a quarter of patients with spontaneous seizures and eeg photosensitivity belong to a variety of idiopathic epileptic syndromes , such as juvenile myoclonic epilepsy , childhood absence epilepsy and epilepsy with grand mal on awakening.1 the overall annual incidence of cases with newly presenting seizures and unequivocal photosensitivity in the united kingdom is 2% of all new cases with epileptic seizures.3 the photoparoxysmal response pattern is usually a generalized polyspikes and slow waves . this wave is highly associated with clinical photosensitivity and clinical events such as jerks or subjective sensations.4 since there have been several reports of photic stimulation induced occipital lobe seizures , some investigators believe that the origin of focal seizures that are photosensitive can only be occipital.1,57 in these cases , corticocortical non - specific visual afferents from the visual area to the frontorolandic cortex seem to be involved in photosensitivity and subsequent spreading occurs through cortical pathways and possibly through thalamocortical pathways.4 there have been a few reports describing photosensitive focal seizure arising from the temporal lobe . benbadis et al . has proposed that true temporal lobe epilepsy can also be photosensitive.7 their case is supported by electroclinical data and confirmed by a freedom from seizures after a temporal lobectomy . in 1999 , another three cases of complex partial seizures provoked by photic stimulation were reported.8,9 the authors suggested the possibility of activation of distant epileptogenic structure in temporal lobe by photic stimulation of the normal occipital cortex through cortico - cortical transmission . inoue et al . presented a case diagnosed from clinical symptoms , eegs , and a single photon emission computer tomography.10 they suggested the photic - driving occipital pathways and afferent pathways from the extraocular muscles or orbicularis oculi can cause temporal lobe seizures . there may be a possibility of the coexistence of idiopathic generalized epilepsy and focal epilepsy.1113 rei enatus reported a 13-year - old girl with well controlled generalized epilepsy and medically - refractory left temporal epilepsy.14 wolf and goosses found that 12 of 103 patients with photic - induced seizures had focal epilepsy . in 8 cases , there was associated evidence of generalized epilepsy and the photic - induced seizures were thought to be generalized . in our case , however , according to medical history , semiology and video - eeg monitoring , there was no evidence of the coexistence of idiopathic generalized epilepsy . the only seizure that we did observe was a complex partial seizure with secondary generalization of left temporal lobe onset . this is a rare case of photic stimulation induced focal seizure from the temporal lobe .

there are only a few case reports of photic stimulation induced partial seizures arising from the temporal lobe . a 12-year - old female with a history of three convulsions was admitted for a diagnostic evaluation . during continuous video - electroencephalogram monitoring , a complex partial seizure with secondary generalization was induced immediately after a photoparoxysmal response with a 15-hz photic stimulation . this is a rare case of photosensitive temporal lobe seizure .

brugada syndrome , first described in 1992 , is a life threatening disease and is usually overlooked during emergency service admissions.1 ) it is characterized by typical electrocardiography ( ecg ) resembling right bundle branch block ( rbbb ) , static or dynamic st - segment elevation in leads v 1 - 3 . there is familial tendency in some cases.2 ) a majority of patients have a structurally normal heart and are likely to remain asymptomatic , however they may present to emergency departments with syncope and various arrhythmias ( atrial fibrillation , ventricular tachycardia , ventricular fibrillation - cardiac arrest and etc.).1)2 ) therefore it is crucially important for emergency medicine physicians not to omit this potential diagnosis . herein we report a case with brugada syndrome which was iatrogenically unmasked after propafenone administration for atrial fibrillation . a 62-year - old male presented to the emergency department of a local hospital complaining of palpitation . because emergency department physicians interpreted the ecg as indicating atrial fibrillation , they administered intravenous propafenone ( fig . 1 ) . after this , the patient experienced dizziness and became hypotensive ( blood pressure : 80/50 mm hg ) . ecg after propafenone infusion showed an rbbb pattern , st - segment elevation in leads v 1 - 3 and st - segment depression in leads dii , diii and avf ( fig . he was thus immediately transferred to our center with the diagnosis of acute myocardial infarction . on admission to our center other than the above mentioned characteristics , ecg revealed atrial fibrillation with a ventricular rate of 155/min . given that he was symptomatic and hypotensive , direct current cardioversion was performed ( 100 joules ) . after shock , the ecg was in sinus rhythm and compatible with brugada syndrome ( fig . 3 ) . after cardioversion , his symptoms subsided and blood pressure returned to within the normal range . ecg changes were in accordance with iatrogenic unmasking of concealed type brugada syndrome . due to ethical reasons we did not give additional propafenone for challenge . from acquiring detailed history , we learnt that one of his sisters had the diagnosis of brugada syndrome ( fig . , we found that his daughter also had the asymptomatic type 2 ecg pattern of brugada . we recommended him for further electrophysiologic evaluation for possible ventricular arrhythmia detection and risk stratification . in the present paper , we report a case with brugada syndrome which was iatrogenically unmasked after propafenone administration for atrial fibrillation . as described in the introduction , the syndrome is commonly overlooked at admission but has several important diagnostic features . it is more common in males and asian populations.1)2 ) in brugada syndrome there are three typical types of repolarization presentation , based on the appearance of the st - segments in leads v 1 - 3 . type 1 has st - segment elevation , ending with a negative t wave , forming a " coved " appearance . type 2 has a downsloping st - segment elevation above baseline forming a " saddle back " st appearance . type 3 has the same saddleback appearance as type 2 , but with less st - segment elevation . ecg readings are dynamic and may transform from one type to another or may even completely normalize . increased vagal activity , fever , tricyclic antidepressant agents , or class i a , ic and iii antiarrhythmic agents may unmask the ecg similarly to that which occurred in our case.3)4 ) in conclusion , in case of admission with atrial fibrillation to emergency department , a commence of class ic antiarrhythmic agent in order to return to normal sinus rhythm may unmask concealed brugada syndrome . the appearance of post treatment st - segments elevation in leads v 1 - 3 with rbbb patterns should alert emergency physicians so that they do not omit this life threatening disease .

brugada syndrome is a life threatening disease that is usually overlooked during emergency service admissions . it is characterized by typical electrocardiography resembling right bundle branch block , static or dynamic st - segment elevation in leads v 1 - 3 . there is familial tendency in some cases . a majority of patients have a structurally normal heart and are likely to remain asymptomatic , however they may present to emergency departments with syncope and various serious arrhythmias . therefore it is crucially important for emergency medicine physicians not to omit this potential diagnosis . herein we report a case with brugada syndrome which was iatrogenically unmasked after propafenone administration for atrial fibrillation .

a semidominant locus in the vicinity of d14rat65 of chromosome 14 has been shown responsible for renal malformation . however , the renal lesion is assumed to occur in a polygenic trait . in the course of the genetic study on thymoma developments of buf / mna rats , we noticed that moderate substrain differences in the incidences of the renal malformations were found among congenic strains of control aci / mna rats with introgressed chromosomal segments from buf / mna rats . the rats of the inbred aci / mna ( aci ) , buf / mna ( buf ) and wky / ncrj ( wky ) strains , and of congenic strains , in which genetic regions of rat nude ( rnu ) , thymus enlargement-1 ( ten1 ) and thymus enlargement-2 ( ten2 ) , thymoma susceptible gene of rat-1 ( tsr1 ) , atrophy of fast - twitch muscle-1 ( aftm1 ) and proteinuria-1 ( pur1 ) were transferred into aci , buf or wky strain , respectively , were used . the buf - rnu/+ rats were raised during the course of the establishment of the buf - rnu / rnu strain , as described in the previous study . they were housed with free access to food ( cmf , oriental yeast , tokyo , japan ) and tap water at the animal facility of fujita health university . all animal experiments were approved based on the approval of the guide for the care and use of laboratory animals of fujita health university school of medicine . rats of these strains , as surpluses in the course of the maintenance of the strains , were killed at the age of 6104 weeks , except 3 rats died of bilateral renal malformations 15 days after birth . 1 ) in 113 % , but buf and wky , and their congenic rats hardly developed . since there were no sex differences in the incidences in renal agenesis and hydronephrosis in these strains , the data for both sexes were accumulated ( table 1 ) . higher incidences of renal agenesis in the right side and hydronephrosis in the left side , respectively , were observed . three rats died 15 days after birth ; 1 developed no kidneys in bilateral sides and other 2 did bilateral hydronephrosis ( table 1 ) . statistically , lower incidences of these developmental abnormalities were found in rats of the aci - pur1 and aci - pur1-ten2 strains than aci , aci - tsr1 , aci - ten1 , and aci - aftm1 strains ( table 1 ) . incidences of renal agenesis and hydronephrosis in rats of both sexes of the aci , buf , and wky strains and their congenic strains the statistical analyses were carried out by the fisher s exact test . highly significant ; significant ; highly significant ; significant ; significant ; significant ; highly significant ; highly significant . the unilateral renal agenesis , hydronephrosis , and associated genitourinary anomalies in aci rats were thought to be a part of a mesonephric duct defect . the inheritance of the unilateral renal agenesis is believed to be polygenic and one of the genes for unilateral renal agenesis was mapped on chromosome 14 . the present study revealed that the renal agenesis and hydronephrosis were significantly suppressed by the insertion of the genetic region of proteinuria in aci - pur1 and aci - pur1-ten2 rats originated from buf rats . conversely , it means that the pur1 region of aci rats contains a second gene for the induction of renal malformations . we previously showed that genetic pur1 region was located on the chromosome 13 and that the genetic pur1 region contained 38 genes , which should be analyzed by molecular methods . the present study also showed that aci rats developed unilateral hydronephrosis more in the left side . the exact mechanism why unilateral hydronephrosis occurs more in the left side is not known .

abstractthe aci rats developed hereditary renal malformations including agenesis and hydronephrosis at moderate penetrance . during construction of a variety of congenic strains based on aci / mna ( aci ) , buf / mna ( buf ) , and wky / ncrj ( wky ) rats , we found that the renal malformations were significantly suppressed by introgression of a segment of chromosome 13 of buf rats containing pur1 locus . it is plausible that this region contain a modifier locus influencing development of renal malformations .

a 14-fr foley catheter was accidentally placed into the right ureter in a 38-yr - old woman . a 38-yr - old paraplegic woman was referred to a hospital emergency room due to lower abdominal pain following a urethral catheter change . five years before , she had developed neurogenic bladder as a result of a 10th thoracic spinal cord injury . a catheter change had been performed by the nursing staff , using 14-fr foley catheter without undue events . in order to determine the location of the tube and rule out bladder rupture , there was considerable leakage of contrast media around the midureter , and the foley catheter tip was observed to be located at the right lower ureter ( fig . the bladder mucosa was intact and both ureteral orifices were in a typical position and normal shape . two weeks later , antegrade urography showed that the injured ureter had healed without leakage or obstruction ( fig . urethral catheterization is commonly performed and is usually safe , although complications , such as infection , bleeding , injury to the urethra or bladder , or catheter malfunction can ensue ( 1 ) . the two most common complications related to foley catheters , particularly in males , are urethral trauma and retention of the catheter balloon in the urethra ( 2 ) . serious complications such as bladder perforation and/or peritonitis ( 3 ) and rectovesical fistula ( 4 ) have been reported . the authors can not explain the cause of the present complication . because the ureteral orifice was orthotopically located , direct insertion of a 14-fr catheter , a large device , into the ureter through the ureteral orifice without pretreatment or mechanical dilatation is completely unexpected . it is unclear whether the right ureteric orifice was patulous at the time of catheterization . urethral catheterization into the ureter is more common when the patient is catheterized on an empty bladder ( 5 ) . the only way to avoid such injury would be adherence to the basic principles of catheterization , e.g. , to confirm the location of the balloon before inflation , by the aspiration of urine . in conclusion , although the current case is an extremely rare example , we should perform urethral catheterization with particular care .

we report an unusual complication caused by urethral catheterization . during a routine urethral catheter change in a 38-yr - old woman , a 14-fr foley catheter was accidentally placed into the right ureter through the ureteral orifice . the position of the catheter was confirmed by retrograde urogram through urethral catheter . percutaneous nephrostomy was performed with subsequent proper replacement of a urethral catheter . two weeks later , the injured ureter had healed without leakage or obstruction .

langerhans cell histiocytosis ( lch ) is characterized by a clonal proliferation of langerhans cells that occurs predominantly in children . lch is categorized as a single - system ( ss ) disease with single or multifocal lesions , and as a multi - system ( ms ) disease with or without involvement of risk organs ( hematopoietic system , lung , liver or spleen ) . although the skin is not categorized as a risk organ , the precise diagnosis of skin lesions is necessary to determine the protocol for the treatment of lch . in this report , we present a case of lch mimicking prurigo chronica multiplex that carried the p. v600e mutation in the braf gene , successfully detected by immunohistochemical staining . a 28-year - old japanese man visited our outpatient clinic with a 1-year history of pruritic papules on his extremities . he had noticed a slow - growing subcutaneous nodule on his right chest 2 years before , and half a year before this nodule had been diagnosed as adult - onset lch and irradiated locally . on his initial visit , physical examination revealed multiple crusted papules on the trunk , lower legs and dorsal region of the foot ( fig . a biopsy specimen showed atypical large lymphocytes infiltrated mainly in the perivascular region of the upper dermis with involvement of the overlying epidermis ( fig . since the infiltrating cells were limited to the perivascular areas and it was difficult to diagnose these eruptions as cutaneous involvement of lch or dermadromes of lch , we employed immunohistochemical staining for braf mutation . we detected p.v600e - mutated cells in the perivascular region of the upper dermis ( fig . , we diagnosed this patient as having adult - onset braf - mutated lch with cutaneous involvement . he was treated for lch with a regimen of the japan lch study group protocol c ( vinblastine , prednisolone , methotrexate , mercaptopurine ) as described previously . one month after the administration of this regimen , in parallel with the induction of complete remission of lch , his pruritic eruption disappeared . lch can roughly be divided into two categories : a ss disease with single or multifocal lesions , and a ms disease with or without the risk of organ involvement ( hematopoietic system , lung , liver or spleen ) . in adults , systemic chemotherapy is required for multifocal ss or ms lch lesions , whereas systemic chemotherapy is not recommended for those with localized ss disease . since cutaneous involvement of adult lch is frequent and its manifestation is variable , exact histological diagnosis of a skin lesion is necessary to determine the clinical type of lch . although lch is characterized by the proliferation of s100 protein , cd1a and langerin - positive cells , sometimes it is difficult to differentiate skin lesions of lch from other inflammatory skin diseases such as prurigo . recent reports suggested that the oncogenic braf mutation could be one of the markers for cutaneous lch . moreover , another report also suggested that braf mutation in circulating cd11c+/cd14 + cell fractions can even determine the prognosis of lch . notably , kobayashi and tojo reported that the braf mutation in circulating cell - free dna could be a biomarker of high - risk adult lch . these reports suggested that the expression of braf could be one of the diagnostic tools for the cutaneous involvement of lch with various presentations . although the skin is not categorized as a risk organ , the exact diagnosis of cutaneous involvement of lch is necessary to determine the protocol for the treatment of lch . since immunohistochemical staining and quantitative rt - pcr were useful for the detection of braf mutation [ 7 , 8 ] , in this report we selected immunohistochemical staining with 3,3-diaminobenzidine tetrahydrochloride and its enhancer . we could not detect p.v600e mutation in the braf gene by quantitative rt - pcr because of the lower cell densities in the skin lesion . our present report suggested that , in such cutaneous lesions , immunohistochemical staining is suitable for the detection of the gene mutation .

langerhans cell histiocytosis ( lch ) is characterized by the clonal proliferation of langerhans cells ; it is categorized as a single - system disease with single or multifocal lesions , and as a multi - system disease with or without the risk of organ involvement . although the skin is not categorized as a risk organ , the precise diagnosis of skin lesions is necessary to determine the protocol for the treatment of lch . in this report , we describe a 28-year - old japanese man with adult onset of brafv600e - mutated lch with cutaneous involvement successfully diagnosed by immunohistochemical staining . our report suggests that immunohistochemical staining for the brafv600e gene could be a diagnostic tool to determine the clinical type of lch .

erysipelothrix rhusiopathiae is a gram - positive , non - sporeforming , slender and straight or slightly rod - shaped bacterium that causes erysipelas in swine and a wide spectrum of diseases in other animals , like sheep , birds , reptiles , amphibians , and some fishes . this bacterium is ubiquitous in environment and in reservoir of asymptomatic carriers among both domestic and wild animals . among the 23 serotypes of e. rhusiopathiae , serotypes 1a , 1b and 2 are the best known as the etiological agents affecting swine industry , , , . e. rhusiopathiaes vary widely in their morphology , host specificity and/or pathogenicity and little is known about the correlation of their proteins to virulence . e. rhusiopathiae gxby-1 , isolated from acute swine erysipelas in binyang county , guangxi province , china , exhibits high pathogenic for mouse and swine . in order to elucidate the genetic background of this pathogenic strain and get deep insights into the virulence - associated proteins of this strain , we sequenced the complete genome of e. rhusiopathiae gxby-1 . the genome of e. rhusiopathiae gxby-1 was sequenced at beijing genomics institute ( bgi , shenzhen , china ) using illumina hiseq 2000 system . a total of 300 million high - quality base pairs were produced with 62-fold coverage of the genome , and then were assembled into 11 contigs by using soap denovo software , , after which the contigs were joined into 2 scaffolds with paired - end information . gene predictions and annotations were performed using the glimmer software ( version 3.02 ) and trna and rrna genes were identified by trnascan and rrnammer , respectively . all genes were further categorized according to swissprot , go ( gene ontology ) and kegg ( kyoto ency - clopedia of genes and genomes ) . the complete genome of e. rhusiopathiae gxby-1 consists of one 1,876,490 bp circular chromosome with no plasmid . and its chromosome contains 1734 predicted open reading frames ( orfs ) and the total length of genes is 1,668,564 bp , which makes up 88.36% of the genome . the g + c content of the chromosome is 36.50% and encodes 57 trna and 27 rrna operons ( table 1 ) . the gxby-1 strain genome not only enriches the genome database of e. rhusiopathiae , but also supports and extends previous studies , and provides fundamental information for further studies . the genome sequence of e. rhusiopathiae gxby-1 has been deposited in ncbi genbank under accession number cp014861 . the authors declare that there is no conflict of interests with respect to the work published in this paper .

erysipelothrix rhusiopathiae ( e. rhusiopathiae ) is an important pathogenic microorganism affecting swine industry . here , we report the finished annotated genome sequence of e. rhusiopathiae gxby-1 , isolated from acute swine erysipelas in binyang county , guangxi , china . the gxby-1 strain , which exhibits high pathogenicity for swine , contains 1,876,490 bp with g + c content of 36.50% , and contains 1734 protein - coding genes , 57 trnas and 27 rrnas . the nucleotide sequence of this genome was deposited into genbank under the accession cp014861 .

inflammatory and pseudodiverticula are the most frequent . only one case of a true diverticulum of the transverse colon has been reported in the literature . we report a case of a 22-year - old woman presenting with constipation and meteorism from childhood . a plain abdominal x - ray showed a round radiolucent air - filled cyst . the postoperative course was uneventful , and she was discharged in 1 week without any complications . in july 2010 , a 22-year - old woman was admitted with a history of constipation and meterorism that were longstanding from childhood . she had been previously diagnosed in other departments , and based on colonoscopy , dolichocolon had been suggested . physical examination revealed a large abdominal mass and cachexia ( bmi = 15.5 kg / m ) . barium enema revealed a single , large diverticulum of the transverse colon ( fig . 1 ) . laparotomy showed a giant diverticulum originating from the proximal part of the transverse colon that was 40 cm long , 1015 cm wide at the bottom and 45 cm wide at the gate ( fig . 2 ) . histopathology revealed that the giant diverticulum contained all four layers of the normal bowel wall ( fig . the postoperative course was uneventful , and she was discharged in 1 week without any complications . 3histopatological examination a giant diverticulum contains all four layers of the normal bowel wall barium enema showing a giant diverticulum of the transverse colon intraoperative image demonstrating a giant diverticulum of the transverse colon histopatological examination a giant diverticulum contains all four layers of the normal bowel wall a giant colonic diverticulum ( gcd ) is defined as a colonic diverticulum measuring 4 cm or larger.1,2 it is a very rare condition , and most frequently , it is associated with colonic diverticular disease . gcd was described first in 1953 by hughes and greene,3 primarily as a solitary air cyst . different names ( giant air cyst or giant cyst ) have been used to describe this condition . according to steenvoorde et al.,2 the term pathologically , gcd is divided into three types : type i ( 22% ) , pseudodiverticulum composed of granulation and fibrous tissue , with chronic inflammatory cells and remnants of muscularis mucosa ; type ii ( 66% ) , inflammatory diverticulum arising from local perforation and communicating with an abscess cavity and type iii ( 12% ) , true diverticulum that contains all the layers of normal bowel wall and being in continuity with the gut lumen.2,4 giant diverticulum located in the transverse colon is extremely rare.57 only one case of a true giant diverticulum of the transverse colon that was accompanied by a right inguinal hernia of the greater omentum has been reported in the literature.5 we present the unique case of an uncomplicated true giant diverticulum of the transverse colon . because symptoms have been remaining from patient s childhood , we believe that this pathology can be congenital due to an intestinal duplication . gcd may be asymptomatic or presents with nonspecific symptoms , such as vague abdominal pain , constipation , rectal bleeding , nausea and vomiting , abdominal distension , diarrhoea and abdominal mass.2 in 28% of patients , complications such as inflammation , perforation , intraabdominal abscess formation and wall infarction occur . 4 a plain supine abdominal x - ray is the radiological investigation of choice for gcd diagnosis.2 preoperative diagnosis may also include barium enema , ct scan or mri.5,8 diverticulectomy in selected cases or partial colectomy with the diverticulum is the preferred method of treatment in uncomplicated gcd . in complicated cases ,

abstractgiant colonic diverticulum is an extremely rare condition in colonic diverticular disease . more than 90% of giant colonic diverticula are found in the sigmoid colon . inflammatory and pseudodiverticula are the most frequent . only one case of a true diverticulum of the transverse colon has been reported in the literature.case reportwe report a case of a 22-year - old woman presenting with constipation and meteorism from childhood . a plain abdominal x - ray showed a round radiolucent air - filled cyst . barium enema revealed a single , large diverticulum of the transverse colon . an extended right hemicolectomy with primary end - to - end anastomosis was performed . the postoperative course was uneventful , and she was discharged in 1 week without any complications . histopathology showed a true diverticulum containing all layers of the colon .

she underwent nephrectomy , and the histology revealed a renal cell carcinoma ( rcc ) of chromophobe type , eosinophilic variant . maximum tumor diameter was 12 cm , with extensive necrosis , grade iv according to fuhrman 's nuclear grading system . pn1 cm0 . neither the renal vein nor the adrenal gland was infiltrated . in august 2007 , treatment with sunitinib 50 mg on a 4 weeks on/2 weeks off schedule was initiated , resulting in stable disease . however , the doses had to be reduced due to grade 3 palmar and plantar toxicity . at the end of march 2009 , after 20 months of treatment , an mri showed progressive liver disease as well as local recurrence and mesenteric lymph node metastases . the initial dose of 800 mg daily had to be reduced to 600 mg due to a hematologic toxicity . no objective response was achieved , and the performance status decreased from 0 to 2 , with a 7-kg weight loss . after 5 months of sorafenib therapy , an mri revealed further progression at all sites ( fig . 1 ; online suppl . video 1 ; see www.karger.com/doi/10.1159/000323804 ) . at the end of august 2009 , temsirolimus was started as a third - line treatment at a dose of 25 mg intravenously every week . in november 2009 , disease stabilization was observed on ct . in february 2009 , a further mri showed a partial remission according to the recist criteria . clinically , the patient had improved considerably ( ps 0 ) , and formerly elevated laboratory values normalized ( ldh from 1,400 u / l , alkaline phosphatase from 142 u / l , gt from 168 currently , the patient continues on weekly temsirolimus , is working full time , and is in excellent physical condition without experiencing any side effects . this clinical observation of an impressive response to third - line temsirolimus in a case of chromophobe metastatic rcc leads us to raise the following questions : first , should the current practice of sequential therapy be revisited ? especially in chromophobe metastatic rcc , mtor inhibition may be considered earlier . in this patient , today , everolimus is the standard in second - line treatment , while temsirolimus is approved only for first - line treatment . second , are all mtor inhibitors equal in terms of efficacy ? would this patient have responded equally well to everolimus ? this question should be addressed by head - to - head comparisons of mtor inhibitors . since 2006 , six molecular targeted agents have been approved for the treatment of rcc ( sunitinib , sorafenib , bevacizumab / interferon , temsirolimus , everolimus , and pazopanib ) . maybe more emphasis should be placed on the biology of rcc as reflected in its histology . rare subtypes like chromophobe rcc or sarcomatoid dedifferentiated rcc may have to be considered separately . current strategies are based on results obtained in the context of clear cell - type rcc . separate trials for rare histologies seem unfeasible and are unlikely to be performed . for these cases , currently , the start trial is studying temsirolimus as first - line and second - line treatment . patients in this trial are stratified by histological subtype , and the results may also answer the question raised by our observation : is chromophobe rcc particularly sensitive to mtor inhibition ?

we report unexpectedly high efficacy of temsirolimus as third - line treatment in a patient with metastatic chromophobe renal cell carcinoma . after failure of two sequentially administered tyrosine kinase inhibitors , treatment with temsirolimus resulted in a prolonged partial remission of 14 months , and the response is still continuing . up to now , no data from randomized clinical studies have been published addressing the question of efficacy of temsirolimus as third - line treatment after failure of tyrosine kinase inhibitors . the case presented here implies that temsirolimus could be a viable option for patients with metastatic chromophobe renal cell carcinoma .

renal transplant recipients have a higher risk for infection and malignancy due to immunosuppression than the general population . kaposi sarcoma is classified in 4 types : 1 = classic kaposi sarcoma , 2 = iatrogenic ( immunosuppressive kaposi sarcoma ) , 3 = aids - associated ( epidemic ) kaposi sarcoma and 4 = endemic kaposi sarcoma . it is well known that kaposi sarcoma is associated with human herpes virus 8 ( hhv-8 ) . in 0.5 - 5% of seropositive transplant recipients reactivated hhv-8 leads to kaposi sarcoma [ 1 , 2 ] . in northern europe the seroprevalence of hhv-8 is 0 - 8% , whereas in the mediterranean area and in the middle east the seroprevalence is 10 - 25% . kaposi sarcoma after renal transplantation is mainly found in patients of the mediterranean area and of middle eastern descent . we report for the first time a case of a successful second renal transplantation using sirolimus in a patient with a history of kaposi sarcoma . the study by stallone et al . showed a complete remission of kaposi sarcoma after conversion from calcineurin inhibitor to sirolimus within 6 months . we report on a 42-year - old male patient who developed kaposi sarcoma with visceral involvement 38 months after living - related renal transplantation . the patient originates from turkey and has been living in germany since he was born . end - stage renal disease was due to chronic glomerulonephritis , and the patient was on hemodialysis for 6 months prior to transplantation . subsequently , the patient lost his graft 30 months after diagnosis of kaposi sarcoma due to chronic rejection . no signs of recurrent disease were present on physical examination and on ct scan 13 months after cessation of immunosuppression . the patient was wait - listed after a period of 10 years and received a second transplant 5 weeks thereafter . his immunosuppressive regimen includes sirolimus ( 2 mg / day ) , mycophenolate mofetil ( 1,000 mg / day ) and prednisone ( 4 mg / day ) . after a follow - up of 4.5 years , the patient showed stable graft function with a serum creatinine of 1.2 mg / dl ( fig . 2 ) . recurrence of kaposi sarcoma is closely monitored by physical examination and ct scans . to our knowledge , this is the first report on a renal transplant recipient with a previous history of transplant - related kaposi sarcoma undergoing de novo renal transplantation with favorable long - term graft function and tumor control . . reported on 15 renal transplant recipients with kaposi sarcoma in whom cyclosporine was replaced by sirolimus to achieve tumor control . a complete remission within several months was documented for the entire patient cohort , with no impact on graft function and no acute episodes of rejection . previously , sirolimus - based immunosuppression has been shown to protect renal transplant recipients from de novo malignancies . in renal transplant recipients diagnosed with transplant - associated kaposi sarcoma , conversion to sirolimus - based immunosuppressive protocols however , the use of less potent immunosuppressives bears the risk of early graft loss due to acute or chronic rejection . an immunosuppressive protocol based on antiproliferative drugs , including sirolimus and mmf , has been shown to protect renal grafts from rejection and to provide reliable tumor control as compared to standard calcineurin - based immunosuppression . our present case and recent literature suggest that de novo renal transplantation in recipients with a history of transplant - associated malignancy is feasible using an immunosuppressive protocol based on sirolimus and mmf .

immunosuppressive treatment increases the risk of infection and malignancy in organ transplant recipients . we report on a 42-year - old male renal transplant recipient who lost his first graft after reduction of immunosuppressive treatment due to kaposi sarcoma and who successfully underwent a second renal transplant 10 years later . the patient 's current treatment consists of low - dose prednisone , and the two antiproliferative immunosuppressants mycophenolate mofetil and rapamycin . 4.5 years after his second transplant , the serum creatinine is 1 mg / dl and the patient has no signs of recurrent disease .

acute suppurative thyroiditis ( ast ) accompanied by an abscess is a rare clinical case . possible reasons for the infrequency of ast include the gland 's abundant blood supply and lymphatic drainage and the antimicrobial action of iodine 1 . ast is found primarily in children . according to miyauchi et al . , the most common mechanism for thyroid infection is the transmission of infective organisms via a pyriform sinus fistula 2 . mainstream management of ast is antimicrobial therapy , directed against the likely bacterial pathogens . here a 65yearold japanese man was admitted complaining of painful swelling in the neck and with high fever . stent placement in left superficial femoral artery for arteriosclerosis obliterans ( aso ) had been performed 45 days before this episode in our hospital . on examination , his height , body weight , and body mass index values were 156 cm , 53.3 kg , and 21.9 kg / m , respectively . a swollen mass was palpable on his left neck with erythematous change , hot feeling , and tenderness . laboratory data revealed white cell count : 12,500 ( 40008000)/l , creactive protein ( crp ) : 26.0 ( 0.30.6 ) mg / dl , blood urea nitrogen ( bun ) : 34 ( 921 ) mg / dl , serum creatinine ( cr ) : 6.89 ( 0.20.9 ) mg / dl , free t4 : 1.69 ( 0.91.8 ) ng / dl , thyroidstimulating hormone ( tsh ) : 0.52 ( 0.343.5 ) iu / ml , hba1c : 5.7 ( ngsp : < 6.5)% , and postprandial blood glucose : 123 ( < 200 ) mg / dl . neck ultrasonography revealed a wellcircumscribed large cystic mass with debris ( 56 51 32 mm ) in left thyroid gland ( fig . 1a and b ) . computed tomography also revealed a lowdensity lesion in left lobe of thyroid gland ( fig . thus , he was diagnosed as suffering from acute suppurative thyroiditis ( ast ) in infected thyroid cyst . intravenous administration of meropenem 1 g / day was started . on the fourth hospital day the aspirated pus from thyroid cyst and blood were sent for microbial culture and sensitivity test . 2 ) . second computer tomography revealed incidental aortic dissection ( stanford type b ) . thoracic endovascular aneurysm repair ( tevar ) surgery was performed for aortic dissection on the 60th hospital day . 2 ) . four months after discharge , neck ultrasonography showed resolution of the supportive thyroiditis ( fig . ( a , b ) wellcircumscribed large cystic mass with debris in left thyroid gland in neck at ultrasonography . ( c ) an abscess in the left lobe of thyroid gland in neck ct ( arrow ) . mepm , meropenem ; vcm , vancomycin ; sbt / abpc , sulbactam / ampicillin ; ampc , amoxicillin ; tevar , thoracic endovascular aneurysm repair . ( a ) cystic area in the left lobe decreased in volume after drainage and antibiotics . ( b ) thyroid gland was almost cured with heterogenous scarred upper middle of left lobe . acute suppurative thyroiditis ( ast ) is a rare infectious disease of the thyroid gland . analysis of 1309 thyroid operations performed during twentytwo years revealed 117 instances of thyroiditis , but only six of these were suppurative 3 . pyriform sinus fistulae related to third and fourth branchial pouch anomalies were one of the causes of ast 4 . in the present case , there were two atypical aspects : ( i ) the onset at middle age and ( ii ) pyriform sinus fistula was not found . no invasive infectious sign on surrounding tissue of the thyroid was revealed by neck ultrasonography . two possible precipitants may be susceptible to infection in hemodialysis and stent placement for aso . some recognized causes of ast are neoplastic thyroid nodule , subacute thyroiditis , hashimoto thyroiditis , and penetration of the thyroid gland by foreign bodies such as fish bones and trauma 5 , 6 , 7 . underlying diseases including tuberculosis 8 , diabetes mellitus 9 , and human immunodeficiency virus infection 10 appear to increase the risk for developing of thyroid abscess . although diabetes is recognized to lead to poor immune response , in this case , the glycemic control ( as represented by hba1c ) was very good . hemodialysis patients are at risk for infections 11 . according to the dialysis patient death registry and the reports on sepsisrelated mortality compared to that of the general population , sepsis mortality was from 100 to 300 times higher for chronic dialysis patients than that for the general public 12 . wang et al . , hypothesized that this association includes increased susceptibility to infection , the presence of comorbidities such as diabetes , and repetitive exposure to pathogens during hemodialysis 13 . mh , nk , ya , hu , kt , and th : were responsible for correction of the article and supervision .

key clinical messageacute suppurative thyroiditis ( ast ) accompanied by an abscess is a rare clinical case . hemodialysis patients are at risk for infections . sepsis mortality was from 100 to 300 times higher for chronic dialysis patients than that for the general public . thus , special care should be taken against infection in patients under hemodialysis .

diogenes syndrome is a rarely reported syndrome of extreme self - neglect , social withdrawal and domestic squalor . other synonyms for diogenes syndrome include severe self - neglect syndrome , aged recluse and social breakdown of the elderly . herein , we report a case of diogenes syndrome presenting with an unusual mask of dirt resembling a carapace . a 55-year - old lady with crusted plaques over head , neck and trunk since 3 months was brought to the hospital by her relatives . the onset of neglect coincided with the awareness of a lump in her right breast . there were adherent crusts over scalp , large dark brown confluent thick crusts forming a mask resembling a carapace over face and ears sparing the perioral area [ figure 1a and b ] . dark brown dirt like papules were found on neck , lower back , intermammary and inframammary area . there was poor oral hygiene and halitosis.a 6 8 cm solitary , firm , non - tender , mobile mass was present in her right breast [ figure 2 ] . ( a and b ) mask of dirt resembling a carapace complete blood counts , fasting blood sugar , liver and renal function tests were within normal limits . a skin biopsy revealed hyperkeratosis , increase in basal pigmentation and focal vacuolar changes [ figure 3a ] . pas stain showed few budding fungal yeast forms in the stratum corneum [ figure 3b ] . ( a ) hyperkeratosis with yeasts in stratum corneum ( h and e 40 ) . ( b ) pas - positive budding yeasts ( pas 40 ) right breast lumpectomy specimen had a biphasic pattern composed of glands and cellular stroma on histopathological examination . the glands showed leaf - like projections due to underlying proliferation of cellular spindle - shaped stromal cells . three months after removal of the lump the patient returned for follow up with a clean face and renewed interest in social activities . the term diogenes syndrome was first coined by clark et al . , in 1975 , to describe elderly patients with severe self - neglect , poor personal and domestic hygiene . it is named after the cynical greek philosopher diogene de sinope who advocated minimizing needs and a natural way of life . nutritional deficiencies of iron , folate , vitamin b12 , vitamin c , vitamin d and serum proteins may be seen . mental illness , withdrawal and denial of need in old age may be triggered by various stressful situations . the cutaneous manifestations of neglect ( dermatitis passivata ) arise from accretion of keratin and dirt and resemble a carapace . sparing of the perioral area could have resulted from her habit of wiping the perioral area after meals . vigorous rubbing with soap and water can cause complete clearance of lesions arising from neglect . terra firma forme dermatosis presents with dirt like patches which can be cleared with isopropyl alcohol but not soap and water . it can be differentiated by the absence of neglect and presence of adequate hygiene . though dermatitis passivata may be commonly seen in clinical practice , awareness about diogenes syndrome with its wider health and psychosocial implications is needed . our patient improved with the manual removal of crusts , psychiatric counseling and removal of the lump . our case is unusual in its mask of dirt resembling a carapace and the presence of a breast lump being a possible stressor . diogenes syndrome presenting with an unusual mask like carapace and responding to clinical and psychological interventions was seen .

diogenes syndrome is characterized by extreme self - neglect , social withdrawal , and poor personal and domestic hygiene . we report a case of diogenes syndrome presenting with dermatitis passivata . an unusual mask of dirt resembling a carapace , onset of neglect after awareness of a breast lump and resumption of personal grooming and social activities after removal of the lump and counseling were seen .

oesophageal stents are used widely to treat benign strictures , oesophageal perforations and fistula and for palliative therapy of oesophageal cancer . oesophageal migration has been reported to occur in 3.6% of cases , and the majority are removed or repositioned endoscopically without complication . this case presentation illustrates a rare and potentially lethal complication of stent migration resulting in mechanical small bowel obstruction . an 81-year - old female with a 24 h history of acute , worsening abdominal pain with associated abdominal distension and bilious vomiting was referred by her gp to the general surgical admissions unit . the patient reported no other gastrointestinal symptoms and there was no prelude to the presentation . she had undergone a billroth i gastrectomy in 1977 for peptic ulcer disease , a splenectomy following blunt trauma in 1989 and further gastric surgery for a cancer in 1999 . during a therapeutic endoscopy for a benign stricture this was treated endoscopically with the deployment of a hanaro - antireflux covered stent , covering the defect that was identified at 35 cm . these margins were chosen in order to give a 3 cm cover of the perforation . the extra 2 cm distal cover was because the stent was the only suitable stent available that would be both wide enough to remain in situ and long enough to give adequate cover of the perforation . there were no concerns regarding the apposition of the stent , and the stent was not sutured in place . routine follow - up was arranged for 46 weeks ; however , for an unconfirmed reason , this follow - up never took place . on presentation , blood results on admission , including liver function , urea and electrolytes , crp , amylase , lactate and full blood count , were within normal limits . abdominal x ray showed multiple distended loops of small bowel with a migrated stent inside small bowel fig . 1 . abdominal films show multiple dilated loops of small bowel with a visible stent in the abdomen . abdominal x ray taken shortly after presentation . abdominal films show multiple dilated loops of small bowel with a visible stent in the abdomen . following the diagnosis of mechanical small bowel obstruction due to oesphageal stent migration , the patient was booked and consented for a laparotomy . at laparotomy , distended small bowel was found , with the cause of the obstruction being found in the distal ileum . post - operative recovery was unremarkable , and the patient was discharged home with regular follow - up in 68 weeks . in cases of iatrogenic oesophageal perforation , stent placement has been shown to be an effective treatment , and has less associated morbidity and mortality than operative repair . however , the main complication of stent placement is migration , requiring repositioning or replacement . if stent migration is a risk due to large defects , or minimal anatomic barriers to migration , some authors advocate suturing stent into oesophagus , which may avoid this complication . in this case , the patient had unusual anatomy due to her previous surgeries . the stent migration may have been aided by the fact that the patient did not have a pylorus , allowing easy passage into the small bowel . clinicians should consider the risks of stent migration , and make the patient aware of these risks . oesophageal stent migration is a known complication of stent placement , occurring in 3.6% of cases of benign oesophageal conditions requiring stent placement . in a case series of 70 patients where migration has occurred , the authors found that the majority of stents migrate no further than the stomach , exit via the rectum or remain in the body with minimal complications . migration occurred in 11 of the 43 patients ( 25% ) with benign cause of strictures and in 591 of the 845 patients ( 7.0% ) with malignant cause . in the 70 patients , only in 3 was it surgically removed due to intestinal obstruction . in a prospective study of 18 iatrogenic oesophageal perforations treated with stents , all the stents were removed at a mean of 50 20 days . in this case , the stent was placed as a temporary measure to allow closure of the perforation . routine follow - up for removal was arranged , but the patient was lost in follow - up . although complications due to stent migration are rare , perforation , obstruction and haemorrhage all carry significant risks , with high mortality and morbidity . this case illustrates the importance of prompt patient follow - up in order for the stent to be removed . this case also illustrates the importance of making patients aware of the small risk of bowel obstruction and seeking urgent medical attention if the symptoms of bowel obstruction are present .

we report the case of an elderly female patient with a history of a previous billroth i gastrectomy who presented with small bowel obstruction secondary to a migrated oesophageal stent . this patient had an iatrogenic oesophageal perforation following therapeutic endoscopy for a benign stricture 4 months prior to presentation . this was treated endoscopically with a covered stent that was not removed as planned . the stent migrated distally lodging in the terminal ileum , causing small bowel obstruction . oesophageal stent migration is a rare but well - recognized complication of stent placement . endoscopic stenting is an effective treatment for oesophageal perforation with lower morbidity and mortality than operative repair . clinicians should be aware that although patients with a history of previous gastric surgery are at no greater risk of stent migration than others , the altered anatomy can affect the final resting place of the migrated stent and hence the clinical effects and sequalae .

amyand 's hernia is a rare presentation of acute appendicitis , in which an incarcerated or perforated appendix is found in the right inguinal canal . only a few cases have been diagnosed either by computed tomography ( ct ) or ultrasonography before operation . we report a 24-day - old neonate who presented atypically with tender swelling in the right scrotum . ultrasonography findings suggested amyand 's hernia preoperatively , which was confirmed by histopathological examination following surgery . a 24-day - old male neonate presented at the department of pediatric surgery of the first hospital of jilin university in changchun , china with right scrotal swelling for 4 days . local examination revealed a firm , tender swelling of 3.0 2.0 2.0 cm in the right inguinal region extending into the scrotum . ultrasonography detected a colon - type echo in the right inguinal canal [ figure 1 ] measuring 30.0 5.0 mm . the wall of the colon - type structure measured 3.5 mm in diameter and was thickened . the right testis measured 9.9 5.2 mm with an anechoic region of 7.0 12.0 mm anterior to the right testis [ figure 2 ] . an ultrasonic diagnosis of right inguinal hernia and right encapsulated hydrocele of the tunica virginals was considered . longitudinal ultrasonography view of the incarcerated appendix ( arrows ) ultrasonogram demonstrates the right testis ( arrows ) and hydrocele of the right testis since the patient 's presentation was not typical of a hernia , a primary clinical diagnosis of testicular tumor or epididymitis was considered . because the neonate was considered to have epididymitis , he was started on antibiotics for 2 days . repeat ultrasonography showed similar features as before , including right inguinal hernia and encysted hydrocele in the right scrotum . in addition , the tunica vaginalis , right testis and epididymis were found to be hyperemic and swollen with discharge of purulent material . the tunica vaginalis also contained a colon - type structure adhering to the swollen and inflamed right testis and epididymis . this colon - type structure extended into the right inguinal region and was identified as the appendix . an appendectomy was performed through the inguinal incision alone , and the right hernia sac was ligated . histopathological examination of the appendix specimen revealed fibrous tissue hyperplasia in the interstitium , with acute and chronic inflammatory cell infiltration , vasodilatation , and congestion . an ultrasonography scan 1 week later revealed a normal scrotum and testes on both sides , and the patient was discharged from the hospital . there have been fewer than 200 cases of amyand 's hernia in the literature ; however , only two cases have presented as scrotal swelling . although acute scrotum is a common clinical presentation of torsion of the testis or its appendages and epididymo - orchitis , incarcerated hernia and scrotal and/or inguinal abscess should also be considered in the differential diagnosis . clinical presentations of torsion caused by undescended testes may be quite similar to those observed in amyand 's hernia . ultrasonography is useful in distinguishing torsion of undescended testes from amyand 's hernia because of its high efficiency , low exposure , and real - time properties . ultrasonography can detect hernias with sacculations and moving content of the colon . in this case , ultrasonography detected a soft tissue structure similar to a thick wall of the colon without sacculations , and the hernia contained an isolated soft tissue with a thick wall and moving contents in the inguinal canal . second , ultrasonography , due to its real - time monitoring , can identify the moving content in the intestine . testicular tumors and epididymitis can be reasonably excluded even though these conditions have similar symptoms . in the present case , ultrasonography identified an inflamed appendix and encapsulated fluid around the right testis that was confirmed by surgery as transudate due to appendicitis . in this case , a neonate presented with right scrotal swelling without any signs of peritonitis . this was most likely because inflammatory transudate surrounded only the testis , and thus irritated the testis and its surrounding tissues . since the appendix , rather than the colon , was incarcerated in the inguinal canal , symptoms of intestinal rhythm alteration were not present . specifically , ultrasonography revealed the presence of an echogenic intestinal canal in the right inguinal canal and encapsulated hydrocele of the tunica vaginalis . in conclusion , we would like to highlight that ultrasonography might be a valuable tool in the preoperative diagnosis of amyand 's hernia .

acute appendicitis secondary to hernia incarceration presenting as scrotal swelling is exceptionally rare in neonates . we report a neonate who presented with tender swelling in the right scrotum . ultrasonography detected features of a rare amyand 's hernia . surgical exploration and histopathological examination confirmed the diagnosis .

a 46-year - old male had been admitted in the intensive care unit ( icu ) for severe chronic obstructive pulmonary disease ( copd ; bronchial asthma ) . based on the clinical picture of acute bilateral proptosis with retracted lids [ fig . 1 ] and patient 's history of similar episode in the past , a diagnosis of globe luxation was made and the globe was reduced by pressing on the superior sclera down and back while pinching the upper lid [ figs . 2 and 3 ] . history from the patient and his wife revealed that he had previous such episodes over past 45 years . the episodes initiated after he started developing wheezing episodes and most of the globe luxation episodes occurred along with exacerbations of copd . ct scan showed a proptosed eye with a stretched optic nerve because patient developed luxation while ct was being performed . right eye still luxated both globes have been reduced ct scan showing bilateral prominent globe luxation with stretched optic nerve during his stay in icu , the patient had repeated episodes of globe luxation ( almost every 2 hours ) which was reduced each time . bilateral temporary tarsorrhaphy was done after five to six episodes of repeated globe luxation to prevent the repeated proptosis . unfortunately , the patient developed complications such as respiratory paralysis and septicemia and expired on the third day of admission . kunesh et al . reviewed and found 26 reported cases of spontaneous globe luxation till 2001 . after that , another four case reports regarding spontaneous globe luxation could be identified in literature review till 2010 . spontaneous globe luxation occurs in patients with thyroid eye disease , proptosis , floppy eyelid syndrome and shallow orbit . luxation can be precipitated by lid manipulation , valsalva maneuver , trauma , general anesthesia and even by contact lens insertion . structural anomalies resulting in laxity of orbital ligaments and extraocular muscles can predispose to globe luxation . once the luxation starts , the orbicularis muscle goes into spasm to maintain the state of luxation . the case reported here had a history of globe luxation whose onset correlated with increased severity of copd . preexistent weaknesses of extraocular muscles and ligaments could have predisposed the patient for globe luxation . review of literature did not reveal any such case of luxation being precipitated and aggravated by copd . management consists of simple reducing maneuvers like asking the patient to look down , pinching the upper lid and pressing on the superior sclera down and back . this simple technique can be taught to the patient also and can help to avoid severe anxiety when the patient develops such an episode where help may not be available . surgical treatment consists of lateral tarsorrhaphy , pentagonal wedge eyelid resection and advancement of orbital wall . but lateral tarsorrhaphy has also been reported to increase the risk of luxation by producing a tighter orbit and increased intraorbital pressure , and once luxation occurs , the smaller remaining palpebral aperture can hinder the reduction of the globe .

spontaneous globe luxation is a rarely reported condition which can lead to complications like optic neuropathy . common causes are thyroid eye disease , shallow orbit and floppy eyelid syndrome . we report a case of spontaneous globe luxation with the onset and severity associated with chronic obstructive pulmonary disease ( copd ) . to our knowledge , this is the first case of spontaneous globe luxation associated with copd .

a 24-year - old woman was referred to our center at 28 weeks gestationbecause of suspected fetal cardiac anomalies . we echocardiographically diagnosed cctga and additional cardiac anomalies in utero at 28 weeks gestation ( gravida : 3 para : 2 ) . associated cardiac lesions includedventricular septal defect , mild pulmonary valve stenosis , tricuspid valvar displacement and moderate tricuspid regurgitation ( figure 13 ) . situs solitus with levocardia . a detailed examination of the ventricular morphology helped to confirm that the right - sided right atrium entered , via a morphologic mitral valve ( mv ) , the right - sided left ventricle ( lv ) , which in turn emptied into the posterior , right - sided pulmonary artery ( pa ) instead the left - sided left atrium was connected , through a morphologic tricuspid valve ( tv ) , to the left - sided right ventricle ( rv ) , which emptied into the anterior , left - sided aorta ( ao ) , ventricular septal defect ventricular septal defect ( vsd ) . note the ventriculoarterial discordance : the aortaarises from the left - sided , morphologically right ventricle instead bifurcating pulmonary artery arising from the morphological left ventricle , ventricular septal defect ventricular septal defect ( vsd ) . the pulmonary bifurcating ( pulm ) is in the middle between ascending aorta ( ao ) and superior vena cava ( svc ) , almost aligned . this fetus experienced supraventricular tachycardia ( svt ) in pregnancy ( at 35 weeks gestation ) ( fig 4 ) . postnatal echocardiography found visceral situs solitus , visceroatrial concordance , and atrioventricular and ventriculoarterial discordance , ventricular septal defect , pulmonary outflow obstruction ( mild ) , tricuspid valvar displacement , mild tricuspid regurgitation . postnatal svt recurred ( figure 6 ) electrocardiographic and holter monitor evidence was confirmed , in which initial treatment with adenosine was ineffective , but amiadarone was effective . congenitally corrected transposition is a rare congenital anomaly , with only a few cases diagnosed and reported prenatally even in the largest fetal series.12 prenatal sonographic diagnosis of ctga is very difficult because the ventricular outflow tract may appear to arise correctly from the right and left ventricles . the long - term prognosis of cctga is dependent on associated other major cardiac anomalies and rhythm disturbances that can influence prognosis.3 svt is reported to account for 47 68 % of cases of fetal tachycardiaand is associated with a low percentage of structural abnormalities ( 2 % of cases).4 fetal tachycardia of any form that is intermittent , not accompanied by cardiac or valve dysfunction , and present < 50% of the time is best not treated , but needs to be monitoredclosely . schnabel reported cctga at 20weeks gestation with recurrent svt.4 some of these cases may undergo major and unexpected changes , particularly with regard to cardiac rhythm and tricuspid valvar function , with concomitant significant changes in prognosis . the diagnosis of these specific types of fetal tachycardia is difficult , if not impossible with the most widely used current technology . the electrophysiologic events are clearly defined in this situation , however , a precise diagnosis is still debatable.4 in this case , postnatal svt recurred , electrocardiographic and holter monitor evidence was confirmed , and the initial treatment with adenosine was ineffective , but amiadarone was effective . the svt shown in postnatal ekg ( figure 3 ) for instance , may be avrt using a concealed accessory pathway . anti - arrhythmic therapy are started when arrhythmias were sustained or associated with hemodynamic compromise prior to 34 weeks gestation . after 34 weeks gestation , such cases are delivered.5 furthermore , some controversy exists in relation to first - line and second - line treatment.regimens for fetal supraventricular tachycardia . in general , digoxin is used as first - line treatment in the us , whereas in europe flecainide is used for this purpose . for second - line treatment of drug - refractory supraventricular tachycardia with hydrops fetalis , maternal intravenous or oral digoxin , alone or in combination with amiodarone can be used without increasing mortality . for fetuses without ventricular dysfunction , flecainide or sotalol have been used as second line agents , and both have efficacies of 6080% . however , for fetuses with ventricular dysfunction or severe atrioventricular valve regurgitation , amiodarone is the most effective drug ( > 93% efficacy alone or in combination ) , and is associated with a low risk of proarrhythmia.56 cesarian deliveries is advocated for fetal distress associated with persistent svt such as in our patient at 35 weeks of gestation . in conclusion , we think thataccurate fetal segmental analysis of the four - chamber and great vessel connections and relations ( l - transposition ) by fetal echocardiography could be highly helpful in improving follow - up of potentially critical neonates ( especially irregular heartbeat ) . the combined presence of svt and cctga are rarely reported in the literature during fetal monitoring.4

the prenatal sonographic diagnosis of congenitally corrected transposition of the great arteries ( cctga ) , a rare form of congenital heart disease is very difficult . a24-year - old woman was referred to our center at 28 weeks gestationbecause of suspected fetal cardiac anomalies . we report a case of cctga with supraventricular tachycardia , ventricular septal defect , pulmonary valve stenosis , tricuspid valvar displacement and moderate tricuspid regurgitation during her pregnancy . the combined presence of svt and cctga are occasionally present in the literature .

we present a case of foreign body granuloma on the eyelids secondary to cosmetic filler injection on the forehead , which developed several years after the injection . filler migration and the clinical significance and microscopic features of granuloma lesions are also discussed . written informed consent was obtained from the patient and this study was approved by the tri - service general hospital review board ( affiliated to the national defense medical center ) . a 72-year - old taiwanese woman was referred to our oculoplastic clinic , with swelling on both upper eyelids , with multiple painless nonmovable palpable masses , without blepharoptosis , which she had for 4 years . the best - corrected visual acuity of both eyes was 6/8.6 , and intraocular pressure was normal . she did not receive the injection around both eyelids , and there were no other specific problems after the injections . the masses were found below the eyebrow border , sized ~2.5 and ~2.0 cm in diameter in the right and left eyes , respectively , with well - defined , irregular - shaped borders firm on palpation . there was no sign of acute inflammation of the overlying skin ( figure 1a ) . the excised mass was yellowish with an irregular border and had a rubbery consistency ( indicated by arrows in figure 1a and b ) . the size of the mass detected in the subcutaneous region in the right eye was 2 cm 1 cm ( figure 1b and c ) . microscopic examination showed that foreign body granuloma was composed of multiple nodules characterized by basophilic amorphous materials surrounded by an irregular fibrous border ( figure 2a ; hematoxylin and eosin [ h&e ] , 100 ) . the fibrous border was composed of histiocytes , multinucleated giant cells , and fibroblasts ( figure 2b ; h&e , 400 ) . after 1 year postoperative follow - up , the patient recovered well without obvious disfigurement and had only slight scar from the surgery . eyelid problems range from benign , self - resolving processes to malignant , possibly metastatic , tumors . in the past few decades , more and more people have been resorting to facial enhancement with dermal fillers for cosmetic purposes . clinically , dermal fillers are composed of autologous fat , bovine collagen , paraffin , fluid silicone , polytetrafluoroethylene ( teflon ; dupont , wilmington , de , usa ) , and polymer and silicone particles . these fillers have been used to correct soft tissue defects as well as to fill in soft tissue volume around the lips and nasolabial region . for this reason , dermal fillers should have properties of biocompatibility , safety , and stability at the implant site and the abilities to maintain their volume , remain pliable , induce minimal foreign body reactions , and not cause foreign body granuloma.1 autologous fat is a nonallergenic , well - tolerated , supple , versatile implant material . autogenous fat injection ( afi ) into the periorbital or midface region is a common type of cosmetic surgery for rejuvenation in middle - aged and elderly western subjects.2 common complications of facial afi include unrealistic patient expectations , bruising , hematoma , undercorrection , overcorrection , and asymmetry contour of injection site.3 often these adverse reactions result from improper use of products performed by unskilled ( or unlicensed ) practitioners.4 the complication of lipogranuloma formation at the periorbital area away from the afi site is rarely reported.5 it is plausible that the injected fat tissue on the forehead could move down to the periorbital area away from the injection site because of the movement of the frontalis muscle and gravity , as in the case of our patient . furthermore , the fat tissue harvested for afi is usually stored frozen at 20c , but during such cryopreservation , ice crystals form inside the fat cell and many fat cells lose viability . also , if the fat tissue without viability is injected , it can increase the risk for inflammation by foreign body reaction.5 the patient in our report was confirmed to have used the cryopreserved afi for the second time , and we consider this to be the cause of the lipogranuloma . in conclusion , there is evidence present to show that potential complications after tissue injection of autologous fat still exist . as we treat similar cases , we should also be concerned with a possible occurrence of migration . in our study , the patient had palpable and visible masses near the upper eyelid that resulted from afi on the forehead 21 years ago . therefore , tracking patient s detailed history and performing a thorough physical examination are necessarily important . if any unknown mass is detected , excisional biopsy should be immediately conducted by experienced clinicians for diagnosis to avoid severe facial disfigurement afterward .

a 72-year - old taiwanese woman had the history of cosmetic cryopreserved autologous fat injection on her forehead ~21 years ago and was referred to our oculoplastic clinic and presented with multiple painless mass , which she had for 4 years , on both upper eyelids . histopathology confirmed the diagnosis of foreign body granuloma on both upper eyelids . clinicians should be aware of the potential complication of granuloma reaction and migration even many years after the injection . we advise that autologous fat injection should be performed solely by trained physicians and it should be made known that there is a possible occurrence of migration .

cat scratch colon is a rare colonoscopic finding first described by mcdonnell et al.1 in 2007 and is defined as bright , erythematous linear breaks that arise spontaneously and resemble scratches made by a cat . sometimes , bright red linear markings with extravasations of fresh blood are seen in the cecum and ascending colon . cat scratch colon was a rare condition that occurs spontaneously or is partially associated with collagenous colitis.1,2,3 a large volume of air entering the lumen of the colon during colonoscopy was believed to cause cat scratch colon.4 the cecum and ascending colon are the most common regions of occurrence of cat scratch colon . however , we here report a case of ischemic colitis so that we can broaden our experience of this endoscopic finding in the transverse colon . a 65-year - old woman was referred for colonoscopy because of abdominal pain and hematochezia . she had a history of end - stage renal disease , hypertension , and diabetes mellitus . laboratory tests revealed a white blood cell ( wbc ) count of 13,900/mm , a hemoglobin level of 9.4 g / dl , and a platelet count of 228,000/mm . serum biochemistry showed creatinine level , 6.6 mg / dl ; albumin level , 3.0 g / dl ; alanine aminotransferase level , 36 iu / l ; aspartate aminotransferase level , 38 iu / l ; bilirubin level , 1.2 mg / dl ; and alkaline phosphatase level , 198 iu / l with normal electrolytes . negative results were obtained for stool wbc , stool culture , and clostridium difficile toxin a / b . colonoscopy was performed after standard bowel preparation and under sedation with 3 mg of midazolam . the colonoscopic findings showed diffuse bowel wall edema . upon examination of the distal transverse colon computed tomography ( ct ) scan showed moderate - to - severe diffuse bowel wall thickening and focally decreased mucosal enhancement in the right colon . the patient was diagnosed with ischemic colitis based on colonoscopic and ct findings , and clinical manifestations . intravenous fluids were administered to treat dehydration , and total parenteral nutrition with prophylactic antibiotics was provided . after 3 weeks , follow - up colonoscopy and ct were performed . on follow - up colonoscopy , the previous severe ischemic colonoscopic findings had disappeared , and ischemic mucosal change showed further resolution ( fig . " cat scratch colon " was described for the first time in 2007 . over the past few years , very few cases have been reported . these lesions have a prevalence rate of 0.25%.1 cat scratch colon is defined as the presence of linear mucosal breaks in the cecum and ascending colon ; the scratches are of variable length , bright red , resembling a cat scratch . although the cause is unclear , there are several hypotheses : barotrauma from air insufflation into a less compliant colon during colonoscopy,1 complications resulting from collagenous colitis,2,3 tension in the wall of a cylindrical vessel,5 lesions related to diversion colitis,4 chronic cholestasis and its complications,6 chronic anti - inflammatory drug ingestion,7 and others . until recently , recently , colonic perforation due to high air pressure was reported following observation of a cat scratch colon.8 this supports the hypothesis that cat scratch colon is commonly associated with barotrauma etiology . however , barotrauma alone is not the sole cause of cat scratch colon . recently , significant hemorrhagic linear marks have been reported to occur during colonoscopy in patients with collagenous colitis.1,2 variable amounts of chronic inflammatory infiltration is typically present in the lamina propria , with an increased number of intraepithelial lymphocytes . in the case documented above , cat scratch colon was seen in a patient with ischemic colitis . the right colon is predominantly involved in the hemodialysis population.9 hemodialysis tends to cause repeated hypotensive episodes and may thus induce induce vasoconstriction of the vasa recta in the right colon , leading to colonic ischemia.10 ischemic colitis is caused by hypoxemia to the terminal vascular distribution of the intestines . the ischemic process is transient and limited to the mucosa , with fibrosis and formation of colonic strictures in the late phase . chronic ischemic change may play a role in cat scratch colon development owing to decreased compliance similar to collagen colitis . cat scratch colon predominantly occurs in the right colon , where the mucosa is less compliant and more susceptible.8 until recently , cat scratch colon was reported in only the cecum and ascending colon . unlike other cases of cat scratch colon , this patient 's lesion was located in the middle - to - distal transverse colon . in this case , the mucosa of the ascending colon and proximal transverse colon was already edematous , because of which barotrauma may have been an unlikely cause . moreover , the ischemic parts of the colon wall were too stiff ( decreased compliance ) , and pressure during air insufflation may have affected the more susceptible distal ischemic area . in our case , we did not perform a mucosal biopsy and could not exclude collagenous colitis entirely . in conclusion , the presence of cat scratch colon during colonoscopy may suggest barotrauma or other colonic pathology that affects wall compliance . ischemic colitis in this patient may have played a role in the development of cat scratch colon .

" cat scratch colon " is a gross finding characterized by hemorrhagic mucosal scratches on colonoscopy . it is usually associated with a normal colon and is rarely associated with collagenous colitis . in a previous report , cat scratch colon was noted in the cecum and ascending colon , but has also been observed in the distal transverse colon . the patient in this study was also diagnosed with ischemic colitis that may have played a role in the development of cat scratch colon .

laparoscopy is a relatively safe procedure , but complications do occur . a giant parietal wall hematoma is a rare presentation of trocar insertion - related injury . bleeding from insertion sites has been reported and can happen with any procedure . we report a patient who presented with a giant parietal wall hematoma in the post - laparoscopic appendectomy period , who was successfully treated surgically . a 34-year - old woman presented with recurrent right iliac fossa ( rif ) pain since three months . , she underwent laparoscopic appendectomy by a 3-trocar technique one infraumbilical , one suprapubic and the third in rif . the telescope was shifted from an infraumbilical to a suprapubic port for the operative procedure , and instruments were used from the other two ports . the mesoappendix was dissected and separated from the appendix by applying clips proximally and using monopolar cautery distally . the patient was on tablet metronidazole , and she developed severe nausea , vomiting and cough for which she did not seek any treatment . on january , 10 , 1999 , she came back to the hospital with severe weakness , malaise , and pain in the abdomen , with distension of the abdomen . , she had a fever , pallor , pulse rate of 120/min . , and systolic blood pressure of 100 mm hg with minimal ecchymosis over the right lower abdomen . her hemoglobin was 8 gm% , and sonography revealed an anechoic mass in the right side of the abdomen , with a suspicion of an extraperitoneal hematoma , as shown in figure 1 . she was transfused two pints of blood , adequately resuscitated and started on a conservative line of management with analgesics and antibiotics . two weeks later , there was a localized lump in her abdomen , 10 cm 15 cm in size , palpable in the right lower paramedian region . about 120 ml of old blood was aspirated from this lump , after which there was a decrease in its size . the patient was well for a week , when a serosanguinous discharge started oozing from the infraumbilical incision . a ct scan of the abdomen was done that confirmed the presence of a large , right - sided parietal wall hematoma , 10 cm 15 cm 8 cm in size , as shown in figure 2 . the patient was then explored by right paramedian incision on the lump ; the hematoma was located extraperitoneally behind the rectus muscle and evacuated . laparoscopic appendectomy is an accepted procedure , but complications as reported do occur , even with experienced surgeons . vascular complications occur because of sharp dissection during the procedure or during the insertion of a veress needle and trocars . minor and self - limiting bleeding from trocar puncture sites has been reported earlier with an incidence of 0.21% . major vascular injuries do occur and are under reported . until now , 25 cases of major vascular injuries during laparoscopy have been reported . of these , only two have been due to an inferior epigastric artery . in our case , trocar - site injury was related to an inferior epigastric artery by infraumbilical trocar , which may have been inserted a little more obliquely , traveling above the peritoneum ; bleeding occurred below the rectus sheath , which was closed effectively . the patient coughing during the postoperative period may have been one of the factors that initiated the bleeding due to the straining that amounted to loosening of the stitch on the sheath . the injury to epigastric vessels can at times be noticed immediately , either by visualization of a forming hematoma or due to persistent dripping of blood around the trocar site . if the vascular injury is detected , the bleeding can be controlled by placing through - and - through sutures on the sheath along the course of vessels , by coagulation at these sites , or by inflating a foley balloon at the trocar site , in case of minimal bleeding . ultrasonography and ct scan of the abdomen were necessary to correctly diagnose and locate the hematoma . we attempted conservative management of the hematoma by aspiration , as the patient was stable . however , she remained symptomatic with pain and oozing of serosanguinous discharge from the trocar site . finally , surgical evacuation was inevitable . delayed recognition of such vascular injury is associated with significant morbidity and mortality . in both the previously reported cases , hence , delayed , severe presentation with failure of conservative management are the conditions that require prompt surgery toward an effective cure . laparoscopic appendectomy is a safe procedure , one that rarely causes any morbidity and mortality if performed by experienced laparoscopists . however , laparoscopists should be aware of this rare and serious and potentially lethal complication that we have reported .

laparoscopic appendectomy is an established procedure in the treatment of appendicitis . complications of the procedure are related to the veress needle and trocar insertions or pertain to actual operative procedures . trocar - related major bleeding is rare , and , if it occurs , is detected on the table or during the immediate postoperative period . delay in recognition may lead to significant morbidity and mortality . we report a case of giant parietal wall hematoma in a 34-year - old female , presenting one week after discharge from the hospital . the hematoma was completely evacuated by exploration through paramedian incision , followed by an uneventful recovery .

star fruit ( averrhoa carambola ) [ figure 1a and b ] , a member of oxalidaceae family , is popular in many tropical and subtropical countries , and its consumption is high in asia , central america , and tropical west africa . in india , it is recognized as kamrakh in hindi and kordoi in assamese . the plant grows primarily in the northeast , the southern states and along the coast , extending from kerala up to west bengal . it is a star shaped fruit with sweet and sour flavor . traditionally use for treating diabetes , fever and sooth eye infections , throat infections , cough , asthma , colic , diarrhea , food poisoning , and mouth ulcer . dilation of renal tubules , interstitial edema , and an interstitial deposition of oxalate crystals ( d : h and e , 40 ) star fruit toxicity in patients with renal failure is well known . these patients mainly presented as neurological involvement as hiccups , vomiting , mental confusion , seizures , coma , and even death . individuals with normal renal function , who ingest star fruit in a large amount , main symptoms are intractable and persistent hiccups . nephrotoxicity in these people is a rare event , and only few case reports publish worldwide . in this report , we present five patients with normal renal function who developed nephrotoxicity after eating star fruit or drinking star fruit juice . all cases in our report had a history of ingesting star fruit and clinically presented as acute kidney injury ( aki ) . baseline characteristics , the amount of star fruit ingested , clinical presentation and relevant investigation of all patients are summarized in table 1 . two patients ( case 1 and 4 ) consumed fruit on an empty stomach and one patient ( case 2 ) after heavy work in hot weather . patients became symptomatic after 1012 h ( except case 5 ) of eating star fruit . the severity of renal failure was maximum in case 1 ( serum creatinine 23.7 mg / dl ) who ingested highest amount of star fruit [ table 1 ] . there was severe interstitial edema along with inflammatory cell infiltration mainly mononuclear cell . in two patients ( case 2 and 3 ) , few irregular crystals were seen in the interstitium and within the tubular lumen [ figure 1c and d ] . ripe fruits are golden yellow and sweet in taste while unripe fruits are lime green and taste sour , due to its high oxalate content as compared to sweet one . to the best of our knowledge , there is no report published yet about star fruit induced renal failure from the indian subcontinent . though case reports are available on aki due to other fruits as irumban puli ( averrhoa bilimbi ) which belongs to the same oxalidaceae family as star fruit with high oxalate content . , oxalate content of various preparations of star fruit is different [ table 2 ] . ingesting star fruit in large amount can cause secondary oxalosis that presents as nephrocalcinosis , urolithiasis , and renal insufficiency . oxalate content of star fruit from different preparations all patients ingested the fruit or its juice or both . the presentation was gastrointestinal ( gi ) upset within hours after ingestion followed by aki , represented as decreased unit output in an otherwise healthy adult . gi symptoms were not due to uremia as uremic symptom can not develop soon after ingestion and suggest direct corrosive injury of oxalate in the digestive tract . the reason may be due to the difference in initial clinical presentation . in the present case report , patients mainly presented with gi and renal involvement as compared to previous case reports where the neurological symptom was more common as first clinical presentation . all patients had a good outcome with complete recovery of their renal function though a long - term follow - up needed to rule out the risk of development of chronic kidney disease in future . various experimental studies show that plant containing high oxalate content may cause nephrotoxicity in animals like rat . according to these studies , large the mechanism by which tubular damage occurred is the obstruction of renal tubules by these crystals . however , it was observed that tubular dilatation was much more extensive than the crystal deposition , especially in human cases and the focal tubular deposition of crystals were not compatible with the profound loss of glomerular filtration rates . studies found that in addition to the obstructive effect of the crystals , the oxalate alone could act as a cellular toxin capable of producing acute cell damage by increasing apoptotic effect on the renal epithelial cell . oxalate crystals were found in two biopsies , but severe aki changes were present in all patients . those patients who ingest star fruit in a large amount , presents as severe aki , though we still do not know the maximum recommended safe amount of star fruit or juice . consuming large amount of star fruit ( either fruit or juice ) , or even smaller amount on an empty stomach or in a dehydrated state ; should be recognized as a risk factor for star fruit toxicity . in country like india where star fruit consumed routinely , physician should educate peoples even with normal renal function , not to ingest star fruit in large amount , especially empty stomach or in dehydrated state to avoid oxalate nephropathy .

starfruit ( averrhoa carambola ) and its juice are popular in the indian subcontinent as an indigenous medicine . oxalate concentration in this fruit and it 's freshly prepared juice is very high . we present a report of patients presenting with acute kidney injury due to oxalate nephropathy admitted in a single center . all patients had history of ingesting star fruit . patients became symptomatic after 1012 h of eating and main symptoms were pain abdomen and decrease in urine output . three patients needed hemodialysis . all improved with complete renal recovery . taking star fruit in large amount on an empty stomach and in a dehydrated state is a risk factor for nephrotoxicity .

lipoma is a thinly encapsulated benign tumour of mesenchymal origin and is found within parts of the body where fat is normally present . accounting for 0.15% of all benign tumours , lipomas may occur either as single or multiple entities . , lipomas tend to arise mainly in the posterior cervical triangle and forehead , commonly in the fourth to sixth decades of life . on rarer occasions , lipomas may occur in the pharynx , larynx , oral cavity , parotid and submandibular areas [ 1 , 2 ] . comprising 0.64.4% of all parotid tumours , a lipoma of the parotid gland is a rare entity . preoperative evaluation techniques including the use of ultrasound ( us ) , fine needle aspiration cytology ( fnac ) , magnetic resonance imaging ( mri ) and computed tomography ( ct ) have enabled a more accurate assessment of these tumours and a rational approach to their management . the aetiology remains largely idiopathic , but literature suggests a plausible genetic predisposition [ 2 , 4 ] . histologically , lipomas contain fat cells although morphological variants containing other mesenchymal tissues can be present such as angiolipoma and osteolipoma . we present a rare case of a post - traumatic lipoma of the parotid gland , describing the diagnostic workup and surgical removal . a 53-year - old female initially presented to the emergency department following an assault in which she sustained a punch to the left side of her jaw . clinical and radiographical examination revealed a left high condylar head fracture ( fig . 1 ) . on 2-month review , she complained of a persistent , painless , slow growing mass in the left pre - auricular area . clinical examination revealed a soft , mobile , non - tender , non - fluctuant , regular mass within the left parotid gland with normal overlying skin and normal sensation . figure 1:orthopantomographic image showing a left high condylar head fracture and a stable occlusion with adequate posterior contact . orthopantomographic image showing a left high condylar head fracture and a stable occlusion with adequate posterior contact . an us scan showed a well - defined non - vascular hypoechoic lesion in the left superficial parotid lobe measuring 3.2 2.2 2.6 cm ( fig . 2 ) . mri showed high signal intensity on t1-weighted images ( fig . 3 ) and a branching vessel - like central structure traversing the lesion . the tumour comprised of a well - circumscribed , soft yellow , fatty mass covered in a thin fibrous capsule ( fig . 4 ) . histopathology showed a homogenous proliferation of mature adipocytes , confirming the diagnosis of a lipoma . figure 2:ultrasound of the left parotid gland showing a well - circumscribed hypoechoic lesion . figure 3:coronal mri showing the extent of the lipoma and vessel - like structure traversing it ( arrow ) . coronal mri showing the extent of the lipoma and vessel - like structure traversing it ( arrow ) . adipose tissue is normally present in the parotid gland ; however , the incidence of lipomas here is very low . different causes of lipomas are mentioned in the literature : heredity , obesity , diabetes , radiation , endocrine disorders , insulin injection , corticosteroid therapy and trauma . as in this case , a preceding trauma to the soft tissues has been hypothesized to result in a haematoma , with subsequent lymphatic effusion , fat necrosis and lipoma formation . as for their rarity in the parotid gland , the principle consideration of an intraparotid mass is to differentiate between benign and malignant salivary gland neoplasia . there is , however , no unique clinical feature by which the lipoma can be separated from other parotid gland tumours . with a sex ratio of 5 : 1 , the majority of patients present with a single lesion usually without any predisposing causal factors . they progress slowly as soft well - delineated asymptomatic masses where the main patient concern is of the associated cosmetic asymmetry . on average , 75% of the lesions are located within the superficial lobe with a further 6.5% in the deep lobe , and 16.5% occurring in both the deep and superficial lobes . as is standard in the workup of parotid masses , us and fnac provide combined imaging and cytological diagnosis although in lipoma the cytology is often inconclusive . an mri scan is the imaging modality of choice for further assessment prior to any planned surgical excision . it will identify soft tissue margin characteristics and accurately help differentiate between benign and malignant lesions . the surgical approach to a parotid lipoma excision should be the same as for any other suspected benign tumour , with due regard to the presence of the facial nerve in the operative field . it is unusual for the facial nerve to be involved preoperatively ; however , a single case with complete palsy has been reported in the literature . during surgery , the facial nerve is identified and followed up to its peripheral branches as far as necessary for complete tumour dissection . as in this case surgeons and physicians alike should be aware of the possibility of lipomas presenting within the parotid gland , and should recognize its formation in the post - trauma situation and its inclusion in the differential diagnosis of parotid lesions . surgeons should be familiar with its management which is in keeping with the overall workup of any parotid mass .

lipoma of the parotid gland is a rare entity . trauma with soft tissue haematoma formation and subsequent lymphatic effusion , fat necrosis and lipoma formation have been postulated as an aetiological pathway . we report a case of a post - traumatic lipoma of the parotid gland to add to the available literature on this uncommon pathology .

neurofibromatosis type 1 ( nf1 , also known as von recklinghausen disease ) is one of the most common autosomal - dominant genetic disorders with many associated ocular findings / disorders , most typically lisch nodules , retinal / choroidal hamartomas , congenital glaucoma , and optic pathway gliomas [ 1 , 2 , 3 ] . nf1 may also be associated with other various ocular abnormalities [ 1 , 2 , 3 ] including , rarely , retinal ischemia or anterior scleral staphyloma . the staphyloma had been missed during repeated previous examinations by ophthalmologists and was eventually found by the patient himself . a 38-year - old man noticed a lump under his left eyebrow after palpation with his finger . he had been diagnosed with unilateral high myopia ( left eye ) as a child , and was seen yearly by an ophthalmologist . he had no difficultly with vision in the right eye , and therefore did not wear refractive correction . however , this time , investigation of the lump led an ophthalmologist to identify a principal lesion in his left eye . 1a ) ; however , when the top lid was pulled up on downward gaze , a dark brown bulge emerged ( fig . the bulge was 10 mm circumferentially , 7 mm radially , and approximately 4 mm in height , and covered by the extended superior rectus muscle ( fig . the proximal end corresponded to the insertion of the superior rectus muscle , approximately 8 mm away from the limbus ; the distance had probably previously hindered detection of the lesion at examinations . the pupil in the left eye was shifted upward , and the iris ectropion was observed on the upper pupillary border ( fig . iris pattern was absent and multiple subtle protuberances were scattered on the pupillary border without apparent nodules . the diagnosis of equatorial staphyloma was made based on coronal t1-weighted magnetic resonance imaging of the orbit showing dilatation of the vitreous cavity ( fig . visual acuity was 20/15 in the right eye without correction and 20/25 in the left eye with 10.00 spheres and 4.00 80 degrees cylinders , and the intraocular pressure was 15 mm hg in both eyes . his past and family medical histories were unremarkable ; his only ailment was myopia in the left eye , and none of his relatives had a known inheritable disease . however , small neurofibromas and caf au lait spots all over his body led to the diagnosis of nf1 . the staphyloma was adjacent to the cornea , and easily to be found on a 28-year - old woman with neurofibromas , caf au lait spots , scoliosis , and intellectual disability . they did not find a precedent report of staphyloma in nf1 and , thus , mentioned that the association between staphyloma and nf1 might be a coincidence . however , they also speculated that the staphyloma might share a similar pathogenetic mechanism as the absence of the greater wing of the sphenoid or orbital roof reported in nf1 , as regional scleral dysplasia is associated with staphyloma . nf1 is caused by a defect in the nf1 gene located on the long arm of chromosome 17 at position 17q11.2 , that encodes a 2,818-amino acid protein , neurofibromin 1 [ 1 , 2 , 3 ] . neurofibromin 1 is a complicated protein with multiple functions involved in the regulation of cellular proliferation . it works as a tumor suppressor ; thus , its absence leads to abnormal cellular proliferation [ 1 , 2 , 3 ] . besides causing various tumors , nf1 may indicate organ dysplasia [ 1 , 3 ] , although the pathogenesis is not fully explained . chen et al . reported a case of anophthalmia ( absent of the whole globe from the orbit ) in a 27-year - old woman with nf1 . similar to kremer and gaton , they also hypothesized that anophthalmia may have been a coincidence finding . in addition , we admit the possibility of coincidence ; however , we still postulate that both staphyloma and anophthalmia could be caused by eyeball dysplasia , a rate phenotype of nf1 . either way , the previous reports [ 5 , 6 ] and unusual staphyloma in this case epitomized the manifestations associated with nf1 , which are extremely variable and unpredictable [ 1 , 2 , 3 , 4 , 5 , 6 ] .

we report a case of a 38-year - old man who presented with a recently self - detected lump under his left eyebrow . previous ophthalmological history was unremarkable except for unilateral high myopia ( left eye ) since childhood . the appearance of the left eye was seemingly normal ; however , with the top lid pulled up on downward gaze , a dark brown bulge emerged . the bulge was 10 7 mm and approximately 4 mm in height , and was covered by the extended superior rectus muscle . the diagnosis of equatorial staphyloma was made after coronal t1-weighted magnetic resonance imaging of the orbit revealed the dilatation of the vitreous cavity . ocular movements were fully maintained and visual acuity was largely spared : 20/15 in the right eye without correction and 20/25 in the left eye with 10.00 spheres and 4.00 80 degrees cylinders . his past and family histories were unremarkable ; however , small neurofibromas and caf au lait spots all over his body led to the diagnosis of neurofibromatosis type 1 ( nf1 ) . from this case , similar to previous reports , we suggest that manifestations of nf1 are extremely variable and unpredictable .

spinal tuberculosis ( tb ) is one of the oldest human diseases ; it has been found in egyptian mummies dating back 5000 years ; the first case of spinal tb was described by percival pott in 1779.1,2 in the developing countries , spinal tb is one of the primary causes of spinal deformity and paralysis . according to the latest global ( tb ) report by the world health organization , an estimated 9.0 million people developed tb and 1.5 million died from tb in 2013 alone.3 tb affects the spine in 3%5% of patients,4 and spinal tb if patients are diagnosed early , they can be treated with the proper pharmaceutical drugs , such as antibiotics only . early and definite diagnosis of this disease is not easy , as the disease progression is slow and insidious . due to this difficulty , many patients in the early stages of the disease receive treatments , such as nonsteroidal anti - inflammatory drugs , physical therapy , or a corset ( spinal support ) , prior to correct diagnosis.6,7 a positive skin test and elevated erythrocyte sedimentation rate ( esr ) may be useful for diagnosing spinal tb , biopsy evaluation and dna amplification techniques ( polymerase chain reaction ) may also facilitate the diagnosis . for histopathological examination , formalin - fixed and paraffin - embedded tissue blocks of biopsied specimens are stained with hematoxylin eosin.8 conventional radiography methods are used as first - step diagnostic methods , providing a good overview . advanced imaging methods , such as magnetic resonance imaging ( mri ) , play an important role in detecting spinal tb earlier than other techniques and provide the possibility of earlier and more effective treatment before significant defects develop.9 three - dimensional computed tomography ( ct ) scans and mri reveal the involved vertebrae and attachments clearly.10 they can also exhibit the degree of intervertebral disk destruction , the scope of central lesions , the locations and number of cavities and abscesses , and spinal cord compression.11 mri and ct facilitate and allow the diagnosis of spinal tb , but histopathological diagnosis remains essential.12,13 pharmacological therapy usually consists of triple - drug antituberculous chemotherapy ( p - aminosalicylic acid , streptomycin , and isoniazid ) and plays a main role in tb treatment,12 if the lesion is without complications and limited to the vertebrae . some studies14,15 have reported the worsening of existing symptoms or the appearance of new lesions in patients who initially responded well to antituberculous therapy . however , with the appropriate indications , surgery is considered superior for preventing neurological deterioration , maintaining stability , and early recovery.1618 surgical treatment of spinal tb aims at abscess debridement , which involves removing purulent necrotic tissues from normal tissue , spinal cord decompression , permanent spinal stabilization , and preventing or correcting deformity . considering the serious burden caused by the high cost of medical treatment and surgical trauma , early diagnosis and treatment to avoid multivertebral destruction are critical to improve the outcomes in spinal tb.11 spinal tb is one of the most challenging diseases for surgeons to treat , and some guidelines are needed for appropriate decision making and treatment . currently , there are few widely accepted classification systems based on objective data that can guide the selection of the proper treatment approach for patients with spinal tb . in 2008 , oguz et al19 developed a classification system ( gulhane askeri tp akademisi [ gata ] ) for spinal tb based on seven clinical and radiological criteria ( abscess formation , disk degeneration , vertebral collapse , kyphosis [ unnatural curvature of the upper back that creates a hunchback appearance ] , sagittal index , instability , and neurological problems ) . they also recommended specific techniques for each type . per the gata classification system , spinal tb is divided into three types ( ia / b , ii , and iii ) using the abovementioned criteria ( table 1 ) . surgery is recommended for type ib ( abscess formation , one or two level disk degeneration , no collapse , and no neurologic deficit ) , type ii , and type iii patients with or without neurological deficit . analgesic therapy alone can not prevent the extensive destruction of vertebral bone and disk material . after cold abscess and two - level disk degeneration , immediate drainage along with medical therapy can protect the patient from vertebral collapse . as long as the treatment plan is fully prepared , the surgical option can achieve a satisfactory curative effect in treating spinal tb , despite some complications . we believe that the appropriate treatment method should be implemented at the early stage of this disease and that the gata classification system can be considered as a practical guide for spinal tb treatment planning in all countries . current treatment modalities ( medical and surgical ) have improved outcomes in the management of spinal tb , even in patients with neurological deficits and spinal deformities . however , there are various surgical or conservative treatment options , and heterogeneous outcomes have been described , which render the decision - making and selection of specific treatment options by surgeons difficult . future investigations and publications require clear definitions of spinal tb to help guide the appropriate decisions and treatment by surgeons .

spinal tuberculosis ( tb ) is a significant form of tb , causing spinal deformity and paralysis . early diagnosis and treatment are crucial for avoiding multivertebral destruction and are critical for improving outcomes in spinal tb . we believe that appropriate treatment method should be implemented at the early stage of this disease and that the gulhane askeri tp akademisi classification system can be considered a practical guide for spinal tb treatment planning in all countries .

emphysematous pyelonephritis ( epn ) is a life - threatening , fulminant , necrotizing upper urinary tract infection associated with gas within the kidney and/or perinephric space . we present the clinical profiles and outcome of eight patients with epn who were managed conservatively without any mortality . during the period 20082011 , a total of eight diabetic patients with clinical features of epn and computerized tomography ( ct ) image showing gas in renal parenchyma , collecting system or perirenal space , and no fistulous connection between urinary tract and bowel were evaluated . all of them presented with fever , loin pain , dysuria , and oliguria . at admission , renal dysfunction was observed in all these patients with a mean serum creatinine of 5.47 2.3 ( 2.37.8 ) mg / dl . most of them had poorly controlled blood sugar levels ( seven out of eight ) . escherichia coli was found to be the causative organism in all the patients as per urine culture . staging class iv ) and unilateral in 4 ( classes i , ii and iii ) . hydronephrosis was seen in fifty percent of patients ( three unilateral and one bilateral ) . there were air pockets in the perinephric collection , proximal ureter , and in the urinary bladder of two patients ( figures 1 , 2 , 3 , and 4 ) . all patients were discharged in a stable condition with a mean serum creatinine of 2.40 0.76 ( 1.63.3 ) mg / dl . epn is defined as a severe necrotizing renal parenchymal infection that is characterized by the bacterial production of gas within the renal parenchyma . the commonest causative organism is escherichia coli , followed by proteus , klebsiella , anaerobic streptococci , and candida . epn has been reported in renal transplant recipients [ 36 ] and in patients with polycystic kidney disease [ 7 , 8 ] . the diagnosis of epn is often delayed because the appearances of symptoms are very hazy and nonspecific . common presentation includes fever , abdominal pain , dysuria , vomiting , depressed level of consciousness , shock , renal angle tenderness , and acute kidney injury . less common presentation includes dyspnea , crepitus over the flanks , and pneumaturia . in our study , all the patients were diabetics , and majority had uncontrolled blood sugars . all of them presented with fever , loin pain , dysuria , pyuria , and oliguria . dyspnea and pedal edema are seen in 5/8 , and one had altered sensorium . the causative organism was found to be escherichia coli in urine cultures in all the patients . ct imaging not only reveals the presence and position of gas in the renal parenchyma but also is useful to assess the extent of renal parenchymal involvement and response to therapy . abdominal ultrasonography ( usg ) and x - ray can also be used to diagnose epn . in our study , ultrasonography gave a clue for the presence of gas in renal parenchyma in five patients . ct showed the presence of epn , even in those situations where usg could not pick up . it is possible that urinary tract obstruction and decreased renal vascular supply due to diabetes might have contributed for the development of epn in our patients . patients with emphysematous pyelonephritis ( epn ) should be treated with aggressive medical management and possibly prompt surgical intervention [ 1 , 13 , 14 ] . active management with appropriate parenteral antibiotics , addressing the fluid - electrolyte and hemodynamic status , stringent control of diabetes with insulin , and relieving ureteric obstruction either by percutaneous drainage or internal stenting has improved the clinical outcome of our patients without any mortality . none of them required any major surgical intervention , even though more than half of the patients were in class 3 and class 4 epn and having more than two risk factors such as elevated serum creatinine , altered sensorium , and shock . recent case reports have also described successful outcome in patients with bilateral epn with medical therapy alone [ 15 , 16 ] . at discharge , all the patients had elevated serum creatinine ; time to normalization of renal parameters could not be assessed as most of these patients were lost for followup .

emphysematous pyelonephritis ( epn ) is a severe , necrotizing renal parenchymal infection characterized by production of intraparenchymal gas . epn predominantly affects female diabetics and immunocompromised patients . in a three - year period 20082011 , a total of 8 patients were admitted to our hospital . all of them were diabetics , and both males and females were equally affected . these patients showed vague symptoms at admission and frequently presented with fever , loin pain , dysuria , and pyuria necessitating urgent medical attention . epn required radiological diagnosis . ct scan revealed bilateral epn with urinary obstruction and hydronephrosis in 50% of patients . escherichia coli was found to be the causative organism in all the patients . treatment comprised of resuscitation , normalization of serum electrolytes and blood sugars , administration of parenteral antibiotics , and relieving ureteric obstruction if present . all the patients improved with conservative management without any mortality .

secondary implications of malignancies are often more life - threatening than the malignancy itself and need early detection and urgent intervention . we report a similar case of a young gentleman , with a bulky friable middle esophageal mass , which invaded the bronchus , resulting in a broncho - esophageal ( be ) fistula , resulting in lung abscess . this highlights the importance of fdg pet / ct in detecting associated life threatening condition in an oncological setting . he also had recent onset high grade fever with chills . nasogastric tube ( ngt ) upper gastro - intestinal endoscopy ( ugie ) showed friable mass in the middle third of esophagus , biopsy from which showed adenocarcinoma cells . staging fdg pet / ct study maximum intensity projection image showed an hypermetabolic area in mid - thorax [ figure 1a - thick arrow ] with diffuse low grade uptake around it [ figure 1a - thin arrow ] . axial fused pet / ct image [ figure 1b and c- arrow ] showed a large intensely fdg avid soft - tissue mass in the middle third of esophagus , closely abutting the right main and segmental bronchi , measuring 32 mm 32 mm , with maximum standardized uptake value of 27.2 . abutting this mass , a centrally necrotic ill - defined mass , with air pockets within and peripheral low intensity fdg uptake in the thick walls ; was seen in the right lung parenchyma , in the lower lobe [ figure 1b and c - arrow heads ] . minimum intensity projection ( minip ) reformatted sagittal [ figure 2a ] and axial ct [ figure 2b ] images showed a definite communication ( arrow - head ) between the mass ( thick arrow ) and the right lower lobar bronchus ( thin arrow ) . patient , when again asked for any complaints , said to have coughing immediately after swallowing . on collating history and imaging findings and also considering the fact that the patient underwent interventions like ngt placement and ugie , a diagnosis of lung abscess secondary to be fistula was made . ( a ) mip image showing intense fdg uptake in mid - thorax ( thick arrow ) with an area of low grade uptake abutting it ( thin arrow ) , ( b and c ) axial pet / ct images show large middle esophageal mass with bronchial invasion ( arrow ) with a centrally necrotic ill - defined mass adjacent to it ( arrow heads ) sagittal ( a ) and axial ( b ) reformatted ct images in minip window showing definite communication ( arrow head ) between the mass ( thick arrow ) and right lower lobar bronchus ( thin arrow ) they present as bouts of coughing while eating or drinking , known as ohno 's sign and sometimes with recurrent pulmonary infections . difference in density and fdg uptake intensity of the two adjacent lesions on ct and pet images respectively and radiological features of central necrosis with few air - pockets in the lung parenchymal lesion , confirmed it to be an abscess . following interventions , especially in the presence of a large tumor with friable margins in close proximity with bronchi , there is a greater risk of perforation and fistula formation . this is of particular importance in cancers involving middle third of esophagus , where there is close proximity of primary mass to trachea and bronchus . since the entire focus is on tumor staging and management , specific symptoms as seen in our patient however , proper assimilation of history and imaging findings , with use of reconstruction techniques on pet / ct helped us pick up this life threatening condition in a case of esophageal cancer , which led to urgent intervention and also a drastic change in the management of primary malignancy .

sinister undesirable pathologies often accompany malignancies . though the entire emphasis is on cancer management , these benign conditions are more life - threatening than the primary malignancy itself . we report an interesting imaging finding of broncho - esophageal fistula leading to lung abscess on 18f fluoro - deoxy - glucose positron emission tomography / computed tomography ( fdg pet / ct ) in large middle esophageal cancer , which due to early detection , was promptly managed .

leiomyomatosis peritonealis disseminata ( lpd ) is a benign condition , and a very rare disease . it is suspected that this disease originates from a metaplasia of submesothelial multipotent mesenchymal cells . since female gonadal steroids play an important role in the pathogenesis of lpd , it is generally associated with high levels of exogenous and endogenous female gonadal steroids . the definite treatment of lpd is transabdominal hysterectomy and bilateral salpingo oophorectomy ( tah - bso ) . however we here describe a case that in spite of tah - bso and removal of all peritoneal myomatous nodules , developed retroperitoneal fibroma six years later . the patient was a 29-year - old woman who complained of the presence of a mass in her lower abdomen , and a right flank pain for the preceding six months . the pain increased gradually , and the patient referred to hospital . at clinical examination , the patient did nt present abdominal distention . she , however , had a diffuse pain , which was most intensely observed in the right lower quadrant . ultrasonography revealed a large ( 7945 mm ) solid oval shape and well - defined hypoechoic mass in the right adnexal site , which most likely was a residue or recurrence of a previously resected pelvis mass . six years earlier , due to diffuse and progressive abdominal pain she had undergone abdominal ultrasonography , which revealed a semi - solid mass ( 5427 mm ) in the left side of adnexa attached to uterus . the laparotomy revealed a very fragile , largely vascular and multi nodular solid mass , which had originated in posterior part of uterus and extended to peritoneum and retroperitonem . tah - bso were performed because of intractable bleeding following the resection of a retroperitoneal mass . the microscopic pathology findings confirmed the mass as leiomyoma . for more than 5.5 years after the surgery , the patient was doing well with no recurrence of the tumor . however , flank pain and mass sensation started and persisted during hormone replacement therapy since six months ago . she underwent the second operation six months ago , and a retroperitoneal solid mass ( 68 mm ) with an irregular border and a pseudo - capsule was found just adjacent to the external iliac artery . histopathogical examination of the pelvic mass exhibited interlacing bundles of smooth muscle cells without cytological atypia , and a few mitoses ( figure 1 ) . immunohistochemical evaluation was strongly positive for the smooth muscle antigen ( figure 2 ) , progesterone receptors ( figure 3 ) , and estrogen receptors ( figure 4 ) , but was negative for cytokeratin . considering the patient s history , the mass was suggested to be a retroperitoneal fibroma , a remenant of previous disseminated peritoneal leiomyomatosis . the patient was , therefore , scheduled for a long - term follow - up . histological examination of the retroperitoneal mass show bundles of spindle cells with eosinophilic cytoplasm ( h&e ) the immunohistochemical evaluation is strongly positive for smooth muscle actin ( cytoplasmic staining ) . the immunohistochemical evaluation is strongly positive for progesterone receptors(nuclear staining ) the immunohistochemical evaluation is strongly positive for estrogen receptors(nuclear staining ) . lpd is a very rare and benign disease , of which less than 140 cases have been reported . it is characterized by the presence of multiple smooth muscle nodules in the peritoneal surface . some authors believe that female gonadal steroids play an important and primary role in the pathogenesis of leiomyomatosis peritonealis . similar to a number of situations such as pregnancy , extended exposure to oral contraceptives and/or combined hormonal replacement therapy , oestrogen - secreting ovarian fibrothecoma , the disease is generally associated with high levels of exogenous and endogenous female gonadal steroids . since , the disease is seen in post menopausal women and in males , the possible causes of it could be divided into hormonal , subperitoneal mesenchymal stem cells metaplasia , genetic , or iatrogenic after morcellation of myoma during laparoscopic surgery . surgeans and pathologisis face a challenge when dealing with lpd because its macroscopic appearances resembles peritoneal carcinomatosis . while this condition is not common , it has to be taken into account in dealing with a patient with abdominal masses , and especially after a previous myomectomy or hysterectomy . the diagnosis of lpd is made on biopsy specimen , which generally demonstrates a benign smooth muscle abnormal growth originating from the multicentric metaplasia of the peritoneal surface . therefore surgical casteration or gonadotropin releasing hormone agonist seems the appropriate treatment of such conditions . leiomyomas should be considered in the differential diagnoses of intraperitoneal or retropevitoneal masses distinct from the uterus .

leiomyomatosis peritonealis disseminata is a very rare condition characterized by the development of multiple smooth muscle - like nodules in the peritoneal cavity . it is associated with increased serum levels of gonadal steroids . the present report describes a 29-year - old patient underwent transabdominal hysterectomy and bilateral salpingo oophorectomy six years ago because of leiomyomatosis peritonealis disseminata . after six years she referred to us again because of retroperitoneal fibroma , another rare entity , during hormone replacement therapy inspite of lack of uterus and previous castration .

various organs can be affected resulting in skin burn , musculoskeletal injury , myoglobinuria and ventricular fibrillation . the electrical surge may also damage the heart conduction system resulting in various types of heart block . in this case , the survivor of electrocution developed persistent low rate sinus bradycardia , a phenomenon not yet described in the literature . he was holding a wire cable when the main electrical switch box suddenly exploded , throwing him a few feet away . there was second degree burn on both hands and his head hair was completely singed with no loss of consciousness or palpitation . he was brought to the nearest district hospital with normal blood pressure but heart rate of about 35 beats per min ( bpm ) . during the hospitalization , there was no other complication of the electrocution and the burn wound was treated with the appropriate dressing . hence , he was transferred to our center after one week of the index event for further assessment and consideration of pacemaker device implantation . at our center the baseline heart rate was 35 bpm and it rose to 60 bpm during stage 3 of the est , achieving 7.10 mets and 32% of maximal age - predicted heart rate . he could only proceed till stage 3 , limited by physical endurance fatigue . when he tried to ambulate to go to the toilet , he experienced a few episodes of near fainting . after waiting for 3 weeks for his burn wound on the hand to heal , we proceeded to implant a dual chamber rate responsive pacemaker sensia sedr01 ( model sesr01 , medtronic , inc . he had been coming back to our pacemaker clinic for yearly interrogation and we found him to be totally pacing dependent . electrical or electrocution injuries are synonymous with excessive , unintended electrical current exposure and subsequent complications . it can be broadly divided into workplace related or environmental related electrical injuries such as caused by lightning . children and adult blue collar males are prone for accidental electrical injuries as a result of careless and inquisitive nature , and exposure to electrical hazards in the workplace , respectively . the factors influencing electrical injury are the type of current [ direct current ( dc ) or alternating current ( ac ) ] , voltage , resistance and total exposure time . the electrical injury can be direct immediate effect on the body , thermal injury or consequent mechanical injury . any organ or body tissue can be affected , either singly or involving multi - organ . it was unfortunate that this electrician was electrocuted while doing some cable wiring work . as he was exposed to ac , this could have stimulated skeletal muscle tetany , persistently prolonging his grip on the culprit cable wire and further damaging electrical exposure , but he was fortuitously aided by simultaneous switch box explosion which generated enough shock wave to propel the patient away from the electrocution source and break his body from being continuously forming part of the harmful electrical circuit . the short contact time may have explained his limited second degree burn on his exposed hands only and other organs mostly unscathed . not surprisingly , skin burn is a common electrocution complication ranging from 57 - 96% of a postmortem study of 220 fatal cases . skin burn may range from superficial first degree burn to full thickness third degree burn in association with the extent of the skin area involved . for any bystander or first medical staff contact , the first important step is assessment of the airway , breathing and circulation before more detail examination . prompt cardiopulmonary resuscitation ( cpr ) should be initiated if there is any hemodynamic instability and cardiac arrhythmias . the most immediate life threatening arrhythmias are asystole and ventricular fibrillation at the time of electrocution . the patient ought to be hospitalized for further observation as some injuries may only manifest later such as rhabdomyolysis , acute kidney failure , head injury and subtle orthopedic fractures . he should be cared in an acute setting to monitor for recurrence of the malignant arrhythmias and evidence of myocardial injury with cardiac enzymes , electrocardiogram ( ecg ) and telemetry . if there is any occurrence of st elevation myocardial infarction ( stemi ) , the aetiology is most probably coronary vasospasm rather than acute thrombotic occlusion . the electrical conduction system of the heart maybe damaged as well , resulting in other dysrhythmias such as first degree heart block , second degree heart block and bundle branch block . from the autopsy report , focal necrosis was found in the myocardium , as well as the sinus and atrioventricular node ( av ) , thus explaining for the heart blocks phenomenon . this patient had sinus bradycardia with every p wave conducted through the av node , resulting in ventricular response rate of 35 bpm most of the time . no doubt the sinus node was damaged from the electrical injury . however , the constant steady heart rate at 35 bpm invariably suggested some form of autonomic system resetting with higher vagal predominance . surely with this new low baseline heart rate , his daily lifestyle would be severely curtailed . he was still young and his work involved active field work . in this instance , a pacemaker device would definitely benefit him . as he was recovering from his burn injury , the timing of pacemaker implantation was of paramount importance as unhealed or active skin infection may lead to pacemaker infection and its complications . this afforded us the luxury of time to wait for the skin to heal . a dual chamber rate responsive pacemaker was chosen with the dysfunctional sinus node in mind , as well as for optimal av synchrony . it was of no surprise that he was now totally pacing dependent with satisfying achievable activities , demonstrating the fact that the damage on the heart conducting system was permanent .

electrical or electrocution injury is a common accidental occurrence and mostly workplace related . fatal arrhythmias , skin injury and sudden death may ensue . however , it is rare for electrocution to result in permanent low rate sinus bradycardia , incompatible with an active lifestyle . the probable mechanisms for this pathological sinus bradycardia are sinus node dysfunction and autonomic dysfunction with vagal predominance . we describe a young patient who suffered a non fatal electrocution with resultant low rate sinus bradycardia and its successful treatment with a dual chamber rate responsive pacemaker .

orbital and infratemporal fossa ( itf ) metastasis as initial presentation of adenocarcinoma of lung is extremely rare . we report a 46-year - old asian woman , who presented with right eye proptosis and diffuse swelling over right cheek [ figure 1 ] . a contrast enhanced computed tomographic ( cect ) scan of face and neck revealed a solid mass in the right orbit with extension to the itf [ figure 2 ] . histopathologic examination of the biopsy from the mass showed poorly differentiated metastatic adenocarcinoma [ figure 3 ] , which was thyroid transcription factor 1-positive [ figure 4 ] . a total - body cect scan revealed a large heterogeneously enhancing mass lesion in the upper and middle lobe of the right lung [ figure 5 ] . ct guided biopsy from the lung mass revealed poorly differentiated adenocarcinoma [ figure 6 ] . therefore , she was diagnosed as a case of metastatic lung adenocarcinoma ( t3n0m1 , stage iv ) , and planned for palliative chemotherapy with pemetrexed ( 500 mg / m ) plus carboplatin ( auc = 5 ) , iv on day 1 , every 3 weekly . one month after completion of radiation , there was complete reduction of proptosis [ figure 7 ] . but , cect thorax , done after 3 cycles of chemotherapy , revealed clear progression of the primary lung mass . the patient died 5 weeks later as a result of respiratory failure . initial presentation with right eye proptosis and right cheek swelling cect scan of face and neck showing a solid mass in the right orbit with extension to the itf histopathologic study of the orbital mass showing proliferation of adenocarcinoma cells ( h and e , 400 ) the tumor cells were positive for thyroid transcription factor 1 ( 400 ) cect scan of thorax showing large heterogeneously enhancing mass lesion in the upper and middle lobe of the right lung histopathologic study of the ct guided biopsy from the lung mass , showing poorly differentiated adenocarcinoma ( h and e , 100 ) one month post radiation , complete reduction of right eye proptosis

orbital metastasis as initial presentation of adenocarcinoma of lung is an extremely rare phenomenon . here , we report a 46-year - old non - smoker asian woman , who presented with right eye proptosis due to right orbital and infratemporal fossa metastasis , as the first presentation of adenocarcinoma of right lung .

mature digestive cd8 tcell lymphomas are rare entities of variable severity ( from indolent to aggressive and destructive disease ) 1 , 2 , 3 . despite their severity we report here an unusual case of severe mature cd8 tcell lymphoma with digestive infiltration that mimicked crohn 's disease , but with favorable outcome upon oral treatment with cyclophosphamide . a 37yearold male with a history of pancolitis consistent with crohn 's disease was successively treated with azathioprine and antitumor necrosis factor ( tnf)alpha antibodies ( adalimumab and infliximab ) with limited results . he developed persistent buccal ulcerations . examination of the oral cavity highlighted the presence of palatal and mucosal ulcerations with fibrin deposits , gingivitis , periodontitis , osteolysis , and total destruction of the uvula ( fig . the patient was treated with mesalamine , methotrexate , and ustekinumab ( antiinterleukin 12 antibody ) , without longlasting effects . diarrhea and oral ulcerations progressively worsened , leading to a weight loss of 8 kg . multiple ulcerations , consistent with pancolitis , were found mainly in the right colon and cecum by endoscopic exploration of the digestive tract . histopathological analysis of the palatal ulcerations showed a diffuse mucosal lymphoid infiltration made of small to mediumsized lymphoid cells ( fig . conversely , they were negative for cd56 , epstein barrencoded small rnas ( ebers ) , and cd30 . the proliferation index , assessed by ki67 staining , was low to moderate ( not shown ) . review of historical colonic biopsies showed the same lymphoid mucosal infiltration with superficial ulcerations without crypt distortion and absence of granuloma . clonal cd8 tcell expansion was assessed by tcell receptor locus rearrangement analysis and was detected in lymphoid cells from the oral cavity and colon ( fig . 1f ) , but not in blood . a computerized tomodensitometry of thorax and abdomen found no additional organ involvement . no somatic stat3 exon 21 mutation was detected in this lymphoproliferative malignancy with expansion of mature and cd8 lymphoid cells 4 . palate biopsy : dense lymphoid infiltrate beneath the normal epithelium and ulcerations with large amount of necrosis within the lymphoid infiltrate ( hematoxylin eosin [ he ] staining ; scale bar 700 ) ( b ) . detail of the infiltrate : small to mediumsized lymphoid cells ( he staining ; scale bar 60 ) ( c ) . immunohistochemistry : lymphoid cells were positive for cd3 ( d ) and cd8 ( e ) ( scale bar 150 ) . tcell receptor locus rearrangement analysis of colon lymphoid cells showing that the cd8 tcell expansion was clonal ( f ) . on the basis of the diagnosis of mature cd8 tcell lymphoproliferative disorder , oral treatment with 150 mg cyclophosphamide per day was started . after 4 months , the patient had already recovered his baseline weight and the oral cavity lesions had completely healed ( fig . we report a very unusual case of mature cd8 tcell lymphoma with digestive infiltration that mimics crohn 's disease . our finding suggests that in patients with atypical features of crohn 's disease , including severe oral ulcerations , and who are refractory to standard treatment , the alternative diagnosis of clonal cd8 tcell lymphoproliferative disorder should be considered . its prognosis can be remarkably favorable upon oral treatment with cyclophosphamide , as observed for our patient .

key clinical messagein patients with atypical crohn 's disease features , including severe oral ulcerations and resistance to standard treatment , the possibility of a mature clonal cd8 + tcell lymphoproliferative disorder should be investigated . clinicians should be aware of this differential diagnosis because cd8 + tcell lymphoma prognosis can be remarkably favorable upon oral treatment with cyclophosphamide .

a 2-month - old girl was brought to us with haziness of both eyes for the past 20 days . she was a full - term baby delivered by cesarean section with a birth weight of 2.75 kg . the parents denied any history of trauma and the child was otherwise healthy . on examination ( under general anesthesia ) , she was noted to have conjunctival hyperemia , increased corneal diameter ( 13.5 mm od , 11 mm os ) , corneal haze with small blood clots in the anterior chamber of both eyes . intraocular pressure ( iop ) was noted to be 20 mm hg od and 8 mm hg os by perkin 's tonometer . a few yellowish - orange skin nodules were noted on the forehead and neck [ fig . 2 ] , that progressively increased on follow - up . remainder of her systemic and neurological examination was normal . excision biopsy of a forehead skin lesion was performed under general anesthesia and histopathological examination of the specimen showed non - langerhans histiocytic proliferation [ fig . 3 ] . immunohistochemistry was negative for cd1a and cytokeratin ; positive for cd68 . with the above clinical picture and laboratory finding left , corneal edema right eye ; top right : hyphema after resolution of corneal edema left eye ; bottom left : development of cataract ; bottom right : after cataract extraction resolution of skin lesion over the period of 1 year histopathology of skin lesion . low power ( 100 ) : structure of acanthotic skin with sub epidermal infiltration of xanthomatous cells ( arrow ) and inflammatory cells with encircling adnexal structure and extending into subcutaneous zone . high power ( 1000 ) : sheets of foamy histiocytes ( arrow ) with interspersed multinucleated giant cells ( block arrow ) she was started on a topical antibiotic steroid combination , cycloplegic and dorzalamide ou . on her review visits , she developed posterior synechiae and a few iris nodules in the left eye , which resolved with intensive topical steroids . she maintained a steady central fixation throughout her treatment and was also on regular review with a pediatrician . four months after her first presentation , she developed bilateral anterior subcapsular cataract , which gradually progressed . topical prednisolone acetate 4 times a day was added to topical antibiotics a day before surgery . after peritomy , the bleeders were cauterized and a limbal groove made with a blade breaker . the anterior chamber was entered by deepening the groove and trypan blue was injected under an air bubble . the anterior capsule was punctured by a bent 26 g needle and capsulorhexis done with a utrata forceps . the bottle height was raised to increase pressure in the anterior chamber and tamponade any bleeding . subsequently , the posterior capsule was punctured by a bent 26 g needle in a sideways fashion and capsulorhexis completed by an utrata forceps . an intraocular lens ( iol ) was not implanted given the age of the patient ( < 1 year ) . after surgery , she was started on topical steroid antibiotic combination and cycloplegics , which were tapered over 6 weeks . the right eye cataract surgery was done 6 weeks after the left eye surgery and a similar procedure was followed . 1 year after surgery , the child was comfortable with aphakic correction and the visual axis was clear . the iop was noted to be normal on all occasions and no antiglaucoma medications were needed . touton 's giant cells were absent in our patient ; however , she had features suggestive of a non - langerhan 's cell histiocytosis as suggested by the absence of a cd1a and cytokeratin . ocular involvement in jxg is in the form of diffuse or discrete vascular iris nodules , which may bleed , resulting in spontaneous hyphema and secondary glaucoma . the condition may also involve the ciliary body , anterior choroid , cornea , lids , limbus , orbit , retina , optic nerve and optic nerve , but intraocular involvement is rare ( < 1% of cases ) . indeed jxg should be included in the differential diagnosis of spontaneous hyphema in this age group and one should make a careful evaluation to detect skin nodules , which can be difficult to detect early in dark skinned individuals . this could be because patient was taken up for surgery once the iris nodules and hyphema had resolved and patient was continued on topical steroids . it is likely that the use of intracameral adrenaline , raising the bottle height and a tight wound closure reduced the risk of bleeding in our patient . in general , bimanual anterior limbal vitrectomy is preferred over a pars plana vitrectomy after primary posterior capsulotomy in pediatric cataract surgery . this is because the technique is simpler and most cataract surgeons are comfortable with an anterior approach . an iol was not implanted in our patient considering the anterior segment inflammation and the bilaterality . furthermore , the safety and benefits of iol implantation in infancy is not well - established . management as detailed above may reduce the risk of bleeding and improve the visual outcome .

there is limited literature on the management of cataracts in juvenile xanthogranuloma ( jxg ) . a 2-month - old girl presented to us with hyphema , secondary glaucoma ou and skin nodules suggestive of jxg . she developed bilateral cataracts during her follow - up and was treated successfully with cataract surgery and aphakic rehabilitation .

myxedema coma is a rare life - threatening complication of long - standing , neglected hypothyroidism . till date we are reporting the case of an elderly lady with life - threatening myxedema coma and septicemic shock who was successfully treated with oral thyroxine . a 61-year - old lady , known case of hypothyroidism for 20 years , was admitted in surgical emergency with complaints of abdominal pain and distension for 3 days . her pulse rate ( pr ) was 68/min . , blood pressure ( bp ) 140/86 mmhg , temperature 36.5c , respiratory rate ( rr ) 10/min . her investigations were hb -8.8 gm% , total leucocyte count-12,400/mm , serum na-112 meq / l ( normal range 135 - 145 meq / l ) , serum k-2.2 meq / l ( normal range 3.5 - 5.5 her liver function tests , kidney function tests and chest x - ray were normal and ecg showed low voltage complexes . abdominal ultrasound revealed a mass in right iliac fossa suggesting appendicular lump along with paralytic ileus . meanwhile her tft showed severe hypothyroidism with t3 being 62 ng / dl ( normal range 100 - 180 ng/ dl ) , t4 - 2.2 g / dl ( normal range 4 - 12 g / dl ) and tsh-18 u/ ml ( normal range 0.5 - 6 u / ml ) . her dose of eltroxin was elevated to 100 g / day . her abdominal distension increased over the next 48 hours and a contrast - enhanced ct scan of abdomen revealed intestinal perforation . general anesthesia was induced using rapid sequence induction . immediately after induction , she had systolic hypotension ( 85 mmhg ) , managed with fast crystalloids . there was a further drop in her blood pressure after the abdomen was opened during surgery , necessitating dopamine infusion along with noradrenaline infusion . surgery lasted for 2 hours and she was shifted to icu for elective ventilation when received in icu , patient was hypothermic ( temperature 34.6c ) and hypotensive ( systolic bp-68 mmhg ) . rewarming was started with of warm fluids , bladder lavage with warm saline and electric blanket . three litres of lactated ringer 's solution was pushed intravenously . adrenaline infusion was now added . unfortunately parental thyroxine was not available so we decided to give eltroxine tablet ( 500 g ) once daily via ryle 's tube . this along with aggressive antibiotic coverage , steroid supplementation and inotropic support gradually improved her blood pressure over the next 24 hours . her eltroxine was titrated to 150 g twice a day now . over the next 2 days finally on postoperative day 5 , she was extubated and shifted to the postoperative ward . myxedema coma is an extreme complication of hypothyroidism in which patients exhibit multiple organ abnormalities and progressive mental deterioration . it occurs almost exclusively in age group 60 years and above , with 80% preponderance in females , as seen in our patient . more than 80% of cases of myxedema coma occur in winters , probably due to age - related loss of ability to sense temperature and lower production secondary to hypothyroidism . other events which can precipitate it include infections , cardiovascular accidents , congestive cardiac failure and certain drugs . we presume that in our patient the precipitating factors included winter season , septicemia and stress of surgery and anesthesia . myxedema coma causes drastic decrease in metabolic rate , hypoventilation , hypotension , hypothermia , decreased mental state progressing to coma and decreased cardiac output . a common misconception is that a patient must be comatosed to be diagnosed with myxedema coma . although we could not get cortisol level determined , empirical steroid supplementation aided in the recovery of our patient . even with optimum therapy , mortality rate as high as 30 - 60% has been reported . did a study to determine the various predictors of outcome in myxedema coma and concluded that various factors associated with increased mortality included bradycardia , need for mechanical ventilation , hypothermia , sepsis , hypotension and intake of sedative drugs . in our patient most of these factors were present but were managed aggressively and judiciously . in literature , intravenous thyroid hormone supplementation has been advised for the treatment of myxedema coma but since it was not available , early supplementation of a higher dose ( 500 g / day ) of oral thyroxine was started via the ryle 's tube . the good outcome of our patient despite severe hypothyroidism and septicemic shock with gangrene occurred most probably due to the fact that she was already receiving eltroxine , though suboptimal dose and our aggressive inotrope therapy , fluid management and multiple antibiotics . to conclude , the combination of myxedema coma and sepsis in a patient can be lethal . early institution of thyroxine supplementation , even via oral route , along with inotropic support and steroid supplementation , can improve the prognosis .

myxedema coma is a life - threatening but uncommon complication of long - standing , neglected hypothyroidism . it was first reported by ord in 1879 . till date only around 200 cases have been reported in literature . the incidence in european countries is 0.22 per million per year . no epidemiological data is available from the indian subcontinent . we are reporting the case of an elderly lady who went into life - threatening myxedema coma along with septicemic shock , and was successfully treated with oral thyroxine .

to report the successful outcome of a rare optic pit - associated maculopathy with an outer retinal hole following 23 g vitrectomy , internal limiting membrane ( ilm ) peeling and fluid - gas exchange without additional endolaser . this case report documents a 56-year - old male patient with complaints of progressive diminution of vision in the right eye more than in the left eye due to an optic disc pit with an outer retinal hole and a cataract . the case report shows the successful outcome of a rare optic pit - associated maculopathy with an outer retinal hole and a cataract following phacoemulsification with 23 g vitrectomy , ilm peeling and fluid - gas exchange without additional endolaser . optic pits occur equally between men and women . they are seen in roughly 1 in 10,000 eyes , and approximately 85% of optic pits optic pits have been associated with serous retinal detachments [ 3 , 4 ] in up to as many as 50% of all cases . imamura et al . reported an outer layer hole in only 27% of eyes with retinal detachment associated with optic disc maculopathy . the detachments associated with optic pits may occur at any age but most frequently present in early adulthood . the most popular theory behind this association is a separation of the layers of the retina , known as retinoschisis , due to fluid ( the vitreous humor ) entering the optic pit and traveling between the inner and outer layers of the retina . we hereby present a case report showing successful management of a rare optic disc pit anomaly with an outer retinal hole and a cataract , which was treated with phacoemulsification vitrectomy with gas tamponade but without endolaser . marked improvement in visual acuity corresponded to the resolution of the outer retinal hole observed clinically as well as on optical coherence tomography ( oct ) . a 56-year - old male presented with a 6-month history of painless , progressive diminution of vision in the right eye more than in the left eye . his past medical history and ocular history were not significant , and there was no history of trauma or surgery . ophthalmic examination showed the best - corrected visual acuity of counting fingers 2 meters in the right eye and 20/40 in the left eye . on anterior segment examination , both pupils reacted normally to light , whereas slit - lamp biomicroscopy revealed grade ii nuclear sclerosis in both eyes dilated fundus examination of the right eye revealed an oval , gray - yellow crater - like depression on the temporal aspect of the optic disc , suggestive of an optic pit ( fig . 1 ) . in the macular area adjoining the disc there was a serous elevation of around 4 disc diameters with central thinning of the retinal layers , giving the impression of an outer retinal hole . oct was done and confirmed the presence of an optic pit with maculopathy and an outer retinal hole ( fig . 2 ) . fundus fluorescein angiography showed a window defect corresponding to the outer retinal hole in addition to pooling of the dye in the late venous phase , suggestive of serous retinal detachment ( fig . 3 ) . pros and cons of various treatment options including no treatment were discussed . after obtaining informed consent , the patient underwent phacoemulsification with intraocular lens implantation with 23 g pars plana vitrectomy with sf6 gas tamponade without additional endolaser . the vitreous was stained with triamcinolone 0.1 ml , and the induction of posterior vitreous detachment was achieved with the vitreous cutter . after completing the vitrectomy , internal limiting membrane ( ilm ) peeling the patient was instructed to maintain a face - down position for 1 week and was followed up closely . after 2 weeks , the gas bubble was completely absorbed , and vision had improved to 20/60 . the oct showed no evidence of serous retinal detachment or retinoschisis , and the outer retinal break was sealed ( fig . the patient was followed up for the next 2 years on a regular basis and there was no evidence of recurrence . this case report documents the successful outcome of a rare optic pit - associated maculopathy with an outer retinal hole and a cataract following phacoemulsification 23 g vitrectomy , ilm peeling and fluid - gas exchange without additional endolaser . imamura et al . reported an outer layer hole in only 27% of eyes with retinal detachment associated with optic disc maculopathy . however , a relatively high incidence of recurrence has been reported after laser treatment alone ; laser treatment also results in temporal scotoma to the patient . hirakata et al . reported the success of vitrectomy with induction of posterior vitreous detachment and gas tamponade , without additional laser treatment in reattaching the macula , and improvement in central vision in most patients with optic disc pit maculopathy . in our patient , his visual acuity remained at 20/60 at the end of 2 years of follow - up . the limited recovery in visual function is probably related to the longstanding nature of the condition . vitrectomy with ilm peeling and gas tamponade without any additional laser photocoagulation may be sufficient for the treatment of optic disc pit maculopathy even with an outer retinal hole . although further studies are required to evaluate the above findings , documenting large series is challenging due to the rarity of cases with optic disc maculopathy with outer retinal holes . none of the authors has a financial or proprietary interest in any material or method mentioned .

objectiveto report the successful outcome of a rare optic pit - associated maculopathy with an outer retinal hole following 23 g vitrectomy , internal limiting membrane ( ilm ) peeling and fluid - gas exchange without additional endolaser.methodinterventional case report.resultsthis case report documents a 56-year - old male patient with complaints of progressive diminution of vision in the right eye more than in the left eye due to an optic disc pit with an outer retinal hole and a cataract . optical coherence tomography confirmed the presence of an outer retinal hole . the case report shows the successful outcome of a rare optic pit - associated maculopathy with an outer retinal hole and a cataract following phacoemulsification with 23 g vitrectomy , ilm peeling and fluid - gas exchange without additional endolaser .

an unusual clinical presentation of cow 's milk allergy ( cma ) in an infant gave us the opportunity to observe a very fast and intriguing decrease of ige concentrations after allergen withdrawal , raising new questions about ige production and metabolism in newborns and infants . a 17-day - old neonate , born to atopic parents at full term , was hospitalized in the children hospital of tours ( france ) for poor feeding and increasing diarrhoea for 4 days , associated with a severe metabolic acidosis , after a symptom - free interval of 2 weeks . erythema and a pustular rash of the face as well as a gloves and socks- type skin rash were noted at 18 days of age , leading to the finding of very high concentrations of total ige ( 1298 kiu / l ) and ige specific to cow 's milk ( cm ) ( 83 kau / l , phadia ) ( figure 1 ) . first - stage formula milk had been introduced on day 11 in addition to breastfeeding according to the mother , although it is possible that this had been given since birth . although the clinical presentation was very unusual and severe [ 1 , 2 ] , the diagnosis of cma was confirmed by a rapid regression of all symptoms after withdrawal of cm proteins . seven days later , total ige fell to 121 kiu / l and specific ige to 1.44 kau / l , and total ige returned to within the normal range ( 10.5 kiu / l ) ( figure 1 ) five days later . the ige detected in the newborn had been self - produced since ige did not cross the placenta and his mother had no cm - specific ige ( not shown ) . this ige immune response thus probably reflected a rapid maturation of ige+ b cells into plasmablasts , as recently evidenced in mice . no less spectacular was the 100-fold decrease in ige concentrations 12 days later ( figure 1 ) , following exclusion of any cm lysate from the diet . high levels of both specific and total ige are regularly observed in infant cow 's milk allergy and are reminiscent of experimental models where their coexistence reflects a genetic ( atopic ) background . however , their parallel evolution , and the fact that they both appeared to be strictly antigen dependent , could also suggest that the ige measured in the total assay is mostly directed against cm . pharmacokinetic modelling techniques were used to describe the elimination kinetics of total ige in this infant . the kinetics model was ln ( c(t ) ) = ln ( cn ) ke t , where c(t ) is the ige concentration over time ( kiu / l ) , cn is the estimated ige concentration just after cm withdrawal , ke ( day ) is the first - order elimination constant , and t ( day ) is time . the ke parameter was estimated using log - linear regression . the elimination half - life ( t1/2 ) was calculated as t1/2 = ln ( 2)/ke and was 1.7 days in this infant . the combined kinetics of ige and omalizumab , an anti - ige humanized monoclonal antibody , were previously described in adults using pk - pd modelling . the parameters describing the interindividual distribution of ige elimination kinetics in this study were used to estimate the confidence interval containing 90% of t1/2 values although low , the t1/2 of ige of our infant was within this ci90t1/2 and thus has to be considered as however , to calculate this elimination kinetics , it was postulated that ige production ceased after cm withdrawal , which is not necessarily the case . it is therefore possible that the true ige t1/2 of this patient may be shorter than 1.7 days . nevertheless , our findings suggest that the ige production stopped rapidly if not immediately after allergen elimination , suggesting that a very limited ige plasmablast life span combined with the very short ige half - life . whether this hypothetical ige plasmablast short life span is restricted to ige or whether it is due a more general immaturity of lymphoid niches in the newborn , as suggested in murine models , warrants further investigations in a cohort of cm allergic newborns .

we report a case of a very fast and intriguing decrease in ige concentrations after exclusion from the diet of any cm lysate in an unusual clinical presentation of cow 's milk allergy in an infant . analysis of ige kinetics after allergen elimination suggests rapid cessation of ige biosynthesis and a short ige half - life .

the success of root canal therapy is dependent upon a thorough knowledge of the root and root canal morphology . this helps us to locate all the canals and properly clean , shape , and obturate the canal spaces in all dimensions.[13 ] slowey has suggested that mandibular first premolars , often called as endodontist 's enigma , may present the greatest difficulty of all teeth to perform successful endodontic treatment . this is because they are anatomically unpredictable , and often present with a wide variety of morphological rarities . one such morphological oddity is the presence of two roots , with a reported incidence of 1.8% . this article reports on the clinical case of a mandibular first premolar with two roots . a 32 year old male patient reported to the department of conservative dentistry and endodontics at rajasthan dental college and hospital , with the chief complaint of pain in the posterior right mandibular tooth for the past 1 week . radiographic examination of the tooth indicated an unusual anatomy of two roots , and also there was widening of the apical periodontium , indicating periapical pathology and the necessity for root canal treatment [ figure 1 ] . pre - operative intra oral periapical radiograph of the mandibular right first premolar in the patient in the case study , showing an unusual anatomy of two roots , and widening of the apical periodontium the clinical examination , radiographic examination and vitality tests led to a diagnosis of acute apical periodontitis of the right mandibular first premolar requiring endodontic therapy . the tooth was anaesthetized by way of right inferior alveolar nerve block using a 2% solution of lignocaine hydrochloride containing 1:80000 adrenaline ( lignox 2% a , warren , indoco ) . endodontic access was prepared with a round diamond bur in a high speed airotor handpiece . the pulp chamber was inspected with the aid of a magnifying loupe ( seiler loupes ) and a sharp dg 16 explorer was used to locate the canal orifice . after obtaining the canal patency , a # 10 k file ( dentsply , maillefer ) was precurved and inserted in a distolingual direction to traverse the canal bifurcation into the second root [ figure 2 ] . a working length radiograph confirmed the presence of a single coronal canal bifurcating in the middle one third , and coinciding with the separation of the two roots . the two canals exited in separate apical foramina located in the respective roots [ figure 3 ] . cleaning and shaping of the canals was performed using crown - down technique under copious irrigation with 5.25% sodium hypochlorite solution . after three days , the canals were obturated with cold , lateral compaction of gutta percha cones ( dentsply ) and zinc oxide eugenol sealer . a post obturation radiograph was taken to evaluate the quality of obturation [ figure 4 ] . files inserted to traverse and confirm the root canal configuration working length radiograph of the mandibular right first premolar in the patient in the case study , showing the presence of a single coronal canal bifurcating in the middle one third and coinciding with the separation of the two roots . the canals exited in separate apical foramina in the respective root post - operative radiograph of the mandibular right first premolar in the patient in the case study anatomical variations , especially extra canals and roots , should always be kept in mind when treating teeth endodontically . canals if left unclean may harbour microorganisms , which have been reported to be a major cause for treatment failure . a study at the university of washington assessed the failure rate of non surgical root canal therapy in all teeth . the root morphology of mandibular first premolar can be highly complex and extra root(s ) can be found . scott and turner describe the accessory root of mandibular first premolar as tome 's root . they observed ethnic differences in the root morphology ; and , reported the highest incidence ( > 25% ) of accessory roots in the australian and sub saharan african populations . the lowest incidence of tome 's root ( 0 - 10% ) occurred in the american , arctic , new guinea , jomon and western eurasian populations . sert and bayrili also reported sex differences in canal morphology , reporting higher incidence ( 44% ) of accessory roots and canals in females as compared to males ( 34% ) . thus , a variety of factors contribute to variations in root anatomy of mandibular first premolars . successful endodontic outcome in such cases is dependent upon careful use of all the available diagnostic aids to locate and treat the entire root canal system . careful interpretation of angled radiographs , proper access preparation and a detailed exploration of the tooth are essential prerequisites for a successful treatment outcome .

thorough knowledge of the root canal morphology , appropriate assessment of the pulp chamber floor , and critical interpretation of radiographs are a prerequisite for successful root canal therapy . the possibility of additional root / canal should be considered even in teeth with a low frequency of abnormal root canal anatomy . this article reports on a case of mandibular first premolar with two roots , which was successfully treated with root canal therapy .

isolated internal iliac artery aneurysms are very rare , accounting for around 0.4% of abdominal aneurismal disease ( 1 ) . with a rupture rate of 38% and associated high mortality , early detection and intervention signs and symptoms at presentation are often nonspecific so a high index of clinical suspicion is required . timely intervention in cases of isolated internal iliac artery aneurysm has the potential to improve survival . an 84 year old , retired electrician , presented to the emergency department with reduced urine output and lower abdominal pain . he reported a three day history of urinary hesitancy and increased frequency , associated with generalised lower abdominal pain radiating to the testes . his medical history included transurethral resection of the prostate in 2005 , inguinal hernia repair and hypertension . the patient lived alone and was an ex - smoker . on examination he appeared well , with stable observations . he was noted to have lower abdominal tenderness , without being peritonitic , and had a palpable bladder . a provisional working diagnosis of urinary retention was therefore made , with no cause identified . axial view of ct abdomen - pelvis showing right internal iliac artery aneurysm with a contained pre - sacral leak ( as indicated by arrow ) the patient deteriorated the following day , becoming hypotensive and anuric , with an increasingly tender abdomen . after initial resuscitation , an urgent ct scan of the chest , abdomen and pelvis showed an 8.5 cm aneurysm of the right internal iliac artery . there appeared to be a contained leak from the aneurysm in the pre - sacral region ( fig . a moderate amount of intraperitoneal free fluid was noted . a right sided hydronephrosis with associated right hydroureter was also noted . an urgent transfer to a vascular facility was arranged and a same day endovascular repair of the aneurysm was performed . prompt identification of the underlying cause is then essential in directing further management . in elderly men causative factors can be grouped into : obstructive , neurogenic and detrusor inactivity . benign prostatic hyperplasia is the most common individual cause but often there are multiple underlying factors ( 2 ) . careful history and examination , combined with the use of specific diagnostic testing will usually establish a cause , and therefore result in the identification of high risk patients ( 3 ) . iliac artery aneurysms account for an estimated 2% of intra - abdominal aneurysmal disease , with a quarter of these primarily affecting the internal iliac artery . typically presentation is late , with an estimated 40% rupturing and a mortality of over 50% in those cases . identifying patients with internal iliac artery aneurysms of any size is challenging , because they often remain asymptomatic . patients may present with non specific symptoms of abdominal pain and tenderness , or symptoms and signs of local compression . this case demonstrates how a rare and potentially life threatening aneurysm manifested as a relatively common presentation abdominal pain and urinary retention . it is likely that the patient s urinary symptoms were caused by local compression exerted by the aneurysm and its subsequent rupture . a triad of symptoms abdominal pain , urinary retention and haemodynamic instability , prompted the clinician to request urgent imaging thus allowing early intervention and a good outcome . the importance of considering a broad range of differential diagnoses is clearly demonstrated particularly when more common causes are less likely . this patient had already undergone a turp making a prostatic cause of retention less probable . aneurysmal dilation and rupture ought to be among any clinicians differential diagnosis for urinary retention , especially in the context of progressive symptoms in an acutely deteriorating , unstable patient . there are a number of case reports on the various different presentations of iliac artery aneuryms : lumbrosacral plexopathy , bowel obstruction and a case of a patient presenting with cullen s sign . these varied presentations collectively demonstrate the difficultly in swift diagnosis of an internal iliac artery aneurysm with potential associated rupture ( 5 - 8 ) . the presenting problem in this case report and of those reviewed remains consistent urinary retention and abdominal discomfort . haemodynamic instability , as seen in this case , is another indicator that the underlying diagnosis may be vascular in origin . we propose that any patient presenting acutely with urinary retention , abdominal pain and haemodynamic instability should qualify for an urgent ct abdomen in order to rule out an iliac artery aneurysm . this case emphasises the importance of maintaining a broad and open mind in approach to the management of urinary retention associated with other symptoms , and reacting quickly to clinical developments .

internal iliac artery aneurysms are rare , but with a mortality approaching 50% in those that rupture , prompt diagnosis is essential . often presentation is nonspecific ; a wide variety of symptoms and signs have been encountered , illustrating a challenge in identification . we report a case of ruptured internal iliac artery aneurysm presenting as urinary retention . the importance of maintaining a broad differential in cases of urinary retention is clearly highlighted .

toxic epidermal necrolysis ( ten ) is a severe and life - threatening condition involving skin and mucous membrane . the drugs most commonly causing ten are sulfonamides , penicillins , other antibiotics , non - steroidal anti - inflammatory drugs , anti convulsants , etc . these drugs reaction are more common in individuals having human immunodeficiency virus ( hiv ) infection / aids than the general population . fluconazole , an antifungal drug of the azole group , is frequently used for management of candidial infections , coccidiodal meningitis , cryptococcal meningitis , and in empiric treatment of critically ill hiv - infected patients . the usual side effects of fluconazole mentioned in the literature are mild like nausea , vomiting , headache , elevation of hepatic transaminases , etc . ten / stevens - johnson syndrome ( sjs ) caused by fluconazole in immunesuppressed patients is very rare and reported in only three reports previously.[46 ] we present an additional case of this type in view of lack of such reports in the indian literature . a 28 year old male patient , taking anti - tuberculosis treatment for last 3 months for pulmonary tuberculosis , now presented with oral thrush . he was not married but gave history of multiple unprotected roadside sexes . on examination , there was pallor and extensive oral thrush over tongue extending up to tonsil , and posterior pharyngeal wall . other systemic examination revealed no abnormality . in view of high risk behavior and the presence of oral thrush , his sera were subjected to hiv1 and hiv2 testing that turned out to be reactive . his other investigations revealed hemoglobin 8 g% with normal leukocyte count , differential count , fasting blood sugar , liver function test , renal function test , etc . a skiagram chest showed regression of pulmonary infiltrates in comparison to previous chest x - ray . the antituberculosis treatment was continued and oral fluconazole 150 mg daily was started with povidone iodine gargles . on the second day of fluconazole therapy , the patient developed generalized body ache along with pruritic rash over face , trunk , and extremities . the rashes first appeared over the trunk and then spread to involve extremities and face . the lesions subsequently ruptured with sloughing of large sheets of skin , leaving behind erythematous areas involving more than 70% of the body surface area . based on clinical course following fluconazole therapy , a diagnosis of fluconazole - induced ten ( naranjo score 6 ) was made . laboratory investigations this time revealed normal total leukocyte counts , but slight elevation of liver enzyme ( alt50 iu / l , ast64 iu / l ) . the patient was managed conservatively with local soothing agents , analgesics , azithromycin , antihistaminics , and eye care with adequate parenteral hydration . the patient improved with above therapy . ten and sjs are acute and life - threatening disorder of unclear pathophysiology , characterized by epidermal necrosis , erosions of mucous membrane , and detachment of epidermis with constitutional symptoms . cases having less than 10% of epidermis involvement are designated as sjs , while those having 30% epidermis involvement are labeled as ten . cases with between 10% and 30% involved areas are defined as overlap sjs ten . the estimated incidence of ten is about 1.171.89 cases per million inhabitants per year in observational studies . the risk of ten is much higher in individuals having hiv infection and other autoimmune disorders.[1012 ] ten is almost always induced by drugs . although a long list of drugs have been implicated as a cause for ten , the drugs commonly associated with ten are sulfonamides , anticonvulsants , allopurinol , non - steroidal anti - inflammatory drugs , etc . ten / sjs induced by fluconazole is very rare and till date only three cases have been reported . the very first case involved a 30-year - old gay , hiv positive man developing sjs following fluconazole therapy for oral candidiasis . the second case involved a 33-year - old hiv positive male who developed ten following treatment with fluconazole for dysphagia and recurrent oral thrush . the third one was a 52-year - old immunosuppressive woman developing ten after fluconazole for the treatment of esophageal candidasis . the exact mechanism for drug - induced ten is unclear but immunological mechanisms , reactive drug metabolites and interactions between the two have been proposed . multiple prior cutaneous drug reaction or prior reaction to a specific drug is also found to be a risk factor for ten in hiv infected individuals . the higher incidence of ten among hiv - infected patients may also be related to glutathione deficiency . b cells which are defectively activated to secrete immunoglobulins leading to circulatory immune complexes and autoimmune phenomenon . all these mechanism makes hiv - infected individuals more vulnerable to ten due to drugs . therefore in all patients with drug - induced ten , an underlying hiv infection should always be ruled out . to our knowledge this is the fourth reported case and probably be the first one in india of ten secondary to fluconazole in a patient having hiv infection .

stevens - johnson syndrome and toxic epidermal necrolysis ( ten ) are rare but serious dermatologic disorders . these grave conditions present as medical emergency , requiring prompt diagnosis and management . these are often drug induced and various groups of drugs , such as sulfa drugs , nsaids , etc . , have been implicated as to cause ten . fluconazole is a commonly used drug with mild side effects . ten caused by fluconazole is rare , and till now only few cases have been reported in the literature . we present a case of ten in a human immunodeficiency virus infected man following fluconazole therapy in view of its rare occurrence .

the totally extraperitoneal ( tep ) method is essentially a laparoscopic modification of stoppa 's repair . we report a case of pneumomediastinum and subcutaneous emphysema of the neck , without pneumothorax , after tep repair . a 52-year - old man , american society of anesthesiologists ( asa ) level i , 75 kg , 173 cm , with left indirect groin hernia was admitted for elective inguinal repair using the tep operation . after an uneventful intubation , tep repair of the hernia utilizing three midline trocars technique was performed . at the beginning of the procedure , so2 was 98% , blood pressure 140/65 and fetco2 was 34 mm hg . operation time was 32 minutes , with a mean intra - abdominal pressure of 11 - 12 mm hg . during the procedure , immediately after extubation , the patient had severe chest pain , o2 saturation decreased to 84% , fetco2 increased to 44 mm hg , and subcutaneous emphysema of the neck was detected . there was no emphysema of the abdomen or of the back . a chest film and thoracic computed tomographic ( ct ) scan ( figure 1 ) confirmed the presence of pneumomediastinum without pneumothorax . after two hours of mechanical ventilation , the emphysema resolved , and the patient was extubated without any problem . theoretical advantages of laparoscopic hernioplasty are less pain and a quicker return to normal activity . complications of tep laparoscopic hernioplasty are reported infrequently . bowel , bladder and vessel injuries , bleeding in the preperitoneal space , severe testicular pain , hemoscrotum or nerve entrapment noted with tep hernioplasty have also have been described in open procedures . the detractors of lh mainly criticize the laparoscopic approach as usually being performed under general anesthesia with higher costs and an unknown recurrence rate . however , large series have been published showing a low recurrence rate ( 0% - 1% ) . tep repair is performed by dissecting the preperitoneal space and insufflating carbon dioxide to maintain an operative space without pneumoperitoneum . this method potentially avoids the risks of intra - abdominal organ lesions and peritoneal adhesions . air may enter the mediastinum from the esophagus , trachea , bronchi , lung , neck , abdomen or retroperitoneal space , producing mediastinal emphysema or pneumomediastinum . co2 usually passes through the hiatus secondary to a congenital anomaly , weak points , defects or a tear in the diaphragm . high - working co2 pressure or a prolonged insufflation time can also lead to pneumomediastinum . a recent article described a tep procedure complicated by hypercarbia and the development of massive subcutaneous emphysema without hemodynamic instability . this was thought to be due to a high intra - abdominal insufflation pressure that ranged between 14 and 18 mm hg . it is thought that pneumothorax as a complication of tep is most likely related to high insufflation pressure and the length of the procedure , itself . both cases occurred in healthy patients without hiatal or diaphragmatic hernia , and the condition was solved without the placement of a chest tube . a retroperitoneal route could be postulated on extraperitoneal procedures , but no retroperitoneal air was present on ct , and no back emphysema was detected . in our case , a hiatal route is the most reasonable explanation for the occurance of pneumomediatinum as insufflation time was short , working pressure was low , and bronchoscopy ruled out an airway disruption . we conclude that pneumomediastinum can occur after extraperitoneal laparoscopic hernia procedures due to small tears in the peritoneum that occur even with low - working pressures .

a 52-year - old man with left indirect groin hernia was admitted for elective inguinal repair using the totally extraperitoneal ( tep ) approach . after an uneventful intubation , tep repair of the hernia was performed with three midline trocars . immediately after extubation , the patient noted severe chest pain . there was a decrease in pao2 saturation , and neck subcutaneous emphysema was detected . there was no emphysema of the abdomen or of the back . a chest film and thoracic computed tomographic ( ct ) scan confirmed the presence of pneumomediastinum without pneumothorax . the patient was discharged without complications .

the european guidelines for the management of bleeding in the trauma patient recently reported by spahn and coworkers is a multidisciplinary , multi - institutional , evidence - based , consensus - driven approach to the diagnosis and management of bleeding in the injured patient . although potentially susceptible to bias introduced by the authors , the grading system used is generally appropriate . the majority of the recommendations are sound and are centered around rapid control of surgical bleeding , proper resuscitation , and transfusion of red cells and coagulation factors . fittingly , the authors make their most important recommendation first ; ' the time between injury and definitive control of bleeding must be minimized . ' although seemingly obvious , in a recent study from a high - volume , mature trauma system , a common cause of preventable death was failure to identify and control surgical bleeding . however , some of the other recommendations based on less definitive data remain controversial and are not necessarily mainstream . also , some of the recommendations need to be placed in context , particularly in terms of the dynamic continuum of patient management over time . for example , the recommendation that red cell transfusion be based on a conservative transfusion trigger ( hemoglobin 7 to 9 g / dl ) is based on solid evidence . however , that evidence applies only to the stabilized ( postoperative ) patient who is no longer bleeding massively . within this context it would be unwise to await laboratory data to decide whether to transfuse an acutely bleeding patient . under such dynamic circumstances , the decision must be based on clinical factors such as vital signs , response to resuscitation , volume of ongoing bleeding , and the success of surgical attempts to control bleeding . likewise , transfusion of thawed plasma under those circumstances should not await the results of an international normalized ratio ( inr , for prothrombin time ) , but rather the decision should be based on clinical factors . once bleeding is controlled and the patient is stabilized , such strict laboratory - guided transfusion practices can be followed . in fact , recent evidence indicates that coagulation products should be infused very early , indeed pre - emptively , in the face of ongoing severe hemorrhage . in an attempt to minimize the coagulopathy associated with severe bleeding and transfusion , protocols for massive transfusion have been developed by a number of institutions as well as the us military in iraq . although the optimal ratio of blood to plasma transfusion is yet to be determined definitively , recent data suggest that this ratio is probably close to 1:1 in the patient with massive bleeding and shock . if they are eventually demonstrated to be effective , such early infusions of plasma would be given long before hemoglobin and inr tests could be performed . regardless of these types of caveats , converting evidence - based recommendations into standard day - to - day operational procedures can still leave plenty of opportunity for interpretation and resulting variability in practice . for example , recommendation 4 from the bleeding management guideline states that , ' we recommend that patients presenting with haemorrhagic shock and an identified source of bleeding undergo an immediate bleeding control procedure unless initial resuscitation measures are successful . ' for the purposes of an operational protocol , how does one define ' successful resuscitation ' ? a definition of successful resuscitation to one surgeon may still be considered a state of ongoing bleeding and continued need for transfusion by another . at the heart of this matter is the presumption that , ultimately , there is a ' best way ' to care for bleeding patients . the goal of evidence - based guidelines is to help develop recommendations not only to identify the best practice but also to decrease variability in delivery of care . however , while more definitive data are lacking , many guidelines including many of those in the present discussion must remain fairly broad in order to accommodate controversial and divergent points of view . accordingly , the european guideline should be viewed as an excellent and timely consensus , but one that will remain a work - in - progress that must continually be refined as new data are accumulated . the authors should be applauded for their tremendous initiative ; moreover , it is strongly recommended that they continue to regroup regularly in order to refine these recommendations further as permitted by the evolving evidence .

the development of evidence - based guidelines has gained popularity as a strategy to reduce variation in practice and to orient clinical care around documentable best practices . based on available data , the new european guidelines for the management of bleeding in the trauma patient do deliver a number of sound recommendations . however , some issues remain controversial and , like many guidelines , the actual translation of these evidence - based recommendations into routine clinical practice protocols continues to leave opportunity for variation . nevertheless , this consensus guideline provides an excellent starting point . as evidence continues to accumulate , future iterations should provide greater specificity and move us closer to the definitive best practice .

fluid management in patients with acute respiratory distress syndrome ( ards ) is particularly difficult . in hemodynamically stable patients fluid restriction is warranted as it decreases the length of need for ventilatory support . however , at the initial phases , patients with ards also often present hemodynamic instability and are at risk of tissue hypoperfusion and even tissue hypoxia , which may further contribute to exacerbation of ards by boosting activation of inflammation and coagulation . guidance of fluid administration is often complicated by the high pleural pressures , associated with high positive end - expiratory pressure ( peep ) levels , that affect measurements of intravascular pressures . multiple studies have shown that static indices of preload , being pressures or volumes , often fail to predict the response to fluids . on the contrary , dynamic indices based on heart - lung interactions , such as pulse pressure variations ( pp ) , have repeatedly been found to reliably predict the response to fluids in mechanically ventilated patients . in patients with ards , ventilation with low tidal volume is recommended . in patients ventilated with low tidal volume , pulse pressure variations do not predict adequately the response to fluids [ 6 - 8 ] . in this issue of critical care , lakhal and lakhal and colleagues observed that pulse pressure variations moderately predicted the response to fluids and that the predictive value was equivalent to that of pulmonary artery pressure . first , this trial confirms that pulse pressure variations fail to predict fluid responsiveness in a large series of patients with ards ventilated according to current guidelines . indeed , it has been advocated that changes in pleural pressure may be preserved , as lung compliance is also reduced in ards patients . the issue is that changes in pleural pressure can not be reliably estimated from the difference between plateau and end - expiratory pressure as the transmission of pressure from airway to pleura markedly varies among patients . even selecting patients with large driving pressure failed to improve the predictive value of pp . these results are in line with the observations of valle and colleagues , who found that correcting dpp by driving pressure failed to improve the predictive value for fluid responsiveness . interestingly , in the few patients with a difference between inspiratory and expiratory pulmonary artery pressure higher than 4 mmhg , the prediction of pp was excellent ( area under the curve 1.0 ( 95% confidence interval 0.73 to 1.0 ) ) . this suggests that these indices can be used when changes in alveolar pressure are effectively transmitted to pleural pressure . unfortunately , this requires invasive measurements of intravascular pressures by pulmonary artery catheter or of esophageal pressure . another important factor may be that respiratory rate is often high when ventilating with low tidal volume . we observed that pp was negligible in fluid responders when the ratio of heart rate to respiratory rate was decreased below 3.6 by increasing respiratory rates . muller and colleagues recently confirmed that pp can be low in fluid responders when this ratio is low . lakhal and colleagues confirmed the combined influence of tidal volume and respiratory rate . using a composite index computed as the product of tidal volume by heart rate divided by respiratory rate , they observed that pp was significantly larger in responders than in non - responders only in patients with above median values of this composite index . it is important to acknowledge the limitations of pp and related indices . when applied correctly more importantly , resuscitation strategies based on these indices are associated with better hemodynamic stability and lower incidence of postoperative organ dysfunction . in patients with ards , the use of these indices is unfortunately limited by several factors , including low tidal volume , high respiratory rate and right ventricular dysfunction .

respiratory - associated variations in stroke volume and pulse pressure are frequently used to predict the response to fluid administration . however , it has been demonstrated that low tidal volume ventilation may limit their use in patients with acute respiratory distress syndrome ( ards ) . in this issue , a trial investigates the value of pulse pressure variation to predict fluid responsiveness in a large series of patients with ards ventilated according to current guidelines .

tuberculous lesions of the liver may be unexpectedly detected during abdominal surgery . also , these lesions can , mistakenly , be diagnosed as tumour - like mass or cystic lesions in the liver on screening investigations . , we discuss two cases of hepatic tuberculosis due to diagnostic dilemma and rarity of these lesions . a 30-year - old woman was admitted to our hospital with a history of abdominal pain , anorexia , weight loss and fever for one month . on physical examination , except hepatomegaly , no other physical finding was detected . liver function tests ( alt 64 u / l , ast 76 u / l ) and alkaline phosphatase ( alp 310 ultrasonography ( us ) of the abdomen revealed a lobulated cystic mass with air in the right lobe of the liver . computerized tomography ( ct ) of the abdomen showed an ill - defined cystic mass with air - fluid level in the anterior segments of right liver lobe ( fig . 1 ) . liver abscess or infected hydatid disease was considered with these radiologic and clinical findings . histopathological examination of the specimen demonstrated granulomas , epithelioid histiocytes and langhans giant cells , and caseating granulomatous inflammation . unfortunately we did not perform culture of the specimen for tuberculosis , because tuberculosis was not taken in account in the differential diagnosis . a 63-year - old man was admitted to our hospital with abdominal pain and fever for one week . except for abdominal tenderness , physical examination was normal . only white blood cells ( 16,400/mm ) and liver function values ( alt 48 u / l , ast 52 abdominal us revealed a tumour - like mass lesion on the right lobe of the liver . histopathological examination of the specimen demonstrated granulomas , epithelioid histiocytes and langhans giant cells , and caseating granulomatous inflammation . tuberculosis is the leading cause of death among infectious diseases . among extra - pulmonary tuberculosis , described three morphologic types of hepatic tuberculosis : ( 1 ) miliary tuberculosis of the liver associated with generalized miliary tuberculosis , ( 2 ) primary miliary tuberculosis of the liver without involvement of other organs , and ( 3 ) primary tuberculous granuloma or abscesses of the liver . our two cases had only hepatic tuberculosis without pulmonary infection , which we accept as type 3 . the symptoms and signs of hepatic involvement are non - specific . similar to our cases mri and ct findings of liver tuberculosis reveal different stages of disease , varying from granulomatous tubercles with or without caseation necrosis to fibrosis and calcification in the healing stage . ct findings demonstrate miliary , hypodense nodular lesions or cystic lesion . in our cases , one of the patients had cystic mass with air - fluid level , and the other patient had tumour - like mass lesion . percutaneous needle biopsy and laparoscopy can be used for the diagnosis and occasionally laparotomy may be required in some patients . infectious and non - infectious diseases which can cause caseating or non - caseating hepatic granulomatous such as leprosy , sarcoidosis , hodgkin 's disease , brucellosis , infectious mononucleosis , inflammatory bowel disease , drug - induced liver damage and syphilis should be considered in the differential diagnosis of hepatic tuberculosis . antituberculous treatment should be given at least for one year ; however , many anti - tuberculous drugs are hepatotoxic and may induce liver damage and jaundice . therefore the response and toxicity should be assessed carefully during treatment . our patients were successfully treated for one year with anti - tuberculous drugs , and no side effect was observed during the treatment period . in conclusion , tuberculosis should be considered in the differential diagnosis of cystic and tumour - like mass lesions of the liver , especially in endemic regions .

hepatic tuberculosis is a rare manifestation of extra - pulmonary tuberculosis . hepatic tuberculous lesions are especially mimicking tumour - like mass or cystic lesions in the liver and so can be misdiagnosed with several diseases . histopathological examination of the specimen is essential in the diagnosis for hepatic tuberculosis . in this report , two cases with hepatic tuberculosis having cystic solid mass and abscess liver lesions are described .

we report a 14 month old girl who developed early pacemaker infective endocarditis six weeks after insertion of the pacing device , and briefly discuss investigation and management . there are no other reported cases of paediatric pacemaker infective endocarditis from haemophilus parainfluenazae in the literature . in march 2007 a 14 month old girl with congenital complete heart block underwent implantation of an endocardial pacemaker into the right ventricle . six weeks after her pacemaker insertion , she presented with pyrexia , lethargy , diarrhoea , vomiting and poor fluid intake . she was pale and lethargic with a liver edge extending to 1.5 cm below the costal margin , and a grade 2/6 systolic murmur . two weeks after initial presentation she became acutely unwell , presenting in shock , with a grade 3/6 systolic murmur . cxr showed an enlarged heart with pacemaker and lead in situ ( fig 1 ) . chest x ray showing cardiomegaly with pacemaker in situ investigations showed a c - reactive protein of 275 mg / l , white cell count of 35.710/l , neutrophils 17.210/l , platelets 11110/l , and haemoglobin of 7.6g / dl . an echocardiogram showed a mobile mass attached to the pacing wire and tricuspid valve , moderate tricuspid regurgitation and a patent foramen ovale 2d echocardiography showing mobile vegetation 7.8 mm 9.6 mm gentamycin was added to the antibiotic regime . blood cultures showed a pure growth of gram negative rods later confirmed to be haemophilus parainfluenzae , sensitive to 3 generation cephalosporins and gentamycin . one week later she underwent open heart surgery under bypass for the complete removal of the infected cardiac pacemaker system ( fig . a follow - up 2d echocardiogram showed moderate tricuspid regurgitation and a small vegetation that was still present near the apical ventricular septum . if untreated pacemaker infective endocarditis is fatal . we discuss some management issues surrounding pacemaker endocarditis in children with emphasis on h parainfluenzae . one study by cohen et al , in a 20 year paediatric cohort , reports an overall incidence of 2.3% of implanted pacemakers.1 case reports of pacemaker endocarditis from h. parainfluenzae are rare . we conducted a literature search using medline ( 1950 to date ) and embase ( 1974 to date ) using the key words pacemaker , endocarditis , infection , haemophilus parainfluenzae . we found only two other case reports of pacemaker endocarditis caused by haemophilus parainflunzae , and both were in adults ( table 1).23 case reports of pacemaker endocarditis caused by haemophilus parainfluenzae haemophilus parainfluenzae is a commensal of the oral cavity and the upper respiratory tract , and belongs to a group of fastidious gram negative bacilli collectively known as hacek ( haemophilus spp , actinobacillus actinomycetemcomitans , cardiobacterium hominis , eikinella corrodens and kingella kingae ) . it is an unusual cause of device related bacterial endocarditis , the majority of which are caused by staphylococcus aureus and epidermidis . the commonest presenting feature is fever which is present in 80% of cases.4 septic shock is the presentation in only 9% of cases.5 when suspected , transthoracic echocardiography ( tte ) is an excellent tool for the detection of vegetations in children , with a reported sensitivity of 81% . transoesophageal echocardiography ( toe ) has been shown to have a superior detection rate in adults , but its role in children has yet to be established . a meta analysis by de costa et al was shown to significantly reduce the incidence of serious infective complications following pacemaker insertion.6 secondary prophylaxis is not routinely recommended , and the american heart academy still classifies this group as a negligible risk category . we could not define a clear source of infection in our patient , and can only postulate haematogenous spread from her middle ear infection . third generation cephalosporins are an excellent choice of antibiotic for hacek organisms which are often resistant to penicillins . however removal of the infected intracardiac device offers the best chance of complete eradication of the infection , with lower morbidity and mortality.78 device re - implantation should be at a new site when the patient is no longer bacteraemic . pacemaker endocarditis should always be suspected in a child with an implanted pacemaker presenting with fever .

we report a case of pacemaker infective endocarditis in a 14 month old girl , caused by haemophilus parainfluenzae . there are no other cases in children reported in the literature . we discuss the issues surrounding the case and the evidence which influenced our management .

fluorine-18 fluorodeoxyglucose positron emission tomography - computed tomography ( f-18 fdg pet - ct ) has evolved as an imaging modality of choice for staging , restaging , treatment response evaluation and surveillance of various cancers . increased fdg uptake is also noted in various benign conditions . we report a case where f-18 fdg pet - ct scan demonstrated residual disease in an operated case of i m . a 36-year - old male presented with slow growing painless lump in the left thigh . magnetic resonance imaging ( mri ) of the thigh demonstrated a well - defined soft tissue lesion in the left rectus femoris muscle with homogenous low - signal intensity on t1-weighted sequences and markedly high signal intensity on t2-weighted sequences . contrast - enhanced t1-weighted mris showed heterogeneous enhancement throughout the mass [ figure 1 ] . excision of the mass was performed and on histopathological examination the tumor showed composition of spindle and stellate shaped cells that were widely separated by myxoid stroma . the tumor cells were immunohistochemically positive for vimentin and cd34 , but negative for s-100 and desmin . however , the cut margins were not free and the patient was referred 10 weeks later for f-18 fdg pet - ct scan for the assessment of residual disease . increased f-18-fdg uptake was noted in the peripheral rim of a well - circumscribed centrally hypodense soft tissue lesion with thin septation in the left rectus femoris muscle [ figure 2 ] . the maximum standardized uptake value ( suvmax ) of the lesion was 3.2 g / ml . these findings were suggestive of residual i m and the same was confirmed on histopathological examination of the postexcision specimen . magnetic resonance imaging ( mri ) of the thigh demonstrated a well - defined soft tissue lesion in the left rectus femoris muscle with homogenous low signal intensity on t1-weighted sequences ( a ) and markedly high signal intensity on t2-weighted sequences ( b ) . contrast - enhanced mris showed heterogeneous enhancement throughout the mass ( c ) increased fluorodeoxyglucose uptake was noted in the left upper thigh on maximum intensity projected ( a ) , coronal positron emission tomography ( pet ) ( b ) and sagittal pet ( e ) images . this uptake corresponds to the peripheral rim of a well circumscribed centrally hypodense soft tissue lesion with thin septation in the left rectus femoris muscle on correlative coronal and sagittal computed tomography ( ct ) images ( c and f ) and pet - ct images ( d and g ) , respectively most of them appear in the fourth to sixth decades of life with a slight female preponderance . i m 's usually arise from large skeletal muscles , the commonest location being the lower extremities , particularly the thigh ( 51% ) , followed by the gluteal region ( 7% ) . the symptoms depend on the site and size of the mass lesion . although majority of the patients were asymptomatic and diagnosed incidentally , few present with slow growing , painless intramuscular tumor . occasionally , the symptoms of nerve root compression may be noted . i m usually occurs as an isolated lesion , but sometimes it is associated with skeletal fibrous dysplasia , when it is known as mazabraud 's syndrome . in such cases , preoperatively , it is difficult to differentiate i m from other malignant myxoid tumors such as myxoid liposarcoma , low - grade fibromyxoid sarcoma , myxoid malignant fibrous histiocytoma and extra skeletal myxoid chondrosarcoma . on imaging , i m appears as hypoechoic mass on ultrasound , shows low attenuation on ct , low - signal intensity on t1-weighted mris , and markedly high signal intensity on t2-weighted mris . contrast - enhanced imaging ( particularly mris ) more accurately reflects the truly solid ( although usually hypocellular ) consistency of the i m because it shows internal enhancement . several studies have been performed to evaluate the role of f-18 fdg pet - ct scan to differentiate benign and malignant myxoid neoplasms . however , the results were not encouraging . the definite diagnosis of i m can only be made from the histopathological examination after its surgical excision , which is considered to be the treatment of choice . complete resection is associated with excellent prognosis and no local recurrence generally occurs during an average follow - up of 7 years after excision . however , incomplete resection is associated with recurrence and completeness of surgery is the most important determinant of the overall outcome of the disease . i m shows low - grade f-18 fdg uptake with suv values between 1.3 and 2.6 . furthermore , whole body f-18 fdg pet - ct has been reported to be useful to detect fibrosis dysplasia and single or multiple i m 's in mazabraud 's syndrome . however , its role in assessing postoperative residual i m is not described in the literature to the best of our knowledge . this case demonstrates the possible role of f-18 fdg pet - ct scan to detect postoperative residual i m .

intramuscular myxoma ( i m ) is a rare benign neoplasm . in a patient diagnosed with i m of left thigh , we report the utility of a postoperative fluorine-18 fluorodeoxyglucose positron emission tomography - computed tomography scan in assessing the efficacy of surgical excision .

a 48-year - old , previously healthy woman was found to have a subepithelial tumor during health screening endoscopy ( fig . 1a ) . abdominal computed tomography demonstrated a 3.6 2.2 cm , well - defined , solid , enhancing submucosal mass on the posterior wall of the stomach midbody ( fig . there was no further history , symptoms , or signs of systemic disease , and laboratory tests were unremarkable . given a presumptive diagnosis of submucosal neoplasm , wedge resection was performed . grossly , the lesion was a poorly circumscribed , yellowish grey , fusiform mass involving the area from the submucosa to subserosa ( fig . microscopically , the mass showed marked fibrosis , often in a storiform pattern of many lymphoid follicles with well - formed germinal centers , and diffuse inflammatory cell infiltration . the infiltrated inflammatory cells were mainly lymphocytes and plasma cells , but some eosinophils were also found ( fig . there were numerous igg4-positive cells throughout the lesion , and the number of igg4-positive plasma cells was up to 210 per high - power field ( fig . the igg4 to igg - positive cell number ratio was about 85% ; however , there were only a few igg4-positive cells in the mucosa . there was no significant myofibroblastic proliferation or immunostaining for anaplastic lymphoma kinase ; therefore , the possibility of inflammatory myofibroblastic tumor was excluded . the patient s postoperative course was uneventful , and she was discharged without any complications . here , we described a case of isolated gastric igg4-rd presenting as a fusiform mural mass mimicking neoplasm , such as gi stromal tumor or neuroendocrine tumor . to the best of our knowledge , histologically , this case demonstrated all the important features of igg4-rd , including dense lymphoplasmacytic infiltration , storiform fibrosis , obliterative phlebitis , and abundant igg4-positive cells . although other diagnostic criteria , such as elevated serum igg4 level or response to steroid therapy , could not be confirmed due to the clinical presentation , typical histopathologic features led us to consider igg4-rd . increased igg4-positive plasma cells can be seen in other organs and in many conditions , including non - specific chronic inflammation , lymphoma , and other malignancies [ 4,6 - 8 ] . however , these lesions lack other characteristic histopathologic findings , such as storiform fibrosis and oblierative phlebitis , as has been described in the consensus statement on the pathology of igg4-rd . although abundant igg4-positive plasma cell infiltration is not uncommon in the gi tract in the setting of autoimmune pancreatitis , the simple presence of igg4-positive cells does not justify a diagnosis of igg4-rd in the absence of other gross and microscopic features , such as tumefactive nature , storiform fibrosis , and obliterative phlebitis . including the present case , there have been nine cases of massforming igg4-rd in the stomach [ 9 - 14 ] . a case of probable igg4-rd that presented as a gastric ulcer has also been reported , but we excluded this case from the present review . as is summarized in table 1 , most gastric igg4-rd was detected in middle age ( mean , 58.8 years ; range , 45 to 75 years ) , and men and women were affected equally , although the total number of patients is likely too small to reveal any meaningful data . seven patients had solitary nodules or masses , whereas two patients had multiple polyps or nodules . the two patients with multiple lesions also had autoimmune pancreatitis and autoimmune polyendocrinopathy , respectively . four of the seven cases showing a solitary lesion had no sign of multi - organ involvement . most cases of gastric igg4-rd ( six of nine ) involved the submucosal layer of the gastric body . most gastric igg4-rd patients were treated surgically except for one patient with autoimmune pancreatitis who was treated with steroid . steroid treatment is the first therapeutic option in igg4-rd , but all reported isolated gastric igg4-rd cases were surgically resected , presumably because they are rare and difficult to diagnose without pathologic examination of a resected specimen . unnecessary surgery might be avoided if the possibility of igg4-rd is kept in mind and careful pathologic evaluation including igg4 immunostaining is performed on a deep biopsy obtained using endoscopic ultrasonography .

isolated gastric igg4-related disease ( igg4-rd ) is a very rare tumefactive inflammatory condition , with only a few cases reported to date . a 48-year - old woman was incidentally found to have a subepithelial tumor in the stomach . given a presumptive diagnosis of gastrointestinal stromal tumor or neuroendocrine tumor , she underwent wedge resection . the lesion was vaguely nodular and mainly involved the submucosa and proper muscle layer . microscopically , all classical features of type i autoimmune pancreatitis including lymphoplasmacytic infiltration , storiform fibrosis , obliterative phlebitis , and numerous igg4-positive plasma cells were seen . she had no evidence of igg4-rd in other organs . although very rare , igg4-rd should be considered one of the differential diagnoses in the setting of gastric wall thickening or subepithelial mass - like lesion . deep biopsy with awareness of this entity might avoid unnecessary surgical intervention .

thyroid hormone levels drop down in starvation and in severe illness without detectable clinical effect of hypothyroidism . this condition was generally known as euthyroid sick syndrome , but was later renamed as nonthyroidal illness syndrome ( ntis ) . ntis is evidenced by low levels of thyroxines and normal or low levels of thyroid - stimulating hormone ( tsh ) and elevated reverse tri - iodothyronine ( rt3 ) level . clinical features of hypothyroidism do not develop within even 2 - 3 weeks , but require much longer period for expression . ramrez et al . studied patients receiving chronic hemodialysis and found a high prevalence of goiter ( 58% ) and low serum tetra - iodothyronine ( t4 ) , t3 , and tsh levels . however , rt3 levels were not elevated in patients with end stage renal disease ( esrd ) . the low levels of thyroid hormones may be due to decreased peripheral de - iodization of t4 and low levels of hormone binding proteins . the nonthyroidal illness serves to defend protein loss and an attempt to supplement hormone causes excess protein loss . the kidneys preserve iodine in patients suffering from esrd and prevalence of goiter and hypothyroidism was observed high in patients with esrd compared with the general population in iodine deficient region . hyperthyroidism is uncommon in patients receiving dialysis , but sporadic case reports are available since 1980 . a 28-year - old female reported in march 2005 for evaluation of renal dysfunction noted during her first pregnancy . she developed bilateral pedal edema , hypertension and proteinuria during the third trimester of the first pregnancy in 1998 . she underwent caesarean section at 8 month of gestation , indication being accelerated hypertension . on evaluation 2 months after delivery , she was found to have persistent hypertension , proteinuria , and elevated serum creatinine ( 1.72 mg / dl ) . tests for hiv , hepatitis b and c were negative . screening tests for systemic lupus erythematosus were found negative . kidney biopsy showed focal glomerular sclerosis with an increase in the mesangial matrix and tubular atrophy , suggestive of focal segmental glomerulosclerosis . she was treated with oral prednisolone , but developed pain in both hip joints after 5 months . regional magnetic resonance imaging showed avascular necrosis of left femur head and so prednisolone was tapered and discontinued . thyroid hormone levels were normal [ table 1 ] . in 2009 , she experienced a sudden loss of weight ( 4 kg over 4 months ) . clinical examination revealed body mass index 18.59 kg / m , with grade i diffuse enlargement of thyroid gland and grade ii inactive orbitopathy . technetium 99 thyroid scintigraphy showed diffuse uptake of 6.8% ( normal 1 - 4% ) indicating graves ' disease ( gd ) [ figure 1 ] . technetium pertechnetate thyroid thyroid scintigraphy she was placed on carbimazole ( 20 mg daily as a single dose ) and propranolol ( 20 mg twice daily ) . she developed relapse of hyperthyroidism upon weaning carbimazole after 14 months , and total thyroidectomy was offered . she developed generalized body pain , asthenia and muscle weakness suggestive of myopathy and became wheel chair bound by 2012 . bone mineral densitometry studies of right wrist and lumbar spines showed t score of 3 and 2.7 , respectively . technetium mibi double phase parathyroid scintigraph after control of thyrotoxicosis , total thyroidectomy and subtotal parathyroidectomy retaining portion of right superior parathyroid gland were done . thyroid gland was diffusely enlarged with nodules in the right lobe and weighed 49 g. histology showed hyperplasic follicles lined by occasional tall follicular cells . the final histology showed features suggestive of gd of the thyroid gland and hyperplasia of parathyroid glands . the first case was reported in 1980 but later about 18 such cases were reported . however , boubakera et al . in a comprehensive review the present case demonstrated clinical features of hyperthyroidism with diffuse goiter and orbital involvement suggesting an autoimmune etiology . the clinical diagnosis of hyperthyroidism in esrd may be delayed due to overlap of symptoms . in suspected patients . the serum iodine level as well as intrathyroidal iodine content can be high in esrd patients and likely to exert transient or extended wolf - chaikoff phenomenon . the excess iodine may induce thyroiditis resulting in hypothyroidism . on the other hand , whether the excess iodine stimulates the gland to a hyperactive state ( jod - basedow effect ) is not clear . carbimazole was used in this patient after dialysis on those days since the drug could be washed off during the procedure . high dose of carbimazole was recommended since failure of conversion to active form is likely due to metabolic acidosis . surgical treatment was planned in this patient due to the presence of active orbitopathy and secondary hyperparathyroidism . all the more use of glucocorticoids was not advisable in case of deterioration of orbitopathy .

thyroid hormone level may be altered in chronic renal failure patients . low levels of thyroxine protect the body from excess protein loss by minimizing catabolism . hyperthyroidism is rarely encountered in end - stage dialysis dependent patients . less than 10 well - documented cases of graves ' disease ( gd ) are reported in literature so far . we report a case of gd in a patient on dialysis .

in 2014 , more than 32 000 women in the usa are estimated to be diagnosed with melanoma , with more than 3000 women dying from melanoma . moreover , nearly one - third of women diagnosed with melanoma are of childbearing age and 8% of malignancies are diagnosed during pregnancy . the treatment of melanoma in pregnant patients remains difficult and controversial as there are obvious concerns for the fetus , which include administering contrast agents , general anesthesia and as well as the stress of a major operation . although open inguinal lymphadenectomy has historically been the standard treatment for metastatic melanoma in the inguinal lymph node basin , multiple studies have reported significant morbidity following the procedure , with wound complication rates of 50% or higher [ 35 ] . videoscopic inguinal lymphadenectomy ( vil ) is a minimally invasive alternative that reports fewer wound complications , shorter hospital length of stay and a similar lymph node yield . this case report further illustrates the safety of vil as it is successfully performed in an expectant mother of 24 weeks . in june 2013 , a 38-year - old woman underwent wide local excision of her right thigh 1.4 mm thick melanoma with a negative sentinel lymph node biopsy ( slnbx ) . one year later and now 20 weeks pregnant , the patient was referred to our clinic with a palpable mass in the slnbx basin . preoperative imaging included computed tomography of the chest and magnetic resonance imaging of the abdomen and pelvis . these studies revealed post - biopsy changes in the right groin without involvement of the deep lymph nodes or other sites of concern . both video - assisted and standard open superficial groin dissections were discussed with the patient , as well as the risks and benefits of undergoing surgery during pregnancy . after conferring with our melanoma multidisciplinary clinic and obtaining approval from anesthesia and maternal fetal medicine , the patient and our team agreed to proceed with vil . at the time of her she was positioned supine on a split - leg table with a bump under her right hip to offload the inferior vena cava from the uterus . the femoral triangle boundaries were mapped out with a surgical pen , and the area was prepped and draped in a sterile fashion . a skin incision was made 10 cm inferior to the femoral triangle apex to accommodate a 10-mm balloon trocar . carbon dioxide ( co2 ) insufflation was started at 20 mmhg for the first 15 min and then lowered to 12 mmhg for the duration of the case . end - tidal co2 never reached above 35 mmhg during the case per anesthesia . under direct visualization using a zero - degree laparoscope , two 10 mm trocars were placed outside the medial and lateral boundaries of the demarcated femoral triangle . we identified the medial and lateral boundaries of the dissection to include the adductor longus and the sartorius muscle fascia . starting at the apex of the femoral triangle , dissection above the deep fascia of the thigh was advanced superiorly to the level of the inguinal ligament with identification of key structures , including the femoral vein and artery . the lymph node packet was removed from our working space and a 19-fr blake drain was introduced through the anterolateral 10-mm port and secured to the skin . both 3 - 0 vicryl and 4 - 0 monocryl sutures were used to approximate the dermal and skin edges . the patient was awakened from anesthesia and transported to the obstetrics recovery unit where fetal heart tones were monitored and a viable fetus was confirmed . her wounds healed well with no complications and the drain was removed 6 weeks after surgery . common complications include seromas , flap necrosis , wound infections and lymphedema . using vil , previous studies have demonstrated both equal and improved lymph node retrieval with decreased morbidity [ 6 , 7 ] . avoiding wound - related complications not only improves recovery time , but also decreases hospital readmission rates and need for postoperative antibiotics . this is most likely due to the fact that since the wounds do not breakdown , the drains continue to work instead of draining by way of an open wound and requirements for a wound vac . an additional potential concern is the retention of co2 during the case secondary to subcutaneous induced emphysematous changes of the dissection . while this remains a real concern , it has not yet required us to abort vil to convert to an open approach . this case demonstrates a safe approach to melanoma management without delaying treatment for an aggressive malignancy in the delicate setting of pregnancy . using this approach , we were able to harvest adequate nodes to fully workup a patient who has had multiple melanomas despite presentation in her second trimester of pregnancy . working in a multidisciplinary setting with inclusion of radiologists , oncologists , surgeons , maternal fetal medicine and anesthesia was essential to easily and safely facilitating the patient 's plan of care . while further research is clearly needed , our case demonstrates that vil can be a safe and effective option for inguinal node harvest for melanoma in the previously excluded population of pregnant women .

lymphadenectomy is the standard of care for metastatic melanoma in the inguinal lymph node basin . historically , open surgery was the only treatment option . however , in recent years , videoscopic inguinal lymphadenectomy ( vil ) has become a popular approach as it offers a minimally invasive alternative , provides similar oncologic control and reduces wound complications . even though the vil approach is being used more frequently , the patient populations that stand to benefit the most from this approach are still under investigation . despite continued advances in safety for laparoscopic surgery , many surgeons are hesitant to perform these procedures on pregnant women . in this report , we present a successful vil in a pregnant patient , describe our technique and demonstrate the safety of performing vil in expectant mothers . to our knowledge , this case represents the first vil performed in an expectant mother .

are extremely rare . early and accurate diagnosis of the condition based on perinatal history , clinical examination , and radiographic evaluation is essential because of high rate of lethal outcome with delayed therapy and avoid unnecessary laparotomy . we present this case focusing on usefulness of percutaneous needle aspiration under ultrasonographic guidance in the management . a 26-days - old male neonate weighing 2.9 kg presented with a history of abdominal distention for the past 4 days . clinical examination revealed an active infant having abdominal distention and palpable abdominal masses in bilateral upper quadrant . ultrasonography was performed with prosound-4000 ( aloka , japan ) ultrasound unit using 3.5 mhz curvilinear trasducer . gray - scale sonography demonstrated cystic masses with internal echoes and debris at bilateral supra renal area measuring right 7.9 4.3 cm and left 4.4 4.6 cm , respectively [ figure 1 ] . transverse ultrasound image reveals anechoic cystic masses containing internal echoes and debris at bilateral supra renal area and right is measuring more than left color doppler and power doppler imaging confirmed avascular nature of the mass . magnetic resonance imaging of the abdomen was performed with a 1.5 t unit ( sigma ; ge , u.s.a . ) revealed well - defined bilateral suprarenal cystic masses measuring 7.5 5.6 cm on the right side and 4.0 4.5 cm on the left side . the masses were hypointense to muscle on t-1 weighted imaging and markedly hyperintense on t-2 weighted imaging [ figures 2a and b ] . coronal t1-weighted image ( tr / te 395/13 ms ) ( a ) of the abdomen showing a bilateral suprarenal cystic lesion with hypointense signal intensity and coronal t2-weighted image ( tr / te 4000/101 ms ) ( b ) of the abdomen showing a bilateral suprarenal cystic lesion with hyperintense signal intensity ultrasonographic guidance aspiration was done through posterolateral approach using a 20 gauge needle [ figure 3 ] . ninety milliliters of thick pus was aspirated from right suprarenal abscess and 60 ml from left side and the aspirate was sent for bacteriological study . abdominal distention was reduced within 24 h. serial ultrasonography showed gradual regression of the residual adrenal cavities . the culture of aspirated purulent fluid revealed no growth and there was no clinical evidence of adrenal insufficiency . after 3-months follow up ultrasonography revealed the complete resolution of bilateral suprarenal mass with foci of calcification . longitudinal ultrasound image reveals anechoic cystic mass with wall calcification and needle in the lesion during procedure two theories have been proposed for the development of neonatal adrenal abscess : first , hematogenous bacterial seeding of a normal adrenal gland and second , the seeding of a neonatal adrenal hemorrhage with subsequent abscess formation . in some cases , it is likely that most adrenal abscesses begin with adrenal hemorrhage that is not infrequently associated with traumatic or difficult delivery , hypoxia , sepsis , and coagulopathy . in most reported cases , bacterial examination of abscess material revealed e. coil , or staph aureus but streptococcus , bacteriodes , echovirus , and herpes simplex could also be isolated . differential diagnosis of adrenal collection includes adrenal hemorrhage , cyst , neuroblastoma , wilm 's tumor , renal duplication with dilatation of the upper segment and hydronephrosis . an early and accurate diagnosis of neonatal adrenal abscess is essential to avoid a potentially lethal outcome , as bilateral adrenal abscess in our case . extension of the abscess with involvement of adjacent organs has been reported . a case of retroperitoneal pulmonary fistula caused by a neonatal adrenal abscess ultrasonography and intravenous pyelography ( ivp ) were shown to be useful for diagnosis . at present , ct scan and/or magnetic resonance imaging ( mri ) should enable one to usually arrive at the correct preoperative diagnosis . mondor et al . , described successful drainage using a pigtail catheter placed under ultrasonography guidance . the authors stressed the need to closely follow up the patient by regularly repeated ultrasonography until the adrenal gland is back to normal size . the mainstay of treatment , especially for large lesions or where differentiation from a malignant lesion is difficult is surgical drainage with or without excision . in concordance with other case reports we would like to point out that percutaneous drainage under ultrasonography guidance of bilateral suprarenal abscess has good response . early diagnosis and early nonsurgical treatment may lead to a successful outcome with decreased incidence of unnecessary laparotomy .

neonatal adrenal abscess is a rare condition . bilateral adrenal abscess are extremely rare . we present this case focusing on the usefulness of needle aspiration under ultrasonographic guidance . the bilateral suprarenal cystic masses are identified by ultrasonography and different sequences of magnetic resonance imaging .

abdominal pregnancy is a rare form of ectopic pregnancy with high morbidity and mortality for both the mother and the fetus . ectopic pregnancy represents about 12% of all pregnancies , with 95% of those occurring in the fallopian tubes . the incidence of abdominal pregnancy varies in different populations , ranging from 1:10 000 to 1:30 000 pregnancies , and being most prevalent in developing countries with limited resources and limited diagnostic facilities . the diagnosis of abdominal pregnancy can be challenging , especially in a resource - limited setting . when discovered early , laparotomy is usually done due to the high risk it carries to the mother . an abdominal pregnancy that reaches term with a healthy mother and fetus , thus , is a rare occasion . a 25-year - old g2p0 , presented to the maternity department in msf agok hospital in south sudan , on 14th of october 2016 with abdominal pain . her pregnancy was estimated to be 3435 weeks , as she did not know the exact date of her last menstrual cycle . she had a previous miscarriage , and was concerned about this pregnancy . on examination , the abdomen was mildly tender , and the fetal head could be easily palpated at the right upper quadrant . ultrasound scan was performed , which confirmed a live term fetus , in transverse lie , with no indication of extra - uterine pregnancy . caesarean section was planned in view of the transverse lie . a pfannenstiel ( transverse lower abdominal ) incision was used . a normal non - pregnant uterus there was active bleeding from the placenta , which was attached to the fundus and left cornual region of the uterus , the small bowel mesentery and the left lateral abdominal wall . left ovary was found to be normal , but the left fallopian tube could not be discerned . the baby was well , weighing 2.5 kg , and showing no obvious external congenital abnormalities . both the mother and the baby were kept in the hospital for 2 weeks postoperatively , during which the mother recovered well from surgery , with no signs of infection or bleeding . the child has shown normal developmental milestones till date ( 6 weeks after birth ) . primary abdominal pregnancy refers to an extra - uterine pregnancy where implantation of a fertilized ovum occurs directly in the abdominal cavity while the secondary abdominal pregnancy is a tubal pregnancy that ruptures with reimplantation within the abdominal cavity usually resulting in tubal or ovarian damage . our patient most likely had a secondary abdominal pregnancy , as the left sided fallopian tube could not be identified intraoperatively . a patient usually presents with signs of intra - abdominal bleeding due to separation of the placenta . abdominal pain , painful fetal movements , abnormal presentation and uneffaced cervix should arouse suspicion of ectopic pregnancy , especially abdominal pregnancy . ultrasonography findings of intra - abdominal , extra - uterine fetus and placenta confirms the diagnosis . but even with routine ultrasonography examination , the diagnosis can be missed , especially in the later stages of pregnancy . in a limited - resource setting , such as a field hospital in south sudan optimal management requires careful evaluation and planning . generally speaking , for previable abdominal pregnancies , i.e. prior to 24 week of gestation , immediate operative intervention is indicated , but for viable pregnancies presenting after 24 weeks of gestation , a more conservative approach is advocated , provided the patient can be under strict observation , preferably in a hospital . bleeding from the placental implantation site is the most serious and life - threatening complication of abdominal pregnancy . usually complete removal of the placenta is not advised , and partial removal is only advised to control bleeding . otherwise , it should be left in situ and is removed by involution and resorption . this is usually monitored by measuring the serum human chorionic gonadotropin levels on follow up . in this case , clinical examination and monitoring of the haemoglobin level were the only means available for monitoring the patient . for the newborn , congenital malformations are common , with reports of fetal malformations as high as 40% , with only 50% of these babies surviving the first week of life .

abstractabdominal pregnancy is a rare form of ectopic pregnancy with high morbidity and mortality for both the mother and the fetus . diagnosis can be challenging , especially in a resource - limited setting . we report a case of abdominal pregnancy that presented to mdecins sans frontires field hospital in agok , south sudan , with abdominal pain . examination revealed a term pregnancy and a live fetus in transverse lie . the diagnosis of abdominal pregnancy was made intraoperatively , with successful management and delivery of a healthy baby .

skin and soft tissue fungal infections were rare in survivors from 2004 indian ocean tsunami . only apophysomyces elegans , , fusarium solani and cladophialophora bantiana associated wound infections have been reported in tsunami survivors . cryptococcus gattii may be found worldwide , not only restricted to tropical and subtropical regions . c. gattii has been isolated from the environment e.g. decayed wood , soil around the trees . c. gattii can be divided into four molecular types vgi , vgii , vgiii and vgiv , . c. gattii associated infections usually manifest as pneumonia and meningitis . we report the first case of primary cutaneous cryptococcosis caused by cryptococcus gattii vgii in a tsunami survivor from thailand . a previously healthy 48-year - old thai woman was admitted to vichaiyut hospital in bangkok on december 27 , 2004 ( day 0 being the day of hospital admission ) for treatment of sustained injuries in the tsunami . one day earlier while walking on the beach in front of the hotel in lanta island , krabi province , thailand , she was hit and carried by the tsunami wave towards the hotel . although she was submerged under the sea water for some time , she did not pass out or aspirate the sea water . despite sustaining injury to her chin , right chest , back , right elbow , right index finger , left wrist and both lower legs , she was able to stand and walk to the high ground and later returned to her hotel . there were multiple skin abrasions of her back and anterior surface of both lower legs . rib cage x - ray revealed fracture of right 7th , 8th , 9th and 10th ribs . initially she was treated with broad spectrum antibiotics ciprofloxacin and clindamycin for 2 weeks . however , the wound of both lower legs gradually got worse . the skin of anterior surface of both lower legs appeared inflammed , red and tender . culture of pus from the left lower leg was sent on day 18 and on day 21 cryptococcus spp . ultrasound of the wound of left shin revealed thickening of skin and subcutaneous layer with irregular hypoechoic area about 0.93 cm . debridement of wounds of both lower legs was done under general anaesthesia on day 23 . moderate encapsulated yeast cells were found on the india ink stain of discharge of the left leg but not of the right leg . culture of pus from the left leg grew cryptococcus spp . which was identified as c. gattii serotypes b and c by using the canavanine - glycine - bromothymol blue agar and later it was determined to be c. gattii molecular type vgii by the ura5-rflp typing method ( fig . she was started on anti - fungal treatment on day 21 with iv amphotericin b 0.5 mg / kg / day and oral itraconazole 100 mg twice a day . on day 42 iv amphotericin b was discontinued and itraconazole was switched to oral fluconazole 200 mg daily . repeated india ink stain of pus on day 43 and day 56 still showed encapsulated yeast cells . on day 64 the wound finally closed and no relapse was seen during follow - up for the last nine years ( fig . the lungs and central nervous system are the most common organs affected by c. gattii acquired through the inhalation . primary skin and soft tissue cryptococcosis can occur after the traumatic inoculation . to our knowledge skin and soft tissue infection caused by c. gattii has not been reported in indian ocean tsunami survivors . our previously healthy patient most likely acquired infection during the 2004 tsunami as we found the small piece of woods inside her wound . culture of pus from the left leg grew c. gattii which belonged to vgii molecular type . cryptococcosis caused by c. gattii is uncommon and the most prevalent molecular type of c. gattii is vgii as in our case . vgii can be found in vancouver island , canada , northwestern united states , brazil , columbia and puerto rico . our patient was successfully treated by a combination of surgical drainage and antifungal treatment including three weeks of iv amphotericin b and three months of oral azoles . no relapse occurred on follow up for nine years . in summary , clinicians should be aware of possibility of c. gattii skin and soft tissue infection in tsunami survivors .

skin and soft tissue fungal infections with apophysomyces elegans , fusarium solani , cladophialophora bantiana have been reported in survivors from 2004 indian ocean tsunami . we report the first case of primary cutaneous cryptococcosis caused by cryptococcus gattii vgii in a tsunami survivor from thailand .

a 50-year - old female patient was referred to our department of oral medicine and radiodiagnosis with a complaint of a painless swelling over the right mandibular region since 3 months . intraoral examination revealed a swelling in relation to the right mandibular posterior region extending from the region of 45 to 48 posteriorly and left mandibular posterior region extending from 35 to 38 [ figures 1 and 2 ] . on palpation , a panoramic radiograph was taken to view the apparently isolated lesion on the mandibular right posterior region . the radiolucency observed in the region of right mandibular posteriors was multilocular with sclerotic borders extending posteriorly up to the anterior border of the ramus with the superior wall scalloping between the roots of 44 and 47 region . in addition to the suspected multilocular radiolucency , orthopantomographs revealed bountiful lesions ; multiple radiolucencies were noted in the anterior maxilla and lower left body of the mandible [ figure 3 ] . computed tomography ( ct ) scan revealed the bilateral involvement of the posterior aspect of mandible [ figure 4 ] and anterior aspect of maxilla [ figure 5 ] showing bicortical expansion in relation to right and left posterior mandible . a clinical diagnosis of the traumatic bone cyst with differential diagnosis of central giant cell granuloma and odontogenic keratocyst was rendered . intraoral examination revealing swelling in the right mandibular posterior region intraoral examination revealing swelling in the left mandibular posterior region orthopantomographs showing multiple radiolucencies computed tomography image showing mandibular lesion computed tomography image showing maxillary lesion patient was offered a choice of surgical exploration and biopsy . curettage of the cavities was done , and the obtained connective tissue material was sent for histopathological examination , which revealed densely inflamed connective tissue stroma with extravasated red blood cells , necrotic changes , and chronic inflammatory cells . surgical exploration of the lesion in right mandible surgical exploration of the lesion in left mandible surgical exploration of the lesion in maxilla patient made an unremarkable recovery after surgery . follow - up radiographs after a period of 6 months showed 90% of bone regeneration [ figure 9 ] . comparing our data with the literature review , an interesting finding of sbcs in an older age group that were not associated with cement - osseous dysplasia is rare , such variation in the clinical presentation of sbcs may be relevant to the better understanding of this underexplored lesion . these lesions showed radiographic characteristics that suggested differential diagnoses of ameloblastoma , odontogenic keratocyst tumor or a central giant cell lesion in view of their location . due to these radiographic characteristics , ct imaging is important for an accurate diagnosis , as this may avoid unnecessarily aggressive surgical approaches , like en bloc resections of the mandible . found that most recurrences were seen within 5 months of the surgical treatment , and this group recommended that follow - up should be continued until healing was confirmed radiographically within 3 years . in our sample , evidence of new bone formation after surgery was noticed within 3 months of the surgery and complete healing was noticed radiographically . the most common radiographic finding in multifocal sbcs was scalloping , consistent with what is traditionally described in single lesions . bony expansion and multilocular radiolucent patterns were observed . in general , sbcs have a tendency to grow along the long axis of the bone , causing minimal expansion ; however , expansion of the involved bone can occur and is more common with larger lesions . in general , the mandibular body is by far the most common location for sbcs , with the maxilla representing an uncommon site . one explanation for this lower prevalence in the maxilla could be related to the possibility that maxillary lesions are obscured by the overall radiolucent appearance imparted by the maxillary sinus . another possible explanation could be due to differences in the quality and amount of bone marrow and vascularity in the maxilla compared to the mandible . in this case , we noted that multiple sbcs in the mandibular body , as well as maxilla . curettage of the bone walls is the most widely accepted approach to the management of sbcs of the jaws , the goal being to promote the formation of a blood clot and subsequent bone repair . in addition , application of gelfoam , grafting of allogenic bone with platelet - rich plasma and intralesional injections of a mixture of blood , porous hydroxyapatite , and bone fragments have been reported to produce good results . in general , all maxillofacial lesions present with a standard set pattern of clinical or radiographic presentations . however , this case defies the regular standards set for a traumatic bone cyst . proper recording of case history , knowledge of symptomatology , and correct radiographic interpretation will be helpful for proper diagnosis and management .

simple bone cyst is an oft - described entity , which goes by many sobriquets and has been presented in the literature as early as 1926 . it is a lesion of unclear etiology with many proposed hypothesis for its pathogenesis and nonspecific histopathological presentation . we present a case of a solitary bone cyst , which clinically presented as a solitary lesion , but radiological examination revealed multiple lesions peppering the maxilla and the mandible , thereby highlighting the importance of atypical presentation and surprise lesions that may show up on radiological examination .

the hh - signaling pathway . ( a ) a diagram of the hh - signaling pathway , showing the site of action of the agonists ( green ) and antagonists ( red ) discussed in the text , as well as many additional factors that affect the pathway . abbreviations : ck1 , casein kinase 1 ; cos2 , costal2 ; dyrk1 , dual - specificity yak1-related kinase 1 ; gsk3 , glycogen synthase kinase 3 ; fu , fused ; gas1 , growth arrest specific 1 ; hh , hedgehog ; hip , hedgehog - interacting protein 1 ; rab23 , a rab - family ras - like gtpase associated with vesicle traffic ; ptc , patched1 ; pka , protein kinase a ; smo , smoothened ; sufu , suppressor of fused . ( b ) a schematic generalized view of the regulation of gli activator ( gli - act ) and gli repressor ( gli - rep ) forms by hh signaling . see [ 2 - 4 ] for further details . examples of diseases caused by loss of or ectopic function of the hh - gli signaling pathway , and the possible agents that could , in principle , be used as therapeutics * hh - ag is the hh agonist described by frank - kamenetsky et al ..

the discovery of small molecules that act as agonists and antagonists of the hedgehog - gli signaling pathway , which plays important roles in the embryo and adult , opens a new avenue for the treatment of diseases caused by aberrant suppression or activation of this complex pathway .

compared to advanced gastric cancer , early gastric cancer ( egc ) shows a favorable prognosis , with 5-year survival rates exceeding 94%.1 lymph node ( ln ) status is the most important prognostic factor . even in egc , the incidence of ln metastasis exceeds 10% ; is reported to be 14.1% overall and appears in 4.8 to 23.6% of cases depending on cancer depth.2 it is important to evaluate ln status preoperatively for proper treatment strategy ; however , sufficient results are not being obtained using various evaluation modalities . according to japanese staging guideline , n3 level node was regarded as distant metastasis.3 it is possible to cure local disease without distant metastasis by gastrectomy and ln dissection . however , there is no survival benefit from surgery for systemic disease with distant metastasis such as para - aortic ln metastasis.4 therefore , whether the disease is local or systemic is an important prognostic indicator for gastric cancer , and the debate continues over the importance of extended lymphadenectomy for gastric cancer . a 72-year - old woman , with no history of neurofibromatosis or other systemic disease , was referred to chosun university hospital ( gwangju , korea ) for egc . abdominal computed tomography did not show the gastric tumor lesion but revealed a well - defined , 1.6 cm sized ovoid retroperitoneal mass located posterior to the duodenum ( fig . an operator and scopist stood to the right side of the patient , and an assistant surgeon stood to the left of the patient . a total of 5 trocars ( two 12-mm trocars and three 5-mm trocars ) were used . the first dissection began with the mobilization of the duodenum from hepatoduodenal ligament and right gastrocolic ligament . an assistant retracted the duodenum left laterally , and an operator dissected between the pancreaticoduodenal unit and inferior vena cava ( ivc ) and right renal vein . 2 ) . several vessels from the mass to the ivc were ligated by the ligasure vessel sealing system ( valleylab , boulder , co , usa ) . the final pathological diagnosis was a gastric adenocarcinoma that invaded a mucosa ( t1a ) without nodal metastasis ( 0/20 , n0 ) and neurofibroma with immunohistochemical stains positive for s-100 protein ( fig . about 15% of egc have ln metastases.2 this makes the prognosis of egc better than advanced gastric cancer . nevertheless , while there is little chance of nodal or distant metastasis in egc , some case reports revealed stage iv egc accompanied by extensive ln and distant metastasis.5 - 8 accurate assessment of ln status is of crucial importance for appropriate treatment planning and determining prognosis in gastric cancer . but abdominal ultrasonography ( aus ) , endoscopic ultrasonography , multidetector - row computed tomography ( mdct ) , magnetic resonance imaging ( mri ) , and 18f - fluoro-2-deoxyglucose positron emission tomography can not reliably be used to confirm or exclude the presence of ln metastasis.9 in this case , preoperatively the patient was diagnosed as egc with distant metastasis by mdct . but a pathological report indicated mucosa - confined adenocarcinoma ( t1a ) without nodal metastasis ( n0 ) and solitary neurofibroma . typically , neurofibromas grow slowly and have minimal potential for malignant transformation unless they are associated with neurofibromatosis type 1 ( formerly known as von recklinghausen disease ) . specifically , solitary retroperitoneal neurofibromas were studied in several case reports.10 - 12 neurofibroma is more common in men , particularly in the 20~40 year age group.13 but in this case , patient was a 72-year old aged woman . retroperitoneal masses not arising from major solid organs are uncommon . because the treatment options vary , it is useful to be able to differentiate these masses by using imaging criteria.14 currently , imaging modalities such as mdct , aus and mri show progress technically . but there is still a considerable overlap of imaging findings for these masses , and histological examination is often required for definitive diagnosis.14 additionally , in gastrointestinal tract malignancy , a retroperitoneal mass is easily confused with ln metastasis . in conclusion , staging laparoscopy with frozen biopsy might be helpful in treating a preoperatively egc with distant metastasis .

neurofibromas are benign tumors that originate from the peripheral nerves , including neurites and fibroblasts . generally , a solitary neurofibroma is located in the skin and rarely in other places . a 72-year - old female suffered from epigastric discomfort for 2 months . endoscopic findings showed an early gastric cancer type iic at the antrum . abdominal computed tomography revealed early gastric cancer with a 1.6 cm - sized metastatic node posterior to the duodenum . laparoscopic assisted distal gastrectomy and retro - pancreatic dissection were performed uneventfully . histological examination revealed gastric adenocarcinoma , invading the mucosa without nodal metastasis , and a neurofibroma . herein , we present a case of a gastric cancer patient with a solitary retroperitoneal neurofibroma which mimicked a distant metastatic node .

the monoclonal anti - c5 antibody eculizumab is an inhibitor of the terminal complement complex formation , successfully tested in atypical haemolytic - uraemic syndrome ( ahus ) , with or without mutations in the regulatory proteins of the alternative pathway of the complement , and less convincingly in enterohaemorrhagic escherichia coli - associated hus [ 13 ] . several drugs , including mitomycin - c and interferon- , may also trigger hus , but the therapeutic impact of eculizumab in this setting remains elusive . here , we show that eculizumab may prove efficient in drug - induced hus unresponsive to plasma exchanges . a female patient developed severe hus during mitomycin - c treatment for relapsing metastatic breast cancer . enterohaemorrhagic hus was ruled out , serum levels of complement components c3 and c4 were normal and screening for mutations in the genes encoding complement regulatory proteins cfh , cfi , mcp , and anti - adamts13 or anti - cfh autoantibodies were negative . before genetic testing and report , all patients gave a written informed consent as recommended by french law . despite termination of drug exposure and daily plasma exchanges , the lack of improvement of haematological parameters and the requirement for dialysis ( see figure 1 ) prompted us to consider eculizumab therapy . following the first infusion ( 900 mg ) , we observed an early and dramatic improvement of haematological parameters with normalization of platelet counts and lactate dehydrogenase levels within 7 days . seven additional infusions of eculizumab were performed , allowing partial renal recovery at 3 months . renal function remained stable ( egfr : 20 ml / min/1.73 m ) and no relapse of hus occurred during 18 months of follow - up . fig . 1.response to eculizumab therapy in a patient with drug - induced haemolytic uraemic syndrome . shown are the data indicating a rapid biological and clinical improvement of thrombotic microangiopathy after administration of the monoclonal anti - c5 antibody eculizumab . lactate dehydrogenase level ( left y - axis ) , platelet count and plasma creatinine level ( right y - axis ) are represented by white triangle , cross and black triangles , respectively . are the data indicating a rapid biological and clinical improvement of thrombotic microangiopathy after administration of the monoclonal anti - c5 antibody eculizumab . lactate dehydrogenase level ( left y - axis ) , platelet count and plasma creatinine level ( right y - axis ) are represented by white triangle , cross and black triangles , respectively . the striking coincidence of eculizumab infusion and improvement of hus parameters suggest a beneficial role of transient complement blockade in this patient . the pathophysiology of drug exposure - related hus remains poorly elucidated . in mitomycin - c - related hus , circulating immune complexes that cause platelet aggregation , complement activation and subsequent endothelial damage have been reported , supplying a molecular explanation for the striking effect of eculizumab . since no change in plasma levels of c3 and c4 was detected , the rapid normalization of haemolysis parameters and platelet counts in both of the patients despite suspension of plasma exchanges might be related to local blockade of complement activation . thus , our data plead for a role of complement blockade in the management of refractory , drug - related hus .

the monoclonal anti - c5 antibody eculizumab has been successfully tested in atypical haemolytic - uraemic syndrome ( ahus ) , with or without mutations in the regulatory proteins of the alternative pathway of the complement , and less convincingly in enterohaemorrhagic escherichia coli - associated hus . here , we report a patient with mitomycin - c - induced hus unresponsive to plasma exchanges . eculizumab infusion was followed by a dramatic improvement of haematological parameters and renal function , suggesting a role of complement blockade in the management of refractory , drug - related hus .

an 18-year - male patient by name vinith came with a complaint of generalized spacing in his upper front teeth . on clinical examination , the case was diagnosed as angles class i molar relation on class i skeletal base with retained deciduous lateral incisor 52 , with missing right side lateral incisor and spacing in the upper anterior teeth and mild crowding in the lower teeth . the central incisor was drifted slightly toward unerupted lateral incisor and lack of space for the lateral incisor to erupt . on palpation , there were no signs of bulge either in the labial vestibule or on palatal vault . on radiographic examination , odontome and retained deciduous tooth was seen along with horizontally impacted lateral incisor [ figure 1 ] . the central incisor was shifted toward the right side and occupied half of the lateral incisor space . same side lingual , opposite side buccal technique with two intraoral periapical radiographs confirmed the presence of impacted lateral incisor on the palatal side . pretreatment osteoprotegerin and lateral cephalogram the treatment options available were extraction of impacted lateral incisor and going for a prosthetic implant by creating sufficient space for the implant placement and residual space closure with the help of fixed orthodontic treatment . second option was a fixed partial denture by closing the spaces in the upper front teeth , and the third option was to extract the deciduous tooth and bringing the impacted tooth into occlusion , but it is time - consuming . the patient was more interested in saving the tooth instead of going for artificial teeth . the treatment plan was to bring the impacted lateral incisor into occlusion and close the spaces in the upper arch . odontome and retained deciduous lateral incisor was surgically excised [ figures 2 and 3 ] . pretreatment intra oral view smile and buccal view showing missing lateral incisor an mclaughlin , bennett , terevisi fixed appliance with 0.022 slot was bonded , 0.016 niti was used for initial leveling and aligning . the central incisors were moved to the left side in order to prevent root resorption , as the lateral incisor was lying in closer proximity to the root of central incisor , and to regain the space for lateral incisor . space closure was done on 0.19 0.25 stainless steel wire , and to provide stability for the adjacent tooth while bringing the impacted tooth into occlusion . once the considerable amount of space obtained for the lateral incisor to bring into occlusion , under local anesthesia surgical exposure was done on palatal side [ figure 4 ] . after elevation of the mucoperiosteal flap , an adequate amount of bone was removed by creating a channel for the tooth to come out with the help of rotary cutting instruments . a lingual button was bonded onto the palatal aspect of the lateral incisor . ligature wire with bull eye holes were attached to the lingual button and tied to the main arch wire . surgical exposure of palatally placed impacted lateral incisor once wound got healed , 0.012 niti wire was used as piggyback wire and engaged it to the ligature wire and tightly tied to the rigid stainless steel main archwire . as niti has shape memory effect , it started moving the tooth into occlusion , this was seen periodically through radiographs . we have increased the force slightly in further visits by increasing the dimension of the wire to 0.014 and 0.016 niti . once the tooth was seen on the labial aspect , 0.016 0.22 tma wire was used with a coil to bring the tooth into occlusion . once the tooth came closer , 0.016 niti was placed directly into the bracket of lateral incisor and brought it into occlusion [ figure 5 ] . a panoramic radiograph shows the dilacerated lateral incisor with uprighted teeth and with proper space closure [ figure 6 ] . the stabilized teeth were retained with upper and lower fixed lingual retainer figure 5 . impacted lateral incisor into occlusion . impacted lateral incisor into occlusion posttreatment osteoprotegerin and lateral cephalogram the patient wanted to retain the tooth instead of going for extraction and was very much cooperative throughout the treatment . the lingual button was bonded onto the palatal side of the impacted tooth because the labial side was facing towards the nasal floor . many precautions were taken during bonding because the chances of debonding were high on the lingual aspect compared to the labial side , due to its irregular morphology . controlled amount of force was applied carefully with the help of ligature wire in every visit to prevent from debonding . as the tooth gets closer , the ligature wire was cut and the loop adjacent to the tooth was tied to the niti wire . complete space closure and a broad smile was achieved within a span of 18 months [ figure 7 ] . impacted teeth can be treated easily in the mixed dentition with the periodic extraction of retained deciduous tooth . if the impacted tooth was in favorable position and time was not a constraint impacted tooth can be brought into normal occlusion with orthodontic tooth movement .

impaction of maxillary lateral incisor with odontome and retained deciduous tooth is not often seen in regular dental practice . impaction of anterior teeth cause generalized spacing which affects the esthetics of the face . here we report a case of an 18-year - old patient with horizontally impacted dilacerated lateral incisor , which was bought into occlusion with the help of orthodontic tooth movement within a span of 18 months .

a 44-year - old male presented with complaints of pain and yellowish discharge in the right eye for one week . he suggested that he may have been infected by his family members or neighbors , all of whom had conjunctivitis . on examination , anterior segment examination of the right eye revealed localized conjunctival and episcleral congestion in the temporal quadrant with an adjacent area of conjunctival ulceration on the bulbar conjunctiva [ fig . 1 ] and the rest of the anterior segment and the posterior segment of the right eye were normal and so was the left eye . localized conjunctival and episcleral congestion with conjunctival ulceration on the bulbar conjunctiva in the right eye conjunctival scraping was obtained from the ulcer under topical anesthesia and was subjected to gram 's stain , koh preparation , and acid - fast stain . 2 ] , while both koh and acid - fast stain revealed no fungal or bacterial elements . gram 's stain showed the presence of gram - negative bacilli after 24 hours of incubation , confluent growth of gram - negative bacilli with smooth , glistening yellow colonies was observed on both blood [ fig . 3 ] and chocolate [ fig . the organism was identified as s. maltophilia ( > 100 cfu/100 l ) using mini api i d 32gn strips ( biomrieux , new delhi , india ) . this was also confirmed by negative oxidase , positive catalase , positive -glucose , positive -glucose , positive -maltose , positive -galactose , positive lipase , positive malonate , positive oxidative fermentative maltose , and negative oxidative fermentative mannitol tests . confluent growth of gram - negative bacilli with smooth , glistening yellow colony was observed on blood agar confluent growth of multiple colonies on chocolate agar susceptibility testing was performed against ampicillin / sulbactum , amoxicillin , amoxy / clavulanic acid , pipercillin , pipercillin / tazobactum , ceftizidime , cefazolin , amikacin , tobramycin , gentamicin , ofloxacin , norfloxacin , ciprofloxacin , gatifloxacin , lomefloxacin , moxifloxacin , chloramphenicol , and co - trimoxazole by the kirby the organism was resistant to aminoglycosides ( amikacin , tobramycin , and gentamicin ) and sensitive to quinolones ( ciprofloxacin , norfloxacin , ofloxacin , gatifloxacin , moxifloxacin , except lomefloxacin ) and co - trimoxazole . topical tobramycin was discontinued in view of aminoglycoside resistance , and ciprofloxacin eye ointment along with oral co - trimoxazole twice a day was added for a period of two weeks . s. maltophilia is an opportunistic ocular pathogen which is known to cause conjunctivitis , keratitis , and endophthalmitis . it was , however , sensitive to most quinolones and co - trimoxazole , and the patient responded well to treatment with both . reported that most infections caused by this organism occur in patients with ocular compromise , and the characteristically resistant antibiogram of s. maltophilia limits the therapeutic options . our case did not have any ocular compromise , but the antibiogram susceptibility showed similar results . timely identification of the organism is important in the management of ocular infections caused by s. maltophilia . das et al . have reported a successfully treated case of endogenous endophthalmitis due to s. maltophilia infection with antibiotic sensitivity - guided antibacterial therapy . stenotrophomonas infections , especially conjunctivitis / conjunctival ulcer , have a good prognosis if treated appropriately . to conclude , we report , to the best of our knowledge , the first case of conjunctival ulcer caused by s. maltophilia that resolved completely without any morbidity owing to early recognition and appropriate treatment . it would thus be prudent to keep in mind s. maltophilia as a possible pathogen in the etiology of conjunctival ulcer .

we report a case of unilateral conjunctival ulcer due to stenotrophomonas maltophilia infection in an immunocompetent individual . a 44-year - old male presented with complaints of pain and yellowish discharge in the right eye for one week . patient underwent complete ophthalmic evaluation and relevant laboratory investigations . anterior segment examination revealed localized conjunctival and episcleral congestion with conjunctival ulceration on the bulbar conjunctiva in the right eye . gram 's stain revealed gram - negative bacilli . culture and sensitivity revealed s. maltophilia and responded well to topical moxifloxacin with systemic co - trimoxazole therapy .

benign tumors of the hypopharynx are very rare , with the most common two being the fibrolipoma and leiomyoma . the ductal cysts are mucous retention cysts that develop after obstruction of the collecting ducts of the submucosal glands . although the oral cavity or larynx are often involved , hypopharyngeal localization is extremely rare . we report a new case of a ductal cyst that developed on the lateral wall of the pyriform sinus of the hypopharynx . there was a delay in the diagnosis because no mass - like lesion was found with routine fiberoptic laryngoscopic examination . a 64-year - old man presented with a four - year history of globus sensation . he was referred for intractable symptoms despite having taken reflux medication for two months . despite continued medication , his symptoms developed to dysphagia , even when swallowing liquid . he had experienced weight loss of 3 kg over the previous 2 months . on his first presentation , flexible fiberoptic laryngoscopy showed pooling saliva , but no mass - like lesion in the hypopharynx . an ovoid submucosal mass with a smooth contour in the left pyriform sinus was revealed ( fig . subsequently , computed tomography ( ct ) scans showed a 34 cm mass with central low attenuation in the left hypopharynx ( fig . it was removed by means of submucosal dissection with the microelectrode . the resected specimen consisted of a 234 cm cystic mass with proteinaceous fluid ( fig . histologic examination showed a true epithelial - lined cyst consisting of cuboidal epithelium ( fig . the final diagnosis , made by a pathologist , was a ductal cyst of the hypopharynx . reported that the ductal cyst is one of the two most common types of cysts in the larynx . pathologically , the epithelium - lined cyst wall consists of a uniform layer of cuboidal , columnar , or non - keratinizing squamous epithelium . ductal cysts usually are slow - growing and painless , appearing as a circumscribed and often fluctuant swelling . the presence or absence of symptoms in patients with benign pharyngeal masses , we missed the mass at the patient 's first visit . despite intractable globus symptoms , we could not find any specific abnormality in the larynx or hypopharynx by routine fiberoptic laryngoscopy . a number of techniques have been suggested to improve the view of the hypopharynx with flexible fiberoptic laryngoscopy . one such technique is letting the patient sustain high - pitch phonation or perform a modified valsalva maneuver . hillel and schwartz suggested performing a trumpet maneuver to fully visualize the pyriform sinus and the postcricoid region . in addition , purser and antippa reported that manual anterosuperior traction applied to the prelaryngeal skin results in a useful view of the hypopharynx . when nothing can be detected despite multiple laryngoscopic methods during outpatient examination in dysphagia patients , double contrast pharyngoesophagography may be useful for evaluating both the structure and the function of the hypopharynx and other organs involved in swallowing . benign lesions typically appear as small , round or ovoid , well - circumscribed , smooth - surfaced submucosal masses etched in white , best visualized on frontal views of the pharynx . ct or magnetic resonance imaging ( mri ) is useful in determining the extent and location of the cyst and distinguishing it from malignant tumors . most hypopharyngeal tumors are squamous cell carcinomas . therefore , hypopharyngeal masses should be differentiated from malignant tumors . therefore , we included the metastatic mass in the differential diagnosis before surgery . generally , there is no reason to resect small pharyngeal retention cysts that are not causing symptoms . the surgical removal of a small cyst is simple , and many of these cysts may be removed by endopharyngeal microsurgery using a direct laryngoscope . marsupialization may be adequate , and it may be necessary to reduce the size of a large cyst by puncturing and draining it before complete removal . we successfully removed the ductal cyst in our patient with a microdissection electrode under direct laryngoscopy . we recommend that , in any patient with a history of intractable globus symptoms , careful physical examination with a flexible laryngoscope and radiologic studies such as double contrast pharyngoesophagogram , ct , or mri should be performed to rule out other benign or malignant lesions .

the ductal cyst of the hypopharynx is a very rare tumor . we report a case of hypopharyngeal ductal cyst in a 63-year - old man presenting with globus sensation . it was removed by a laryngomicrosurgical technique , using a microdissection electrode . masses of the hypopharynx may not always be easily visible on routine examination of the hypopharynx with flexible fiberoptic laryngoscopes . particularly in cases of benign tumors , the diagnosis may be delayed due to a prolonged history of mild and subtle symptoms . we missed the hypopharyngeal mass at the initial presentation , but could detect the mass in the pyriform sinus with a double contrast barium swallow study . we describe the diagnostic method to detect hypopharyngeal tumors and the treatment of benign hypopharyngeal masses .

lap - protector was developed as a safe and useful device that may help to prevent infections and cancer cell contamination of minilaparotomy wounds . we applied this device to the gastric anterior wall of the incision in the first step of surgical transgastric cystogastrostomy for a patient with an infected pancreatic pseudocyst . recently , disposable circular stapler is very popular in digestive organ anastomosis . in 1999 , yunoki et al . a 69-year - old man presented to our hospital with a 5-day history of high fever , loss of appetite , and hiccups . laboratory data on arrival were as follows : white blood cell count , 24,800/l ; c - reactive protein , 13.8 mg / dl . computed tomography ( ct ) showed an infected pancreatic pseudocyst which compressed the gastric wall ( fig . we made a preoperative diagnosis of the recurrence of infected pancreatic pseudocyst ; therefore , emergency operation was performed . according to the jedlicka 's method , transgastric cystogastrostomy was selected . after performing minilaparotomy with upper median incision , a small incision with a length of 3.0 cm was made on the anterior gastric wall . placing the lap - protector over the incision , the bottom ring is pushed into the lumen of the stomach through the incision . the top and bottom rings of the lap - protector formed a sandwich around the anterior gastric wall ( fig . the posterior gastric wall was punctured toward the retrogastric space by a sheath of needle . a small incision was made on the posterior wall to reach the abscess wall ( fig . while preventing pollution in the peritoneal cavity , the abscess space was irrigated with saline . using an auto suture pceea 25 , an anastomosis was approximated between the abscess wall and the posterior gastric wall ( fig . four days after the operation , upper gastrointestinal examination and ct using a water - soluble contrast agent showed no anastomotic leakage and complete drainage ( fig . two months after discharge , follow - up evaluation with upper endoscopy and ct revealed no sign of recurrence ( fig . a variety of treatments in infectious pancreatic pseudocyst have been reported , such as percutaneous drainage , endoscopic cystogastrostomy , and conventional surgical cystogastrostomy . however , we believe that surgical cystogastrostomy still plays an important role in the management of selected cases of pseudocyst of the pancreas . in this case , we performed cystogastrostomy easily without any contamination using lap - protector and circular stapler .

lap - protector , which is an abdominal wall sealing device , is usually used for wound protection from implantation of malignant cells or pyogenic fluid . a circular stapler is a common easy - to - use device for anastomosis of the digestive tract . we report the case of an infected pancreatic pseudocyst which was treated by surgical procedure using these useful devices . a 69-year - old man was followed up in our hospital after severe acute pancreatitis . he had undergone drainage surgeries twice for intractable pancreatic abscess followed by severe acute pancreatitis . he was admitted to our hospital complaining of loss of appetite , hiccups , and high fever . computed tomography of the abdomen revealed an infected pancreatic pseudocyst which compressed the gastric wall . internal drainage into the stomach was performed using lap - protector and circular stapler . the patient recovered uneventfully . recently many endoscopic or laparoscopic procedures in cystogastrostomy are reported ; however , a conventional open surgical approach is also important . this easy method may be useful for operative cystogastrostomy .

a 55-year - old woman suddenly developed motor weakness of the right side and dysarthria 1 day prior to admission . on admission she had mild dysarthria , especially of the lingual components , hemiparesis of the right side ( motor grade iii ) , and right facial palsy . diffusion- and t2-weighted mr images obtained the same day revealed high signal intensities in the left pontine regions , consistent with an ischemic lesion ( fig . 1-a ) . the source images for both contrast - enhanced mr imaging and three - dimensional ( 3-d ) mr angiography displayed narrow segments of the basilar artery and double - lumen signs ( fig . these mr findings were consistent with a long intimal dissection or fenestration of the basilar artery . using virtual endoscopy ( rapidia ; infinitt , seoul , republic of korea ) , a diagnosis of an extreme fenestration because of the complete proximal and distal fusion shown by virtual endoscopy , we speculated that this finding was a basilar arterial fenestration of the true duplication type . two weeks after the onset of symptoms , hemiparesis of the right side improved ( motor grade v- ) and she was discharged . the reported incidence of fenestration of the basilar artery has varied from 0.3% in angiographic examinations to 5.26% in autopsy series.6,7 basilar artery fenestration is the most commonly observed form of fenestration of the cerebral arteries.7 the fenestration is usually short and occurs primarily in the proximal segment of the artery,8 with its clinical interest attributable to the possible association with aneurysms localized at the junctions of the fenestrated segments.9 there is known to be a defect in the media of the arterial wall that has been thought to be the predisposing factor for the prevalence of saccular aneurysms together with turbulent blood flow at the joining points.10 in the present case , the lesion was a true duplicationt - type fenestration , with each lumen sharing the medial wall . this finding was consistently observed from the joining portion of the two vertebral arteries to the top of the basilar artery , and each of the posterior cerebral arteries was branched from the adjoined portion of the basilar artery . there has been speculation about associations between either vertebrobasilar artery fenestrations and brainstem ischemia,10 or infarctions,5,11,12 although their relationships are controversial . among these reports , some authors showed basilar artery fenestration and associated brainstem infarctions.5,11 one study showed an arterial slit - type fenestration that was confirmed by virtual arterial endoscopy,11 and another study showed a true duplication - type that was confirmed by autopsy.5 the latter case was a true duplication - type , nonseparated form of fenestration that shared the medial wall that was confined to the caudal half portion of the basilar artery . the autopsy findings showed a partially organized thrombus that occluded both halves of the duplicated portion , suggesting possible hemodynamic disturbances and turbulent blood flow at the site of fenestration . another case of extreme fenestration of the basilar artery was reported in a retrospective mr angiographic study.4 in that study , 10 basilar artery fenestrations were observed from 600 brain reviews ( 1.7% ) and one case showed a total duplication . the shape of the fenestration was different in the proximal and distal parts of the basilar artery ; in the proximal two - thirds of the artery , a true duplication - type fenestration that shared the medial wall was noted , but in the distal one - third of the artery , the artery was fully separated , with each basilar artery terminating in a posterior cerebral artery . however , it was not mentioned whether this finding was combined with a brainstem infarction . because of the ' double - lumen sign ' in the present case , a basilar artery fenestration might be misinterpreted because it is known as a pathognomonic sign of arterial dissection.13 even though neuroimaging has improved in both resolution and 3-d capabilities , it remains difficult to diagnose fenestrations of intracranial arteries . this is consistent with the large discrepancy in the reported incidence of fenestration of the basilar artery between angiographic examination and autopsies.6,7 virtual endoscopy is a new diagnostic method that uses computer processing of 3-d image datasets , such as ct or mr imaging scans , to provide simulated visualization of specific organs.14 the ability to obtain an internal view of a vessel facilitates qualitative analyses and could improve the pretherapeutic visualization of vascular variants , stenosis , and obstruction of cerebral vessels.15 virtual endoscopy in patients with fenestration , as in the present case , has both improved the anatomical identification of vessels and prevented misdiagnoses of fenestration as either dissection or thrombus . in summary , we have reported a case of a patient with a duplication - type , nonseparated fenestration of the basilar artery and associated brainstem infarction . even though a causative linkage between arterial fenestration and cerebral ischemia was not established , a reliable diagnosis of the vascular condition in the affected lesion was important clinically . from this point of view , virtual arterial endoscopy may enhance diagnostic capabilities , especially in certain cases .

we present a case of a 55-year - old woman with pontine infarction and fenestration of the basilar artery that was demonstrated using virtual endoscopy . the patient had motor weakness of the right side with sudden onset . diffusion- and t2-weighted mr images revealed high signal intensities in the left pontine regions , and an mr angiographic examination showed the double - lumen sign of the basilar artery . an extensive duplication - type , nonseparated fenestration of the basilar artery was diagnosed by virtual endoscopy and craniocaudal communications with discrete openings were observed . we report a case of a patient with basilar artery fenestration and associated brainstem infarction .

in the 1970s , max cooper , dennis osmond , and others discovered that immunoglobulin ( ig)-bearing b cells arose from ig - negative precursor cells in the bone marrow . these precursor cells dubbed pre - b cells by cooper had synthesized part of the receptor ( ig heavy chains ) , which could be detected in the cytoplasm , but had yet to assemble a complete receptor on the cell surface ( 1 ) . osmond later pinpointed a more primitive population of cells ( pro - b cells ) that had not yet synthesized heavy chains ( 2 ) . but the cellular phenotypes that might be lurking between the pro - b and pre - b cell stages remained mysterious , as there was no way to pick apart this mixed bag of cells . as a result , many of the early insights into b cell development including the ordered rearrangement of the v , d , and j gene segments of the ig locus came from studies using transformed b cell lines . the discovery that stromal cells could support the growth and differentiation of primary b cell precursors soon overcame this obstacle ( 3 , 4 ) . then came multicolor ( multilaser ) flow cytometry , a technique that revolutionized the study of b cell development ( and immunology as a whole ) by allowing single cells to be stained simultaneously with multiple fluorescently tagged mabs . hardy a chemist by training used one of the first multilaser facs ( fluorescence activated cell sorter ) machines to investigate mature b cell subpopulations as a post - doc with leonore and leonard herzenberg in the early 1970s ( 5 , 6 ) . with these techniques in hand , plus a battery of lymphocyte - specific mabs ( generated by the herzenbergs , john kemp , and others ) , hardy by then a new faculty member at fox chase cancer center ( philadelphia)set out to explore the diversity of b cell precursors . he started by dividing b220 bone marrow cells based on their expression of cd43 , bp-1 and cd24 . sorting the cd43 populations of cells and culturing them in vitro revealed their order of differentiation : fraction a ( bp-1/cd24 ) begot fraction b ( bp-1/cd24 ) , which in turn begot fraction c ( bp-1/cd24 ) . later ( cd43 ) populations could be distinguished by their expression of surface igm and igd . it was so striking the way you could sort these cells and see the progression , says hardy . hardy confirmed the developmental order of the cells by amplifying the ig genes from each fraction . the ig genes from fraction a cells were in a germline configuration , whereas those from fractions b and c had d - j , but not v - d - j rearrangements , consistent with previous studies . hardy published these data , along with his fractionation scheme , in 1991 in the journal of experimental medicine ( 7 ) . hardy was not alone in his attempt to bring order to early b cell development . fritz melchers and colleagues in basel , switzerland , who discovered the surrogate light chain , added c - kit and cd25 to the growing list of b cell markers ( 8) . that list now goes on and on ( and facs is up to 17 colors ) , with each new marker revealing new layers of diversity . as melchers puts it , it 's a mess , but it 's a beautiful mess . hardy prefers to call it a beautiful complexity , adding , it 's like a fractal the deeper you look , the more you see .

in the early 1990s , richard ( randy ) hardy and colleagues divided b cell precursors into subpopulations the hardy fractions based on the cells ' expression of various cell surface proteins . this classification helped lay the groundwork for our present - day understanding of the molecular events that control early b cell development .