Rapunzel syndrome is a rare type of trichobezoar with an extension of the hair into the small bowel. Clinical presentation is deceptive and vague ranging from abdominal mass to gastrointestinal symptoms.We present a 7 years old girl with Rapunzel syndrome, where the trichobezoar was not suspected at all especially with negative history of trichophagia. In majority of the cases the diagnosis was made very late in the history of the disease, at a stage where surgery is the only cure for this syndrome.In the paediatric age group with a long history of gastrointestinal symptom, endoscopy is a diagnostic as well as a therapeutic modality and may reduce surgery in trichobezoars. Bezoar is a tightly packed collection of undigested material that is unable to exit the stomach, Most bezoars are of indigestible organic matter such as hair-trichobezoars; or vegetable and fruit the – phytobezoars; or a combination of both but other rare substances has been also been described in literature. Trichobezoars, commonly occur in patients with psychiatric disturbances who chew and swallow their own hair. Only 50% will have history of trichophagia. Trichobezoars have been described in literature and they comprise 55% of all bezoars. In very rare cases the Rapunzel Syndrome hair extends through the pylorus into the small bowel causing symptom and sign of partial or complete gastric outlet obstruction .7 year old girl was referred to our paediatric clinic in 2007, with a history of abdominal pain, distension, weight loss and attacks of vomiting which was not persistent but it comes after a meal and fluid intake. This history was on and off for almost one year. She was treated in different primary health care as a case of gastroenteritis and parasitic infestation. Despite the treatment she was receiving there was no improvement in her condition, in fact she was getting worse.In the paediatric clinic, abdominal palpation revealed an oblong mobile well-defined mass occupying the upper half of the abdomen, the mass was not tender and was firm in consistency. Upper Gastrointestinal endoscopy revealed a Trichobezoar occupying almost the whole gastric cavity, an attempt to remove it by foreign body forceps failed and the forceps was barley pulling few fibres of this huge ball of hair.The patient was referred to Surgery, and through upper mid line incision the stomach was opened (Gastrotomy) between two Vicryl stay suture. A huge Trichobezoar was identified which took the shape of the stomach Figure , 2. TherThe common presentation of trichobezoar is in young females usually with an underlying psychiatric disorder. In our case the presentation is in a very young age with hair extending down to the small bowel, causing symptoms, which could mimic gastrointestinal infections and infestation especially in endemic areas. Rapunzel Syndrome is a rare form of trichobezoar. It is named after a tale written in 1812 by the Brothers Grimm about a young maiden, Rapunzel, with long hair who lowered her hair to the ground from a castle, which was a prison tower to permit her young prince to climb up to her window and rescue her.In recent years more and more cases has been described . This syMajority of cases of trichobezoar present late, due to the low index of suspicion by the physician. Of 131 collected cases of trichobezoar, a palpable abdominal mass was present in (87.7%), abdominal pain (70.2%), nausea and vomiling 64.9%), weakness and weight loss (38.1%), constipation or diarrhoea (32%) and haematemesis (6.1%). The laboratory investigations revealed low haemogiobin in about 62% (average) [.9%, weakThe complications of Rapunzel syndrome ranges from attacks of incomplete pyloric obstruction to complete obstruction of the bowel to perforation to peritonitis and mortality -7.Patient with Trichotillomania , around 30% will engage in trichophagia, and of these, only 1% will go on to eat their hair to the extent requiring surgical removal . Less thEndoscopy is diagnostic, in almost all cases while Ultrasound has not much to offer as a diagnostic tool. CT scan with contrast will delineate the extension of trichobezoar.In the early stages endoscopic removal is not with out risk of bowel perforation and should be resolved for small Trichobezoars only . Other mA long history of gastrointestinal problem, in a pediatric age group with history of trichophagia, early endoscopy is recommended. All patients with Trichobezoar should be referred for psychiatric evaluation after surgery to avoid recurrence.The authors declare that they have no competing interests.Written informed consent was obtained from the parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors were involved in patient management or writing of the manuscript.AW and JS performed the surgery and the follow up of the patient, HK diagnosed the case, did endoscopy and refered the patient for surgery. AS did the workup preoperatively and was the Major contributor in writing the manuscript. All authors read and approved the final manuscript.


Desmoid tumors are soft-tissue neoplasms arising from fascial or musculo-aponeurotic structures. Most reported thoracic desmoid tumors originate from the chest wall. However, intrathoracic desmoid tumors are rare. We present a case of a 35-year-old male patient complaining of mild shortness of breath. The patient was diagnosed to have a huge intrathoracic desmoid tumor, which was successfully resected. Desmoid tumors are soft-tissue neoplasms arising from fascial or musculo-aponeurotic structures.2 Most reA 35-year-old male patient was admitted to the emergency room complaining of mild shortness of breath of 12 weeks’ duration. He had history of road traffic accident about 1 year back, with unilateral lower limb fracture. Otherwise, the history was unremarkable.Clinical examination of the patient showed stable vital signs, one small left supraclavicular lymph node, trachea shifted to the right and decreased air entry on the left side with dull percussion note; otherwise, no abnormality was detected. Blood work-up did not show significant changes. Chest x-ray was done, and it showed homogenous opacity on left hemi-thorax and shifting of the mediastinum to the right. So, CT scan of the chest was done, and it revealed huge left-side intrathoracic mass pushing the mediastinum to the right side, with complete collapse of the left lung . Fine-neThe patient was prepared for surgery, posterolateral thoracotomy was done and about 20 × 15 × 20 cm lobulated intrathoracic tumor was found. The tumor was attached to the apicolateral portion of the chest wall near the brachial plexus; so we excised the tumor completely except a very small part which was adherent to the area of brachial plexus, and decortication of the left lung was done . The hisJohn Macfarlane first described desmoid tumors in 1832. The over6They most often arise from the musculo-aponeurosis of the abdominal wall. Common s1014Several pathophysiological hypotheses are suggested. Abnormal62216Complete resection of the tumor with a clear surgical margin is currently the mainstay of curative treatment for desmoid tumors. The recurrence rate is high and varies from 29% to 54% in some reported series.


We present a case of fibrovascular polyp, a rare submucosal tumour of the oesophagus that has been reported only sporadically in the literature. The biapproach for surgical removal of fibrovascular polyp has only been mentioned once in the literature.A 65-year-old Greek man presented with a 9-month history of gradually progressive intermittent dysphagia. Radiologic work-up with oesophagogram and computed tomography revealed a large, sausage-shaped intraluminal polyp extending from the level of the cervical oesophagus to the level of the upper body of the stomach. The diagnosis of giant fibrovascular polyp was made radiographically and confirmed by endoscopic biopsy. The polyp was removed using a biapproach surgical technique: pharyngotomy and subsequent gastrostomy.Fibrovascular polyp is a rare submucosal tumour. Proper treatment depends on accurate assessment of the origin, size, and vascularity of the pedicle and the size of the tumour. Choice of the appropriate surgical approach depends on the correct diagnosis, which can usually be indicated radiographically by the presence of a smooth, sausage-shaped defect with a discrete bulbous tip. A fibrovascular polyp (FVP) is a rare, benign, intraluminal, submucosal tumour-like lesion, characterised by the development of pedunculated, intraluminal masses that can exhibit enormous intraluminal growth. These lesions are composed of loose or dense fibrous tissue, adipose tissue, and vascular structures and are covered by normal squamous epithelium. The most common location is the upper third of the oesophagus, near the cricopharyngeus. Dysphagia, vomiting, weight loss, and respiratory symptoms are the most frequent complaints. However, long pedunculated lesions can regurgitate into the pharynx or mouth and cause death from asphyxiation if the larynx is occluded . We presA 65-year-old Greek man presented with a 9-month history of gradually progressive intermittent dysphagia. He also reported significant weight loss but no haemetemesis or melaena. The rest of his medical history was not significant. No specific abnormality was revealed during the physical examination.Radiologic work-up with oesophagogram showed a dilated oesophagus, air bubbles with a mottled appearance, and contrast-filling defects from the cervical oesophagus to the upper body of the stomach Figure . The comCT with multiplanar reformatting provided valuable information regarding the location of the lesion as well as the size and anatomical attachment proximally, which was pivotal in directing surgery Figure . The intA biapproach surgical technique was selected. The origin of the pedicle was attached to the anterior wall of the hypopharynx. The broad base of the stalk was divided by performing cervical vertical oesophagostomy. However, the head of the polyp was too large to be removed through pharyngotomy; therefore, the entire polyp was removed via gastrostomy Figure . The lenFVPs are rare submucosal tumours of the oesophagus almost always originating from the cervical oesophagus; they are benign but potentially life-threatening lesions. In the past, these lesions have been variably classified as 'lipomas', 'fibromas', and 'fibrolipomatous' polyps -5. They A FVP usually presents as a large, pedunculated lesion, and symptoms occur only once the polyp has become sufficiently large. Patients usually complain of dysphagia, substernal discomfort, and the sensation of a mass. Many cases of FVP have presented as regurgitated masses in the mouth, others have led to airway obstruction when the mass impacted on the larynx. Asphyxiation can result from impaction of the polyp in the glottis and is the most feared complication . HistoloUnless regurgitated, the presence of FVP can be difficult to diagnose, and patients may die without a correct diagnosis. FVPs can sometimes be identified during chest radiography by the presence of a right-sided superior mediastinal mass, anterior tracheal bowing, or both. At oesophagography, the lesion usually appears as a smooth, expansile intraluminal mass that arises in the cervical oesophagus and extends into the thoracic oesophagus. On CT scan, FVPs containing abundant adipose tissue may appear as soft-tissue-attenuated lesions (abundant fibrovascular tissue), with a paucity of fat, that expand the lumen of the oesophagus.Although most FVPs have an attachment site in the cervical oesophagus, barium studies often fail to demonstrate a proximal pedicle. Accurate diagnosis is best established with endoscopy, although this technique is not completely reliable for diagnosing FVP. The differential diagnosis for FVP includes achalasia, extrinsic masses that compress the oesophagus, giant coalescent air bubbles, and other polypoid intraluminal tumours including lymphoma, spindle cell carcinoma, malignant melanoma, and leiomyosarcoma. The correct diagnosis can usually be suggested radiographically by the presence of a smooth, sausage-shaped defect with a discrete bulbous tip .Removal of these lesions is usually recommended because of the progressive and eventually debilitating nature of the symptoms and the small but known risk of asphyxiation and sudden death. The most common therapeutic approach is surgical cervical oesophagostomy with complete excision of the stalk. The location of the stalk and the vascularity makes surgical resection the preferred mode for removing these unusual polyps. Endoscopic resection is possible but is generally avoided because of the potential for haemorrhage from the feeding vessels in the stalk. However, complication-free endoscopic resection of large oesophageal giant FVPs has been reported.Generally, small polyps less than 2 cm in diameter and with a thin pedicle can be removed by endoscopic ligation and electrocoagulation of the pedicle. Polyps larger than 8 cm long or those with a thick, richly vascularised pedicle should be removed by surgical excision, and usually through a cervical incision. When the head of the polyp is too large to be removed through pharyngotomy, removal via gastrostomy is recommended. We could identify only one report in the literature about the surgical resection of FVP using the biapproach . A biappGiant FVP of the oesophagus is a very rare entity and few reports on this lesion exist in the literature. Diagnosis can be difficult for physicians who are unfamiliar with this type of tumour. The most common location is the upper third of the oesophagus, near the cricopharyngeus. Dysphagia, vomiting, weight loss, and respiratory symptoms are the most frequent complaints. However, long pedunculated lesions can regurgitate into the pharynx or mouth and cause death from asphyxiation if the larynx is occluded. The details of our case should raise awareness for both radiologists and clinical physicians. Oesophagogram and CT are essential when evaluating a patient with such symptoms.The authors declare that they have no competing interests.DC analysed and interpreted the patient data and was a major contributor in writing the manuscript. AD analysed the patient data and contributed in writing the manuscript. Both authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


The association of paraneoplastic hypoglycemia [Doege-Potter syndrome] and finger clubbing [Pierre-Marie-Bamberg syndrome] with pleural solitary fibrous tumour is rare. We present a previously unpublished but typical example of this rare occurrence together with a detailed updated literature review of previously published cases of pleural SFT discussing the histopathology of SFT; pathophysiology of the hypoglycemia and finger clubbing; treatment and outcome of pleural SFT. The patient, a 57-year-old African male was admitted at our hospital with recurrent episodes of hypoglycemia. He was found to have digital clubbing and decreased breath sounds in the right lower chest but no other significant clinical findings. His insulin level measured during an episode of hypoglycemia was undetectable. Chest radiograph and CT-scan revealed a lobulated mass in the right chest which was diagnosed to be SFT on histology. Surgical excision of the mass resulted in cure of the hypoglycemic episodes and rapid regression of the clubbing. Less than 65 cases of pleural SFT manifesting with hypoglycemia with or without finger-clubbing have been published in the English literature. The mean diameter of these tumours manifesting with hypoglycemia is 20 cm, 54% being benign while 42% were malignant. They predominantly present in the 6th-8th decade, average age of 64 years and a slight male preponderance at 58%. Complete surgical resection remains the most important predictor of clinical outcome in terms of recurrence and metastases, while providing instant cure for the hypoglycemia and rapid resolution of the finger clubbing. The occurrence of hypoglycemia with an intrathoracic tumour was first reported by Doege and Potter independently in 1930 hence the eponym Doege-Potter syndrome [DPS] . HyperthA 57-year-old male was referred to the Chris Hani Baragwanath Hospital with recurrent episodes of symptoms of severe hypoglycemia including syncope. He had a 20 pack-year history of smoking. He was found on examination to have marked finger-clubbing and decreased breath sounds in the right lower chest, but no other significant clinical findings. Sputum cytology was negative for malignant cells. His blood glucose levels recorded over three consecutive days whilst remained low despite receiving continuous intravenous infusion of 10% dextrose table .A percutaneous needle biopsy of the mass showed morphological and immunohistochemical features of a benign SFT. The patient underwent thoracotomy with excision of the bulk of the mass. A small remnant attached to the diaphragm could not be removed. The tumour was large, well-circumscribed and encapsulated, with a tan-coloured whorled cut surface figure . It weigMicroscopic examination of the tumour revealed a bland spindle cell proliferation arranged in short intersecting fascicles with a patternless architecture, and hypo- and hypercellular areas separated by dense collagenous fibrous stroma figure . There wThe episodes of hypoglycemia abated postoperatively and the digital clubbing resolved within three months.et al which had analysed 360 cases in the literature and 8 additional cases from their series , high cellularity, nuclear pleomorphism and necrosis ,42. HoweHypoglycemia is rare in pleural SFT, occurring in approximately 5% of cases ,51. DoegParaneoplastic hypoglycemia results from secretion of an unprocessed or incomplete high molecular weight [HMW] form of insulin-like growth factor type II [IGF-II] . This HMRecently it has been shown that SFT cells have a markedly greater expression of IGF-II mRNA and a lesser expression of pro-hormone convertase 4 [PC 4] mRNA in the tumour tissue compared to normal placental tissue, raising the possibility that defective PC4 gene expression in these tumours may underlie the impaired processing of IGF-II . Pro-horThe detection of HMW IGF-II requires immunoblot analysis to distinguish it from normal IGF-II; unfortunately this technology was not available to us at the time and there was no pre-operative blood specimen preserved to enable IGF-II determination. Nevertheless all the features in this case fit with the diagnosis of DPS including severe intractable hypoglycemia with suppressed insulin secretion, decreased C-peptide and immediate resolution of the hypoglycemia following excision of the tumour.et al reported hypoglycemia in 12 out of 223 cases [5.2%] from a multi-institutional case series [3. Rena et al reported hypoglycemia in 3 out 21 cases [14.3%] [et al found only one case of hypoglycemia from a series of 60 cases [1.7%] [et al found one out of 14 cases [7.1%] in their series which at 33 cm also happened to be the largest tumour in that series  survival rates for pleural SFT vary from 75% to 100% in various series ,13,39,72Pleural SFT is an uncommon but not rare tumour that is now known to be of mesenchymal fibroblastic rather than mesothelial origin. Paraneoplastic manifestation of hypoglycemia and finger-clubbing in pleural SFT are exceedingly rare yet important clinical features in terms of diagnosis and follow-up. Hypoglycemia with or without finger-clubbing has been recorded in <80 cases in the English Literature. Both hypoglycemia and finger-clubbing resolve following excision of the tumour. In this updated review, hypoglycemia with and without finger-clubbing was found to associated with large tumours [>10 cm], but does not appear to be significantly associated with histological features of malignancy. The hypoglycemia is caused by HMW IGF-II which results from defective enzymes due to PC4 gene abnormalities in the tumour cells. The finger-clubbing results from abnormal hyaluronic acid deposition due to VEGF and PDGF cytokine expression. Histological features for malignancy and size, though important, are not truly predictive of local recurrence or worse clinical outcome. Complete surgical excision of the tumour remains the best treatment and most important predictor of clinical outcome. Long-term follow-up is recommended due to varyingly high recurrence rates particularly for tumours that were difficult to completely excise and have histological features for aggressive or malignant behaviour.CT: Computed tomography; DPS: Doege-Potter syndrome; IGF: Insulin-like growth factor; HMW: High molecular weight; HOA: Hypertrophic osteoarthropathy; MSA: Muscle-specific acting, PDGF: Platelet-derived growth factor; PMBS: Pierre Marie-Bamberg syndrome; SFT: Solitary fibrous tumour; SMA: Smooth muscle acting; VEGF: Vascular endothelial growth factor; WHO: World Health Organisation.The authors declare that they have no competing interests.All authors helped to draft the manuscript, read and approved the final manuscript. AK, MJH and JM were involved in making the pathological diagnosis. MW and TH were involved in the case report, clinical workup and management of the patient. AK and JM did the literature search and review. AK coordinated the drafting of the manuscript and its preparation for publication.Written informed consent was obtained through the University of the Witwatersrand Ethics Committee for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Tumor rupture and pulmonary metastasis in patients with hepatocellular carcinoma are both associated with poor prognosis and treatment strategies are controversial.Here we report a 50-year-old man with survival of over 90 months after undergoing an extended right lobectomy for a ruptured hepatocellular carcinoma and then repeated resections for pulmonary metastasis during the followup period.This case report shows that surgical resection can be an effective treatment for patients with both ruptured hepatocellular carcinoma and pulmonary recurrences. Hepatocellular carcinoma (HCC) is the most common primary hepatic tumor and one of the most common cancers worldwide. Spontaneous rupture is a life-threatening complication of HCC. The overall incidence of spontaneous rupture of HCC varies from 5% to 26%, with a mortality rate of up to 67%, especially in patients with poor liver function -4.The treatment of a ruptured HCC is controversial. Previous studies have suggested that emergency liver resection is feasible in patients with a small tumor and satisfactory liver function (Child-Pugh A or B grade). Surgical resection is currently the only way to achieve long-term survival .The lung is the most common site of metastasis in patients with HCC. These are often unresectable as most pulmonary metastases are multiple . Neverth3 mass in Couinaud segments 7 and 8 and ascites accumulation  on admission showed a 12 × 12.5 × 5 cm2) and cholecystectomy, on 26 February 2000. The tumor was resected with margins greater than or equal to 1 cm. Six hundred cubic centimetres of bloody ascites were removed during surgery.The patient underwent a right extended lobectomy, with partial resection of the diaphragm (about 4 × 4 cmHistopathological examination revealed a poorly differentiated HCC Figure . Direct The patient's postoperative course was smooth and he was discharged on 13 March 2000. After surgery, he had regular followups with serum alpha-fetoprotein levels, chest radiographs, and abdominal ultrasonography every three months in our hospital. A small intrahepatic recurrence was found and treated twice by ultrasound-guided percutaneous alcohol injection.3. Histopathological examination confirmed the presence of metastatic HCC (Figure The patient presented with mild hemoptysis 30 months after the hepatectomy, and follow-up chest CT demonstrated a metastasis in the right lower lobe the of lungs. The metastasis was removed with wedge resection measuring 2 × 1 × 1 cmC Figure .A follow-up chest radiograph and CT revealed a 2.9 cm solitary metastasis in the right upper lobe of the lung without any sign of liver recurrence 52 months after hepatectomy. A wedge resection of the right upper lobe of the lung was performed through a thoracotomy and the tumor was confirmed as a metastasis of HCC.A further follow-up chest CT scan disclosed tumor recurrence in the right upper lobe of the lung and pleural seeding 80 months after hepatectomy Figure . The serNo intrahepatic recurrence of HCC was found after the last percutaneous alcohol injection of the liver and the patient has remained disease-free for 10 months since the last resection of pulmonary metastases, i.e. 90 months after the initial hepatectomy for a ruptured HCC.The exact mechanism of spontaneous HCC rupture is still unknown although hypotheses include rapid growth of tumor with necrosis, erosion of a vessel, occlusion of the hepatic veins by a tumor thrombus, and coagulopathy . The proIn our patient, HCC with direct invasion to the diaphragm was seen and was resected with clear margins, and intrahepatic recurrence was controlled by local ablation. Pulmonary metastases were diagnosed 30, 52, and 80 months after hepatectomy and were removed by repeated wedge resections.The lung is the most common site for extrahepatic spread of HCC and leads to a poor prognosis . Some auSome reports support the effectiveness of pulmonary resection of metastases from HCC. Tomimaru et al.  reportedFactors for good prognosis after pulmonary metastasectomy of HCC include a patient's disease-free interval greater than 12 months and alpha-fetoprotein levels less than 500 ng/ml . For ourFew reports described long-term survival after resection of pulmonary metastases from HCC . Our casThis case report suggests that surgical resection can be an effective treatment for patients with both ruptured HCC and pulmonary recurrences. When intrahepatic recurrences are not present, a previous episode of HCC rupture is not a contraindication for pulmonary metastasectomy in patients with HCC.CT: computed tomography; HCC: hepatocellular carcinoma.The authors declare that they have no competing interests.KLS and YYC examined the patient, reviewed the literature, and wrote the manuscript. The manuscript was reviewed and edited by YYC and MSS. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Extremity liposarcomas can metastasize to different areas of the body but have rarely been demonstrated to metastasize to the liver. Due to the unusual occurrence of isolated metastatic extremity liposarcoma to the liver, the optimal treatment of this condition is unknown.Less than one year after resection of a myxoid/round cell liposarcoma of the left lateral calf, a 61-year-old male presented with a CT scan showing a 2 cm low-density lesion in the right lobe of the liver. The lesion tripled in size over the next few months. An extensive evaluation revealed isolated disease to the liver. The lesion was surgically removed with a right hepatic lobectomy and the pathology was consistent with metastatic myxoid/round cell liposarcoma.Although extremity liposarcoma rarely metastasizes solely to the liver, the best chance at cure is with complete resection. Unfortunately, cure rates are very low in the setting of metastatic disease. As expected, the patient experienced progression of disease at sites outside of the liver 5 months after the liver resection. Extremity soft tissue sarcomas are rare mesenchymal tumors with 3,900 new cases being diagnosed each year and are typically malignant fibrohistiocytoma, liposarcoma, or synovial sarcomas. Myxoid liposarcomas make up 38% of liposarcomas with round cell tumors 11%) and mixed lesions (8%) less commonly present 1% and mi.Prognostic factors that impact on survival include histological grade and size . The hisManagement of metastatic disease is a difficult problem with no clear consensus. Chemotherapy has had limited results outside of case reports ,7. The mHepatic metastases from a primary extremity soft-tissue sarcoma are rare . There iA 61-year-old male noted a mass in his left lateral calf. A biopsy was performed revealing a myxoid liposarcoma. The patient subsequently underwent resection of an 8.0 × 6.0 × 3.5 cm tumor with wide margins Figure , and lesThe patient did well for 14 months, but follow-up computed tomography (CT) demonstrated an asymptomatic 6.3 by 6.8 cm mass in the right lobe of the liver that was growing in size  frequently occur in cases of visceral and retroperitoneal tumors. It is uncommon for extremity soft-tissue sarcomas to spread to the liver (< 0.5%) . It is eThere are few treatment options for metastatic soft tissue sarcomas (STS) to the liver. Conventional chemotherapy has not impacted survival in patients with metastatic STS to the liver. The group at MSKCC treated 52 of 65 patients with STS with hepatic metastases with a doxorubicin-based chemotherapy and the partial response rate was 6% with no complete responders [There is a single case report of a patient with an isolated hepatic metastasis from an extremity liposarcoma who underwent resection and remained alive for 22 years . This paComplete resection of liver disease is the only means for long-term survival in metastatic extremity liposarcoma but cure is rarely achieved. Unfortunately, the patient in this case report had a short disease free interval following liver resection and was identified to have disease on the chest wall and spine after 5 months. Consideration should be given for "adjuvant" therapy following resection of metastatic sarcoma. However, the most active chemotherapeutic options for sarcoma are of limited value and are associated with serious and potentially life-threatening toxicity. Median survival from the time metastases are recognized is on the order of 12 months, although 20% to 25% of patients with metastatic sarcoma are alive 2 years after diagnosis. In fact, the most recent study which was the largest undertaken of adjuvant chemotherapy in soft tissue sarcoma failed to demonstrate a survival benefit .Optimal treatment of patients with unresectable or metastatic soft tissue sarcoma requires an understanding of the natural history of the disease, close attention to the individual patient and an understanding of the benefits and limitations of the therapeutic options.The authors declare that they have no competing interests.CAG assembled data and participated in drafting of the manuscript. JDR assembled data and reviewed pathology. SRG participated in writing the manuscript and critical review. SNH conceived concept, assembled data, participated in drafting of the manuscript and critical review.Patient consent could not be obtained as the patient died, the case was presented to the Health centre Institutional Review Board of University of Florida and an IRB exemption was obtained.


We report on a case of gossypiboma. A 54-year-old woman was admitted to our hospital with abdominal mass. She had undergone a caesarean operation 23 years previously. The mass in the right abdominal quadrant was suspected by abdominal computed tomography and magnetic resonance imaging. The mass was removed by laparotomy excision and the final diagnosis was gossypiboma. Gossypiboma is derived from gossypium (“cotton” in Latin) and boma  and is a rare complication of surgery –4. The r We herein report a gossypiboma resulting from 3 retained surgical swabs that had been left in peritoneum for 23 years after a caesarean operation.A 54-years-old female patient was admitted to our policlinic with intermitent abdominal pain and complaint of rigidity around her umbilicus. It was learned from her past history that she had a Caesarean operation 23 years ago. On her a physical examination a pfannenstiel incision scar and a 15 by 10 cm diameter rigid, mobile mass located right abdominal cavity were found. On abdominal computed tomography (CT) scan the 6 cm-by-6 cm in diameter calcific mass had smooth edges and a central lipophilic density located at right quadrant was thought as a lipoma. On magnetic resonance imaging (MR) scan the 43-by-42 mm diameter mass that did not take intravenous contrast agent was evaluated as a gallbladder stone or myoma uteri. We operated on the patient. The abdominal cavity was explored. On exploration in the right abdominal quadrant the 15-by-10 by 6 cm diameter a rigid and mobile mass that conglomerated with ileum, transvers colon and caecum was found . No dilaGauze, ped, compress, and clamps are the most forgotten materials after surgical operations. The incidence of gossypiboma is difficult to estimate because of not being reported, but it has been reported that varies from 0.01% to 0.001%. Most of the gossypiboma are identified only after abdominal or pelvic surgery , 7. Reta Gossypiboma induce two types of reaction. First is exudative in nature and leads to formation of abscess. An aseptic fibrinous response, which creates adhesions and encapsulation and eventually results in the developmentof a foreign-body granuloma . Abdominal gossypibomas may be asymptomatic or may present with abdominal pain, abdominal mass, intestinal obstruction, gastrointestinal hemorrhage, intraabdominal sepsis, granulomatous peritonitis, and fistulization into surrounding structures .Because the symptoms of gossypiboma are usually nonspecific and may appear years after surgery, the diagnosis of gossypiboma usually comes from imaging studies and a high index of suspicion. In most countries, surgical sponges contain radiopaque material that facilitates detection by standard abdominal radiography. Such sponges can also be identified readily in CT images. However, surgical sponges without radiopaque markers are still used in many institutions, and this type of sponge is very difficult to identify by using standard radiographic and CT imaging. Therefore, retained surgical foreign bodies often make diagnosis intractable .The granulomatous reaction and surrounding tissues caused by foreign body may reach huge volume and, may lead to misdiagnosis as a tumor. This huge mass sometimes causes to bowel occlusion. If the gossypiboma consist of metal, possiblity of perforation will increase and, mortality due to this condition increases . After the diagnosis of gossypiboma is confirmed, removal of the retained sponge surgically, endoscopically or laparoscopically is accomplished in order to prevent severe morbidity or mortality may lead to death .In conclusion, gossypiboma can be diagnosed early postoperative stage providing that usig materials having radiopaque markers. However, materials having radiopaque markers are not used in the most of health service in our country. In order to prevent these types of complications, we have to control all of the surgical materials before and after surgery, which is the main principle in all procedures.


Duodenal atresia is a common cause of intestinal obstruction in neonates. It is associated with other congenital anomalies like Down's syndrome, annular pancreas etc.We present a case of a two days old male baby presented to us with bilious vomiting since birth. It was associated with Down's syndrome, Annular pancreas and Malrotation.Duodenal atresia is associated with other congenital anomalies but more than one congenital anomalies in a single patient is very rare. The duodenum is the most common site of intestinal obstruction accounting for nearly half of all cases . The incDuodenal atresia is associated with Down's syndrome (30%), annular pancreas (23%), congenital heart disease (22%), malrotation (20%), oesophageal atresia (8%), others (20%) .The mainstay of treatment is surgical intervention. Duodenoduodenostomy is most frequently performed operation.We report a case of duodenal atresia associated with more than one congenital anomalies.A male baby 2 days old presented to us for bilious vomiting since birth. On examination patient has features of Down's syndrome and no other obvious anomaly. A nasogastric tube is passed and 40–50 ml bilious fluid aspirated. Xrays abdomen erect film showed a double bubble sign. Provisional diagnosis of duodenal atresia made and patient was optimized for surgery At operation we found duodenal atresia, annular pancreas and malrotaion of gut.Duodenoduodenostomy was peformed. Recovery of patient was uneventful.Duodenal atresia is frequently associated with other congenital anomalies but combination of anomalies in a single patient is very rare. This report illustrates a rare setting in which three associated congenital anomalies were present in a single patient of duodenal atresia.Written informed consent was obtained from the parents for publication of this case report.The authors declare that they have no competing interests.BM presented the case history, researched the topic and helped draft the manuscript. LI reviewed the literature and drafted the manuscript. MS was the supervising consultant paediatric surgeon who supervised the surgery. AS is head of department helped and guided for preparation of case report. All authors read and approved the final manuscript.


Splenosis is the heterotopic autotransplantation of splenic tissue, mostly found after splenic trauma or surgery in the abdominal, pelvic or thoracic cavity. Here we report a patient with a history of splenectomy after polytrauma with chronic hepatitis C and liver cirrhosis presenting with an hepatic mass of unknown origin.The lesion could not be exactly classified by ultrasound, computed tomography, angiography and biopsy, classical features of malignancy were not fulfilled, and on the other hand a neoplastic process could neither be excluded. After revision of a MRI performed in our centre it appeared that the liver mass contrasted in the same way as the remaining accessory spleens in the left upper quadrant. A selective Tc-99m-labelled heat-denatured autologous red blood cells scintigraphy of the spleen was performed and showed both the accessory spleens in the left upper quadrant and spleen-typical tissue in projection to the left liver lobe and confirmed the diagnosis of splenosis.Although intrahepatic splenosis represents an extremely rare condition, this diagnosis should always be taken into consideration in patients with history of abdominal trauma with splenic involvement presenting with an indeterminate focal liver lesion. The diagnosis of splenosis may then be reliably confirmed by Tc-99m-DRBC scintigraphy. Intrahepatic masses occurring in patients with liver cirrhosis and hepatic viral infections have a high likelihood to be regenerative nodules or HCC. The differential diagnosis of benign pseudo-lesions may not be obvious in this context and comparably rare. Diagnostic imaging such as ultrasound, liver MRI and transfemoral arterial angiography play a pivotal role but may not always be conclusive at first glance. Diagnostic interpretation in the clinical context is always warranted.We report a case of intra-hepatic splenosis in a patient with hepatitis C, liver cirrhosis and a history of severe abdominal trauma with multi-organ contusion and splenic rupture followed by splenectomy. The value of various diagnostic modalities in this rare distinct pathology is discussed.A 43-year-old Caucasian man from Germany with liver cirrhosis and chronic hepatitis C virus infection was referred to our tertiary centre presenting with a tumour in the left liver lobe of indeterminate origin. Infection with HCV occurred in 1979, following multiple transfusions in the context of a severe polytrauma with liver rupture, diaphragmatic rupture, left kidney rupture and splenic rupture, the latter requiring subsequent splenectomy. In 2001, a liver biopsy was performed confirming the presence of liver cirrhosis. The hepatitis C genotype was 1a according to Simmonds. Antiviral therapy was started immediately with pegylated interferon and ribavirin but was not successful due to virus relapse at the end of the therapy.In October 2004, a second try of antiviral therapy was planned. Routine ultrasound of the liver at that time revealed a polylobulated mass with regular margins in segment II of the left liver lobe. Subsequent abdominal MRI confirmed the presence of multiple confluent nodular lesions in the left liver lobe measuring 2.5 × 7.0 cm. These lesions showed hypervascularity on contrast-enhanced liver MRI and malignancy was suspected. The patient was admitted to our hospital to confirm the diagnosis and to establish appropriate treatment of suspected HCC.At admission, the patient did not complain about any symptoms except for increased fatigue. He did not suffer from weight loss, fever or nocturnal sweats. The physical examination was normal. Clinically, the cirrhosis was graded Child A. Serum aminotransferases were slightly elevated . AFP was normal with 6.4 ng/ml.MRI was repeated in double contrast technique including dynamic imaging after intravenous application of standard Gadolinium-DTPA  and SPIO - enhanced imaging in the same examination . Besides the signs of subtle micro-nodular regenerative liver cirrhosis of the entire liver parenchyma, multiple nodular lesions between 4 and 36 mm in diameter were identified in the subcapsular region of segment II. The partially confluent nodules showed pronounced slightly inhomogeneous hypervascularity in the early arterial phase of dynamic Gd-enhanced liver MRI and a lack of contrast uptake of the liver specific superparamagnetic iron-oxide (SPIO) containing contrast agent .Enlarged paracaval and hilar lymph nodes and accessory splenic tissue in the left subdiaphragmatic area were identified at the same examination. The suspicion of a potentially malignant process concerning the hepatic nodules was sustained and further investigation was considered necessary to confirm or rule out hepatocellular carcinoma (HCC). Transfemoral intraarterial angiography of the liver was performed as a primarily diagnostic and potentially therapeutic procedure. It showed regular branches of the hepatic artery but no pathologic vessels or parenchymal foci of hypervascularity.CT-guided biopsy was then considered and attempted although the peripheral location of the nodules offered limited approach. The histologic findings showed cirrhotic liver tissue with chronic periportal inflammation and steatosis of up to 30 % of the hepatocytes. There was no confirmation of malignancy.Despite the negative results of all diagnostic and imaging modalities, the potential malignancy of the lesions could still not be ruled out at this point. Therefore, the MRI which was performed in our center, and all other investigations were revised for a second time.Facing a similar arterial contrast enhancement behaviour of the intrahepatic nodules on MRI when compared to the residual and accessory splenic tissue observed in the left subdiaphragmatic region, a selective scintigraphy with Tc-99m-labelled heat-denatured autologous red blood cells (Tc-99m-DRBC) was performed in the attempt to investigate also for the rare differential diagnosis of intra-hepatic splenosis in this context. Tc-99m-DRBC finally confirmed this differential diagnosis showing uptake in both of the accessory spleens in the left upper quadrant and in the area projecting to the left liver lobe . A folloSplenosis is the heterotopic autotransplantation of splenic tissue and occurs in 16 to 67% of the patients after traumatic spleen rupture or spleen surgery .Splenosis is a rare entity. To date, around 100 cases were reported in the literature until 2004 . HoweverHematogenous spread of splenic pulp is supposed to be a possible mechanism of seeding into the liver. The subcapsular location of the splenic tissue in our case may suggest the implantation on the inferior surface of the diaphragm which was ruptured in the same trauma mechanism rather than by hematogenous spread into the liver. However, the marked indentation of the nodules into the liver parenchyma made exact localization difficult.In the vast majority of cases with splenosis, patients are asymptomatic and an undetermined mass is found incidentally. Sometimes, non-specific symptoms such as abdominal pain or diarrhea may lead to the diagnosis. Gastrointestinal bleeding after rupture of a splenic implant nodule and intestinal obstruction are rather rare complications associated with splenosis.Chronic hepatitis C has a prevalence of about 1% in the Middle-European population. Transmission of the hepatitis used to occur frequently after transfusion in polytraumatized patients. 20% of the patients develop liver cirrhosis. The incidence of HCC in patients with liver cirrhosis and hepatitis C is about 7 % in 5 years [The spectrum of differential diagnosis for focal liver lesions in diagnostic radiologic imaging is wide but may be narrowed down in patients with hepatitis C and liver cirrhosis to hemangioma, adenoma, focal nodular hyperplasia, regenerative nodules, dysplastic nodules and hepatocellular carcinoma.The EASL (European Association of the Study of the Liver) criteria for the diagnosis of HCC recommend a coincidental presence of arterial hypervascularity of a suspect nodule in two imaging techniques or an AFP level over 400 ng/ml and concomitant arterial hypervascularity in one imaging technique. According to Bolondi, in a study of small nodules in patients with liver cirrhosis, 38 % of the HCC nodules measuring 1-2 cm and 16 % of the HCC nodules measuring 2-3 cm were not satisfying the EASL criteria concerning hypervascularity. All nodules larger than 2 cm resulted to be HCC [The respective nodules in our patient showed slightly inhomogeneous but marked contrast enhancement in the early arterial phase of dynamic imaging after Gadolinium-DTPA, evocative of HCC. The additional lack of uptake of the SPIO contrast agent further supported the suspicion of malignancy.Double contrast MRI of the liver using Gadolinium-DTPA and SPIO as liver specific contrast agent has proven to enhance the diagnostic accuracy in the detection and characterization of focal liver lesions and HCC  and is wThe liver biopsy performed in our centre showed cirrhotic tissue and did not reveal any components of splenic tissue. As a consequence, it has to be assumed that the lesion of interest was missed at biopsy. The limited access to the lesion in its subcapsular and subdiaphragmatic location in the left upper abdominal quadrant may be the most plausible explanation to this negative result. According to the literature, in CT-guided liver biopsies the diagnostic yield is 67 to 96 % ,9.It remains an interesting observation that a large number of the reported cases of hepatic splenosis are patients with hepatitis B or C with or without liver cirrhosis . It coulThe specific diagnosis of splenosis is most reliably established with help of Tc-99m-DRBC scintigraphy. Tc-99m-DRBC scintigraphy has shown to be more sensitive than Tc-99m-sulfur colloid scintigraphy . About 9As splenosis is a quite rare disease there exist no studies with a larger cohort of patients to evaluate sensitivity and specificity of Tc-99m-DRBC scintigraphy. Yet, Tc-99m-DRBC scintigraphy is still considered as the gold standard in the non-invasive diagnosis of splenosis.Although intrahepatic splenosis represents an extremely rare condition, this diagnosis should always be taken into consideration in patients with history of abdominal trauma with splenic involvement presenting with an indeterminate focal liver lesion.At contrast enhanced MRI with Gadolinium-chelates, splenosis shows a signal behaviour and enhancement pattern similar to that of potentially present accessory spleen but does not necessarily exhibit iron uptake on SPIO enhanced imaging. The diagnosis of splenosis may then be reliably confirmed by Tc-99m-DRBC scintigraphy and will help to avoid unnecessary major surgery or other invasive therapeutic procedures.Splenosis per se does not require particular follow up with diagnostic imaging or other investigations. Interferon therapy of hepatitis C in the cirrhotic patient with splenosis is safe, however, careful follow-up is crucial.


Residual appendicitis involving the stump of the appendix has been well described in the literature in the past.We report the case of a 43 year old male with acute onset of abdominal pain who had undergone an appendicectomy ten years previously. Ultrasound revealed the presence of an inflamed tubular structure. Subsequent laparotomy and histology confirmed that this structure was an inflamed residual appendiceal tip.Residual tip appendicitis has not been reported in the literature previously and should be considered in the differential diagnosis of localised peritonitis in a patient with a history of a previous open appendicectomy. Acute appendicitis is a common surgical emergency requiring intervention. The lifetime risk of developing appendicitis is approximately seven per cent . DespiteWe report the case of a 43 year old male who presented with sudden onset of severe abdominal pain. His symptoms were consistent with acute cholecystitis. He was unable to speak English and communicated via an interpreter. On examination a Battle's incision was noted. He claimed that this was due to a previous cholecystectomy completed ten years previous in his home country. He demonstrated signs of localised peritonitis in the right upper quadrant. Inflammatory markers were elevated. A clinical diagnosis of acute cholecystitis was made and broad spectrum antibiotics commenced. An ultrasound was performed and this confirmed the presence of a non-inflamed gallbladder which did not contain gallstones. However, it did reveal an inflamed tubular, non-compressible, non-peristalting, blind-ending structure intimately related to the liver suggesting sub-hepatic appendicitis Figure .Laparoscopy revealed dense adhesions throughout the abdominal cavity and the presence of purulent intra-abdominal fluid. We proceeded to a laparotomy through a midline incision. Following extensive adhesiolysis the caecum was identified, however only a non-inflamed appendiceal stump remained. Further dissection revealed evidence of pus in the right sub-hepatic space. The pus was originating from a tubular structure which was adherent to the lateral abdominal wall. An intra-operative diagnosis of residual tip appendicitis was made. The tubular structure, measuring 2.5 cms in length, was removed and was confirmed histologically as appendicitis with evidence of acute inflammatory changes. The patient made an uneventful recovery and was discharged home six days post-operatively.Appendicectomy is usually performed either via open or laparoscopic surgery. At open surgery the conventional incisions are of either the Gridiron or Lanz variety. A Battle's incision, which employs a vertical para-median incision with temporary retraction of the rectus muscle medially, was initially described in 1895 . In the There are thirty seven cases of residual appendicitis in the English literature. The majority of these case reports involve stump appendicitis. Stump appendicitis remains, however, an under reported condition. It occurs when there is incomplete resection of the inflamed appendix. The reported interval between operations varies between two months and 50 years ,5. In ouResidual appendicitis may be diagnosed using radiological imaging. Ultrasound imaging is useful and was diagnostic in this case. A pre-operative ultrasound, which was done primarily to confirm the presence of a gallbladder, visualised a sub-hepatic appendicitis. Computed tomography (CT) is also useful in diagnosing residual appendicitis. CT features of stump appendicitis are similar to those of acute appendicitis. They include pericaecal inflammatory changes, abscess formation, fluid in the right paracolic gutter, caecal wall thickening, and an ileocecal mass. Barium studies have also been reported as being diagnostic in diagnosing stump appendicitis . There aThe surgical treatment of residual appendicitis is most commonly reported as an open operation but cases have been successfully treated using laparoscopic intervention. This case required a laparotomy as there were dense adhesions from previous surgery and the exact source of purulent fluid was not evident laparoscopically. Residual appendicitis has been reported most frequently in cases of appendicitis treated with laparoscopic surgery. There is a high morbidity rate in this setting due to a combination of delayed diagnosis and a high rate of stump perforation . The ratResidual tip appendicitis has not been reported in the literature previously and should be considered in the differential diagnosis of localised peritonitis in a patient with a history of a previous open appendicectomy.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the journal's Editor-in-Chief.The authors declare that they have no competing interests.EM - Assistant surgeon, JC - Radiologist who conducted ultrasound, IW - Consultant who operated on patient. All authors read and approved the final manuscript.


Paragangliomas that originate from the urinary bladder are extremely rare. In most series, bladder paragangliomas often cause micturitional attacks. Treatment modalities include transurethral resection and cystectomy . Prognosis of bladder paraganglioma is similar to that of adrenal pheochromocytoma.A 55-year-old Chinese woman presenting with the sole complaint of lower abdominal pain for one month was admitted to our hospital. Ultrasound and computed tomography revealed a mass on the dome of the bladder measuring 4.0 × 3.0 cm. The tumor was completed removed by laparoscopic partial cystectomy. Histological examination of the tumor indicated paraganglioma of the urinary bladder. The clinical features, diagnosis, management and pathological findings of paraganglioma of the urinary bladder are discussed.Bladder paraganglioma should be considered as a differential diagnosis to neoplasm in the urinary bladder, although there is no characteristic symptom. Laparoscopic partial cystectomy may be the first choice in treating paraganglioma of the urinary bladder, offering several advantages such as less invasion, rapid recovery and early discharge from the hospital. Paragangliomas, which arise from the chromaffin tissue of the sympathetic nervous system in locations outside the adrenal gland, are referred to as extra-adrenal pheochromocytomas. The first case of paraganglioma of the urinary bladder was reported by Zimmerman in 1953 . In mostA 55-year-old Chinese woman complaining of lower abdominal pain for one month was admitted to our hospital. Her family history was unremarkable and she had no previous medical problems. Her blood pressure was 110-130/70-80 mmHg, and her heart rate was in the normal range. Physical examination showed no evidence of hypertensive disease. Both ultrasound examination and computed tomography (CT) scan demonstrated a mass on the dome of the bladder, measuring 4.0 × 3.0 cm  during the second to fourth decades of life [131-methyliodobenzylguanidine (I131-MIBG) and positron emission tomography (PET)-CT help to evaluate its function. The significance of cystoscopy is limited and biopsy is not recommended since the tumor is usually located in the submucosa with an intact surface, and biopsy may provoke a hypertensive episode in patients who have not had proper medical treatment. Additionally, artifactual changes in a small biopsy may confuse diagnosis [With the advances in imaging technology, ultrasound, computed tomography (CT) and magnetic resonance imaging (MRI) may be useful in localizing the tumor, while Iiagnosis . In our In some asymptomatic bladder paragangliomas, it is very difficult to have an actual pre-operative diagnosis. Histological and immunohistochemical diagnosis become the last choices. Paragangliomas of the urinary bladder show histological features similar to adrenal pheochromocytomas. The tumor cells usually grow in a nested, zellballen pattern with delicate fibrovascular stroma, although they may also grow in a diffuse pattern . Immunohet al. [Treatment modalities include transurethral resection and partial or total cystectomy combined with pelvic lymph node dissection, especially in the presence of proven metastasis . Howeveret al.  first reBladder paraganglioma may be misdiagnosed when characteristic symptoms are absent. Laparoscopic partial cystectomy for bladder paraganglioma may be the first treatment of choice, offering several advantages such as less invasion, rapid recovery and early discharge from the hospital.The authors declare that they have no competing interests.DFX performed the surgery. MC collected the patient data and wrote the manuscript. YSL was a major contributor in writing the manuscript. YG performed the surgery as first assistant. XGC performed the surgery as second assistant. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


A 23-year-old male patient with right nasal Juvenile Nasopharyngeal Angiofibroma (JNA) developed Central Retinal Artery Occlusion (CRAO) during embolization of the tumor using polyvinyl alcohol particles before endoscopic excision. Classic CRAO management was initiated by an ophthalmologist after 12 h. Retrospective evaluation of the angiograms revealed a tiny communication between the external carotid and ophthalmic arteries which had not been noticed before embolization. During endoscopic excision, the tumor was found to originate extraordinarily from midline structures. It was concluded that CRAO might be a rare complication of JNA embolization. Careful preoperative angiographic evaluations to detect communicating arteries and immediate ophthalmologic consultation in case of developing visual symptoms during the procedure are necessary. Central Retinal Artery Occlusion (CRAO) is one of the most sudden and dramatic events seen by ophthalmologists. One of the major causes of CRAO is embolism. There are many procedures associated with embolic complications and interventional radiological procedures are among the rare ones.Juvenile Nasopharyngeal Angiofibroma (JNA), one of the common benign nasal cavity tumors of adolescence, exhibits a strong tendency to bleed during surgical removal. Nowadays, preoperative embolization is commonly used to minimize such intraoperative bleeding; however, this technique is not without compli-cations.3 Here, wA 23-year-old male patient presented with the com-plaint of right nasal obstruction. After observing a purple pink mass completely filling the right nasal cavity, the diagnosis of JNA was made. Computerized tomography revealed a nasal cavity mass extending partially to the sphenoid sinus, pterygomaxillary fossa, and infratemporal fossa.In order to reduce bleeding during the later endoscopic resection, the patient underwent a transarterial particulate embolization procedure as a part of preoperative preparation. After catheterization of both external carotid arteries with 5 F MP catheters (Cordis) via right transfemoral approach, selective angiography was performed. Abnormal tumor blush [After 12 h, the patient was examined by an ophthalmologist in another hospital. Left eye visual acuity (VA) was counting finger at 0.5 meter and the relative afferent pupillary defect was positive in this eye. Left fundus examination revealed retinal edema and a ″cherry-red spot″ appearance of the macula with narrowed vessels, which were compatible with the diagnosis of CRAO. Ocular massage, anterior chamber paracentesis as well as systemic therapy with carbonic anhydrase inhibitor and mannitol were initiated. During the follow-up, VA stabilized at 20/200.Retrospective and precise reevaluation of pre-embolization angiograms revealed a suspicious tiny communication between the external carotid artery and ophthalmic vessels on the left side , arrow. Four months after the embolization process, the patient underwent a successful endoscopic angiofibroma en bloc exci-sion. Interestingly, the tumor was found to originate from midline structures and attached to the posterior free border of the nasal septum.Fourteen months after embolization, VA was 20/200. Cherry-red spot disappeared leaving attenuated retinal arterioles and optic disc atrophy.Many CRAOs are embolic in origin. Radiologic procedures including hysterosalpingography, arteriography, cardiac catheterization, and interventional procedures are among the many different conditions that can cause embolus formation. Preopera4et al. presented a patient with JNA who developed CRAO following preoperative embolization. They could not demonstrate any responsible communicating artery and therefore assumed the existence of a branch of the internal maxillary artery supplying the intraorbital contents and the retina in their case. Hence, they recommended more detailed assessment of these possible dangerous collaterals by superselective catheterization of the internal maxillary artery branches with microcatheters.[It has been known that inadvertent embolization of the brain or eye during JNA management occurred via dangerous collaterals from the internal maxillary artery to the intracranial/intraorbital contents. Önerci atheters. In our cThe other pitfall encountered in our case was the delay in the management of CRAO. It was mostly due to the lack of an ophthalmologist in the primary center in which radiologic intervention was performed. To make this presentation concise we will not elaborate on the management of CRAO. However, since the precious time for keeping the retinal cells alive by restoring the retinal artery flow is estimated to be about 90 min, this rep7During surgical removal of the tumor, it was noted that the tumor had an extraordinary origin from the midline as opposed to the usual lateral location of the tumor pedicle in the majority of JNA cases. The association of this rare tumor position with the existence of a collateral vessel in our case might have happened by chance and we could not reach any conclusion in this regard.This case report introduces a patient with CRAO as a rare but an important complication following preoperative embolization of an unusually located JNA tumor. This case also highlights the necessity of careful evaluation of angiograms for detection of any abnormal collateral vessel(s) prior to embolization and the importance of immediate diagnosis and treatment in patients who develop ocular symptoms during or shortly after the interventional procedures.


Iatrogenic injuries of the membranous trachea have become increasingly common and may trigger a cascade of immediate life-threatening complications.A case of a 48-year-old man with an iatrogenic membranous tracheal wall rupture after double-lumen intubation during Ivor Lewis esophagogastrectomy is presented. Tracheal injury was successfully managed surgically with the use of bovine pericardial patch and reinforcement with the gastric conduit which was moved toward the posterior wall of the membranous trachea sealing the wall laceration.Our technique was proved to be safe, effective and not technically demanding. Early recognition with prompt surgery is the gold standard of managing such cases, although small tears can be managed conservatively. Iatrogenic injuries of the membranous trachea are rare but potentially lethal and can complicate procedures such as endotracheal intubation, mediastinoscopy, percutaneous tracheostomy and excision of neoplasms with mediastinal involvement. Statistically women are more predisposed to this than men . FurtherA 48-year-old man was admitted to our department with the diagnosis of an adenocarcinoma of the gastroesophageal junction. Preoperative staging was negative for distant metastases. Endoscopic ultrasound findings confirmed the presence of a locally advanced tumor (type I – Siewert classification). It was an adenocarcinoma of the distal esophagus arising from an area with intestinal metaplasia and infiltrating the esophagogastric junction from above. As a consequence preoperative synchronous chemoradiotherapy was proposed. The scheme was consisted of fluorouracil (15 mg/kg of body weight) daily for five days and cisplatin (75 mg/m2 of body surface area) on day 7 and a course of radiotherapy  over a a three week period, beginning concurrently with the first course of chemotherapy. After the completion of chemoradiotherapy, the stage of the disease was reevaluated . Restaging was completed with no evidence of distant disease and surgical removal of the primary tumor and lymphadenectomy was proposed. The patient was eligible for an Ivor Lewis esophagogastrectomy.He was intubated with a left-sided 37-F double-lumen endotracheal tube by a specialist anesthesiologist experienced in cardiothoracic surgery anesthesia. The technique includes the following points. The tube was held with the bronchial curve concave anteriorly. As the tip was passed through the larynx, the tube was rotated 90 degrees to direct the endobronchial part to the intended side. The tube was then connected to the breathing circuit via a double catheter mount.The abdominal part of the operation was uneventful. During the second phase and while the intrathoracic esophagus was being mobilized a hissing noise was heard and a careful inspection revealed part of the double-lumen tube and the inflated tracheal cuff protruding through the ruptured membranous tracheal wall into the operating field. This resulted in an approximately 3 × 1 cm oval-shaped laceration of membranous trachea, extending to 1 cm above the carina  declare that they have no competing interests.NB, GS, DP, CL and TB took part in the care of the patient and and contributed equally in carrying out the medical literature. CT participated in the care of the patient and had the supervision of this report. All authors approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Diffusion-weighted magnetic resonance imaging is a very sensitive tool for the early diagnosis of acute ischemic stroke. This is employed in some stroke centers as the primary screening tool to select patients eligible for thrombolysis.We present the case of a 49-years old Chinese man whose diffusion-weighted magnetic resonance imaging performed 12 hours of symptom onset was negative.Although the initial diffusion-weighted magnetic resonance imaging was negative, the imaging repeated after 4 days despite static neurological symptoms and signs, could demonstrate an acute medullary infarction.Diffusion-weighted imaging may not be100% sensitive in very early stages, especially in posterior circulation strokes. Our case serves as a reminder that clinical assessment still retains priority until a diagnostic modality offering 100% sensitivity and specificity is discovered. Diffusion-weighted magnetic resonance imaging (DWI-MRI) is considered to be a very sensitive tool for the early diagnosis of acute ischemic stroke. This is employed in some stroke centers as the primary screening tool to select patients eligible for thrombolysis. However DWI may not be100% sensitive in very early stages, especially in posterior circulation strokes. We present a patient in which DWI-MRI was negative even after 12 hours of symptom onset and only the imaging repeated after 4 days could demonstrate an acute infarction.A 49-year old Chinese man, office clerk by occupation was admitted with sudden vertigo and unsteady gait. The vertigo was unremitting with eye closure and not posturally related. There was no trauma, neck pain or illness. There was no significant medical illness in past and accordingly, he was not on regular medication and did not smoke. Clinical examination revealed right lateral gaze-evoked nystagmus, right upper limb dysmetria and ataxic gait. Sensation to pain and temperature was intact and there was no evidence of a Horner's syndrome.An emergent computerized tomography (CT) of the brain was unremarkable. An MRI of the brain including diffusion-weighted imaging (DWI) 24 hours after admission did not reveal an acute lesion. However, an absent right vertebral artery flow void on axial T2-weighted images was observed Figure . CervicaOur patient demonstrates that DWI-MRI performed early may fail to demonstrate an acute infarct especially if it is small and located in territory supplied by posterior circulation. This may have an impact on the clinical decision making process in centres using DWI-MRI as a tool to guide aggressive therapy in patients with acute stroke . Our casIn acute cell death, failure of sodium-potassium ATPase pumps result in the shift of water from extracellular to intracellular compartments, reflected as a hyperintense lesion on DWI . DWI proabsence of a DWI-lesion does not completely rule out an ischemic state.Technical reasons have been postulated for the occasional failure of DWI-MRI to demonstrate an acute infarction including poor spatial resolution, high signal-to-noise ratio and magnetic susceptibility artefacts around the brain stem . AnotherThis may be argued that the first MRI could have missed the particular section of the brain that underwent the ischemic infarct. This possibility, although very unlikely, can not be completely excluded. Another explanation for DWI-negative MRI could be an interval infarction between the two MRI scans performed in our case. However, our patient did not develop any new symptoms or signs suggestive of a new neurological injury.In conclusion, if there is a high index of suspicion for an acute cerebrovascular event especially involving the posterior circulation with an unremarkable initial MRI-DWI, a repeat study after 24 hours of a fixed neurological deficit may be more demonstrative. Our case serves as a reminder that clinical assessment still retains priority until a diagnostic modality offering 100% sensitivity and specificity is discovered.DWI: Diffusion-weighted imaging; MRI: Magnetic resonance imaging; CT: Computerized tomography.The authors declare that they have no competing interests.RR Patient management, initial draft of the manuscript, VKS Patient management, draft and review of manscript, conceptualization and final approval, BPLC Patient management, conceptualization, final approval.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Adrenal incidentaloma can be described as adrenal lesions that are incidentally diagnosed during abdominal laparotomy or any abdominal screening without prior suspicion of adrenal disease. It is important to diagnose adrenal lesions to learn if they are hormonally active or malignant. The most common clinical sign of pheochromocytoma is sustained or paroxysmal hypertension, and the most common symptoms are headache, excessive truncal sweating, and palpitation. In some cases, the clinical symptoms are not clear. Roughly 70% of adrenal incidentalomas are non-functional. A small group of 5–7% of the functional ones (30%) may exist as pheochromocytoma. Ten percent of pheochromocytoma cases are diagnosed incidentally during computed tomography (CT) or magnetic resonance imaging (MRI) screenings for other reasons. The most frequent symptom of the pheochromocytoma is hypertension, and 90–100% of cases exhibit it. The literature indicates that incidental pheochromocytoma cases that are smaller than 1 cm have no clinical symptoms. Rarely, some large pheochromocytoma cases do not show any clinical symptoms, and it is difficult to diagnose very small ones.A 45-year-old male patient experienced an epigastric ache and distended stomach for 7 years. The serum cortisol level was 19.2 ng/dL , and urinary free cortisol excretion was 25.00 μg/24 h . The serum basal level of adrenocorticotropic hormone (ACTH) was 21 pg/mL . Plasma cortisol was under 1.00 μg/dL after low dose (1 mg) overnight dexamethasone suppression test. 24 hours urinary catecholamines level were vanil mandilic acid (VMA) 8.90 mg/day , metanefrin 330 μg/day , epinefrin 13 μg/day , norepinefrin 41 μg/day . During abdominal ultrasonography (USG), a tumor was diagnosed in the right perirenal space. A regular-shaped mass (dimension 36 × 35 × 35 mm) with a homogeneous and solid structure was diagnosed in CT. The density of the mass was 80 Hounsfield units (HU) in postcontrast CT. The patient was given a diagnosis of a non-functional adrenal incidental lesion, underwent a right adrenalectomy. Histopathological data correlated with pheochromocytoma as wellPheochromocytoma can be diagnosed by establishing an increase in catecholamines and metabolites in the plasma and urine. The level of catecholamines and metabolites in the plasma and urine provide 95% of the evidence of the disease. Because the dimensions of the lesion were large and the HU was very clear, the patient was underwent surrenalectomy. During laboratory investigation, there was no evidence of abnormality; we, therefore, think that these cases can be named sublaboratory pheochromocytoma. Adrenal incidentaloma can be described as adrenal lesions that are incidentally diagnosed during abdominal laparotomy or any abdominal screening without prior suspicion of adrenal disease. This definition does not include adrenal lesions that are diagnosed for oncological metastasis. In the last 30 years, case diagnosis of adrenal incidentaloma increased about 20 times because of technological development. It is important to diagnose adrenal lesions if the lesions are hormonally active or malignant. The number of adrenal incidental cases increase with age and application technique. Statistical autopsy data suggests that these lesions occur in 1% of people at age 30, but 7% at age 70 with no difference in gender [Pheochromocytoma is a tumoral disease that originates from chromaffin cells in the sympathoadrenal system. These tumors secrete epinephrine, norepinephrine, and in some cases, dopamine and catecholamines. They secrete other active peptides and cause symptoms of hypertension. Because pheochromocytoma is a rare disease, it is present in only 0.1–0.2% of hypertension patients. Although there is no difference in age or gender, occurrence increases in the 4th and 5th decades. Most pheochromocytoma cases localize to the abdomen (90%), and 90% of abdomen-localized cases are in the adrenal glandulas. The most common clinical sign of pheochromocytoma is a sustained or paroxysmal hypertension, and the most common symptoms are headache, excessive truncal sweating, and palpitation. Angina pectoris, abdominal pain, queasiness, anxiety, and paleness are other symptoms of it as well. In some cases, clinical symptoms are not clear until they are diagnosed incidentally during computed tomography (CT) or magnetic resonance imaging (MRI) .Roughly 70% of adrenal incidentalomas are non-functional. A small group of 5–7% of the functional ones (30%) may exist as pheochromocytomas . We revi2. His right hand tension arteriel was 110/70 mmHg and his left hand tension arterial was 115/70 mmHg; when standing, these values were 110/70 and 110/70 mmHg, respectively. Heart rate was 80 beats/minute, and no pathologic data was found in the system examination. The laboratory data showed that hemogram and urine findings were normal. His lipid profile was normal. Other findings were as in Table A 45-year-old male patient experienced an epigastric ache and distended stomach for 7 years. During abdominal ultrasonography (USG), a tumor was diagnosed in the right suprarenal fossa. He did not use any drugs and there was no characteristic of past medical and family illness history. He was 1.74 cm and weighted 81 kg, with a body mass index (BMI) of 26.5 kg/mPlasma cortisol was under 1.00 μg/dL after low dose (1 mg) overnight dexamethasone suppression test. 24 hours urinary catecholamines level were vanil mandilic acid (VMA) 8.90 mg/day , metanefrin 330 μg/day , epinefrin 13 μg/day , norepinefrin 41 μg/day . Hormonal levels were as in Table There was a 34 × 26 mm solid lesion in the right perirenal space.Liver parenchyma was homogeneous. In the right lobe of the liver, there was a two millimeter subcapsular calcification. A regular-shaped mass (dimension 36 × 35 × 35 mm) with a homogeneous and solid structure was diagnosed at the right suprarenal fossa. The density of the mass was 80 Hounsfield units (HU) in postcontrast CT and 40 HU without contrast CT Fig. .After all these examinations, the patient was given a diagnosis of a non-functional adrenal incidental lesion. Because it was 80 HU in contrast CT, 40 HU without contrast CT, and 36 × 35 × 35 mm, the patient underwent a right adrenalectomy Fig. . There wThe number of incidentally found adrenal lesions is growing. The most important problem in appraising adrenal incidental cases is diagnosing the lesion as benign or malignant. To distinguish malign and benign lesions, screening methods must be used. CT is an ideal screening method for the adrenal glandula and the most helpful technique for distinguishing cysts, bleedings, and myelolipomas. On the other hand, MRI is successful in distinguishing adrenal adenomas, carcinomas, and metastatic masses. But CT has gained widespread acceptance as the technique of choice for distinguishing between adrenal adenomas and metastasis .Adenomas frequently contain sufficient intracytoplasmic fat to produce lower attenuation values. With an increasing proportion of lipid mass, the density will be lower. Although the lipid proportion is high in benign cases, it is lower in malignant ones. No adrenal malignancy with a density of 0 HU has been reported in the literature. It is suggested that 4–20 HU density cases must be suspicious and patients in these cases undergo MRI. The cases in which the density is higher than 20 HU are rated as malign in some other studies . In someThe dimensions of lesion and a history of cancer are important factors for distinguishing whether masses are benign or not. While the carcinoma prevalence is 2 % for the masses smaller than 4 cm, it is 6 % for the ones 4–6 cm and 25 % for those bigger than 6 cm. It is impossible to morphologically distinguish adrenal metastases from benign masses. The most reliable technique for distinguishing these two masses involves conducting a percutan biopsy after elimination of the diagnosis of pheochromocytoma. If the mass is pheochromocytoma, percutan biopsy will cause a hypertension crisis. Biopsy is not an adequate technique for distinguishing adrenocortical adenomas and adrenocortical carcinomas . While oThe second important question in adrenal incidentaloma is whether a lesion is functional or not. No functional scanning is needed for myelolipoma and adrenal cysts, which were diagnosed with screening methods alone. According to the increase in the sensitivity of the endocronologic tests used for differentiation diagnosis, the prevalence of the subclinical syndrome and/or a silent preclinical one is increased in these cases.About 5–10 % of the adrenal incidental cases are subclinical instances of Cushing syndrome. In all cases, Cushing screening should be conducted with a 1 mgr Dexametazone test. In this test, the limit for the pressured plasma cortizol level is 1.8 μg/dL [The level of the plasma potassium and renin-aldosterone must be checked in hypertensioned adrenal incidental cases. If the level of renin-aldosterone activity is greater than 30 and the level of plasma aldosterone is higher than 20 ng/dl, there is an autonomic aldosterone secretion. Roughly 80% of the primary aldosteronism are adenomas and 1% of the cases are adrenocortical carcinomas. In cases of adrenocortical adenoma with primer aldosteronism, the dimensions of the adenomas are typically small . BecauseTumors producing androgens are rare. They may be malignant or benign, and clinically, they have virilization effects. DHEA-S provides the most important evidence for the secretion of adrenal androgen. The level of DHEA-S should be measured in adrenal incidentaloma cases. There was no increase in androgen function due to the lack of hyperandrogenic findings, and the levels of DHEA-S were normal in our case.Pheochromocytoma is a rare catecholamine secreting tumor derived from chromaffin cells. It is thought that 5–7 % of functional adrenal incidental masses may be pheochromocytomas. Although pheochromocytoma is not one of the main causes of hypertension, it will be fatal if not controlled. About 10 % of pheochromocytoma cases are diagnosed incidentally during CT or MRI that are screened for other reasons . On the The literature states that incidental pheochromocytoma cases that are smaller than 1 cm have no clinic symptoms . Rarely,Pheochromocytoma can be diagnosed by establishing the increase of catecholamines and metabolites in the plasma and urine. The level of catecholamines and metabolites in the plasma and urine provides 95 % of the evidence for the disease. Free catecholamines (epinephrine or norepinephrine) or metabolites  can be measured in the urine, and the levels of metanephrine or catecholamines can be measured from the plasma. Measurement of metanephrine in the urine is the most useful test . If the The dimension of the mass was > 3 cm but the HU was up to 40 HU. Our patient underwent surrenalectomy even though there was no incidental pheochromocytoma. Laboratory findings support the absence of a pheochromocytoma. Since the operation material of the patient showed some findings related to pheochromocytoma, the patient was scanned for multiple endocronological neoplasias (MEN) in the postoperative period. Calcitonin and PTH levels detected for screening MEN. No clinical or diagnostical findings have been observed.All incidental adrenal cases must be examined very carefully, because the chance of mortality is high when they are not diagnosed. Incidental pheochromocytoma cases have nonhomogenic, cystic, and hemoragic images in screening methods. Because our case was homogeneous, we did not consider pheochromocytoma as a diagnosis for our case. The image of a bilateral surrenal incidental lesion is one of the most important symptoms of pheochromocytoma. The image of the left adrenal was normal in our case.During the laboratory investigation, there was no evidence of abnormalities. We, therefore, conclude that these cases can be regarded as sublaboratory pheochromocytomas. However, further studies of more cases must be done to clarify this subject.CT: Computed tomography; MRI: Magnetic resonance imaging; BMI: Body mass index; USG: Ultrasonography; VMA: Vanil mandilic acid (VMA); PTH: Parathyroid hormone; ACTH: Adrenocorticotropic hormone; HU: Hounsfield Unit; DHEA-S: Dehidroepiandrosteron sulfate; ECG: Electrocardiography.The authors declare that they have no competing interests.MO was involved the clinical preparation and prediagnosis of the patient, SB was involved the preparation of pathological findings. All other authors conducted the operation and prepared the manuscript.We have obtained written, informed patient consent for publication of the report and any accompanying images.


Soft-tissue perineuriomas are rare benign peripheral nerve sheath tumors in the subcutis of the extremities and the trunks of young patients. To our knowledge, this the first presentation of the computed tomography and magnetic resonance imaging of a soft-tissue perineurioma in the retroperitoneum with pathologic correlation.A 63-year-old Japanese man was referred for assessment of high blood pressure. Abdominal computed tomography and magnetic resonance imaging showed a well-defined, gradually enhancing tumor without focal degeneration or hemorrhage adjacent to the pancreatic body. Tumor excision with distal pancreatectomy and splenectomy was performed, as a malignant tumor of pancreatic origin could not be ruled out. No recurrence has been noted in the 16 months since the operation. Pathologic examination of the tumor revealed a soft-tissue perineurioma of the retroperitoneum.Although the definitive diagnosis of soft-tissue perineurioma requires biopsy and immunohistochemical reactivity evaluation, the computed tomography and magnetic resonance imaging findings described in this report suggest inclusion of this rare tumor in the differential diagnosis when such findings occur in the retroperitoneum. Soft-tissue perineuriomas are rare benign peripheral nerve sheath tumors with perineurial cell differentiation -4. They To the best of our knowledge, this the first presentation of the CT and MRI images of a soft-tissue perineurioma in the retroperitoneum with pathologic correlation.2-weighted, and hypointense on T1-weighted MR images (Figure A 63-year-old Japanese man was admitted to our hospital for assessment of hypertension. Abdominal CT and MR imaging revealed a 4 cm well-defined mass at the anterior portion of the pancreatic body. The mass was hypodense with ca.12 HU on plain CT Figure , and onlTumor excision with distal pancretectomy and splenectomy was performed, as a malignant tumor of pancreatic origin could not be ruled out.Macroscopically, the tumor was a well-circumscribed, whitish myxoid mass with no focal degenerative changes Figure . It was Microscopically, the tumor consisted of spindle cells with oval nuclei and thin bipolar elongated cytoplasmic processes loosely arranged in abundant myxoid stroma Figure .The tumor was immunohistochemically characterized as a soft-tissue perineuroma, given its positive immunoactivity for CD 34, EMA, glut1, claudin-1, and negative immunoactivity for S-100. Ultrastructurally, the tumor cells possessed markedly attenuated bipolar cytoplasmic processes running in parallel, which are the characteristics of perineurial cells.Although the tumor was firmly attached to the pancreas, no direct invasion was noted. Sixteen months after his operation, follow-up CT showed no evidence of a recurrent tumor.Perineuriomas are uncommon benign peripheral nerve sheath tumors that include four subtypes: soft tissue, intramural, sclerosing, and reticular. Soft-tissue perineuriomas are the most common subtype and show distinctive morphologic, ultrastructual, and immunophenotypic features that distinguish them from the much more common nerve sheath tumors, schwannomas, and neurofibromas. The tumors show a morphologic spectrum ranging from hypercellular lesions with collagenous stroma to hypocellular tumors with myxoid stroma. The immunohistochemical studies are often necessary for the diagnosis of soft-tissue perineurioma ,4.1-weighted images, and hyperintensity with low-intensity capsule on T2-weighted images. Only contrast enhancement on fat-suppressed T1-weighted images was more prominent in our report. A dynamic MR study revealed the gradually enhancing nature of this myxoid tumor.To the best of our knowledge, MRI and CT images of soft-tissue perineurioma in the retroperitoneum have not been reported. The soft-tissue perineurioma in this report was similar to the one in the subcutis of the groin : well-deNearly half of soft-tissue perineuriomas are hypocellular, and 20% are markedly hypercellular. The stroma can vary from a more collagenous to a myxoid appearance. Whereas the stroma in most tumors is collagenous, 40% show at least focally myxoid stroma, and 20% are nearly exclusively myxoid [Radiologic differential diagnosis of retroperitoneal soft-tissue perineurioma includes neurilemmomas, neurofibromas , ganglio2-weighted images. The tumor signal intensity on T2-weighted images depends on the proportion of myxoid stroma to cellular components and collagen fibers. MR dynamic enhancement patterns of the two entities are similar: they lack early enhancement, but delayed enhancement increased gradually. However, one of the MR imaging characteristics of ganglioneuroma is curvilinear bands of low signal intensity on T2-weighted images [In contrast, the current case of soft-tissue perineurioma showed no definite focal degeneration or hemorrhagic areas. Cellular/intramuscular myxomas arise in the muscle, and are rarely found in the retroperitoneum . The mosd images , which wd images , and thed images .Other differential diagnoses include malignant tumors such as perineurial malignant nerve sheath tumor , malignaDynamic MR imaging better provides the gradually enhancing nature of a myxoid tumor. Soft-tissue perineuriomas with marked myxoid stroma may be mistaken for cysts on plain CT.Although the definitive diagnosis of soft-tissue perineurioma requires biopsy and immunohistochemical reactivity evaluation, the CT and MRI findings described in this report suggest inclusion of this rare tumor in the differential diagnosis when such findings occur in the retroperitoneum.The authors declare that they have no competing interests. Funding was neither sought nor obtained.MY conceived the study. YK and RM performed the literature review. AA and KK performed histopathologic and immunohistochemical analyses. All authors read and approved the final version of the manuscript.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Congenital polyps of the posterior urethra are rare lesions, and have often been described in boys. The polyps are benign lesion usually lined by a transitional epithelium, but cases of squamous or intestinal metaplasia have been reported.We report a case of a 10 years old boy referred for hematuria and dysuria.The voiding cysto urethrogram revealed a filling defect in the posterior urethra. At cystourethroscopy, a polyp of the posterior urethra was found and resected transurethrally. Histological examination showed a polyp with a fibro muscular core covered by focal gastric metaplasia with fundic gland. To our knowledge this is the first reported case of gastric metaplasia of urethral polyp. Urethral polyps are a rare cause of bladder outlet obstruction in the pediatric age group. They are benign fibroepithelial polyps usually diagnosed in the first decade. They have been described often in boys and the posterior urethra is the predominant location .Most patients present with varying degrees of irritative and obstructive voiding symptoms and hematuria .A 10 year old boy was referred to our hospital with complaints of dysuria and hematuria. He had a 6-month history of progressive loss of force and interruption of his urinary stream. He had also a recent history of urinary tract infection treated by antibiotics. Clinical examination and ultrasonography revealed no abnormalities.A voiding cysto urethrogram showed no reflux or urethral valves but revealed a filling defect in the posterior urethra. Cystourethroscopy revealed the presence of 0,5 cm polyp arising from the posterior urethra. The polyp was resected in its entirety at the base using a 9F resectoscope.Histopathological examination showed the lesion to be a benign fibroepithelial polyp covered by transitional epithelium with focal areas of gastric metaplasia with fundic glands figure  and 2. UPolyps derived from the lower urinary tract are not as frequent when compared with those derived from the upper urinary tract . Polyps The presence of large polyps in healthy newborns and infants is a strong argument in favor of a congenital origin. It has been proposed that they may arise from mesonephric remnants .Concerning metaplasia of the urothelium, three basic types are described: squamous, intestinal, and nephrogenic metaplasia .Intestinal metaplasia of the superficial epithelium of the prostatic urethra was reported, and can be associated with dysplasia .Gastric metaplasia is extremely rare and has been previously recognized in the urothelium of the ureter  and the To our knowledge, the present case is the first reported case of urethral gastric heterotopia.The etiology of the metaplasia of the urothelium is still controversial. Congenital, irritative, infectious, obstructive and traumatic causes have been proposed . MetaplaIn our observation, gastric metaplasia of the posterior urethral polyp can be linked to obstruction and chronic inflammation.Transurethral resection has provided an adequate therapeutic approach in this case. Considerable caution must be taken because of the proximity of the external sphincter to the verumontanum.Other surgical options include Bugbee fulguration of the polyp, laser excision and open suprapubic transvesical excision .Regardless of the treatment, there have been no reported recurrences when the polyp has been removed completely .Written informed consent was obtained from the parents of the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.MJ and AH collected the data and literature review, and wrote the manuscript. AS, WH and NBS revised and provided comments on the manuscript. FM was the attending doctor, carried out the surgical procedure and literature review. All authors read and approved the final manuscript.


A 29-year-old female presented with complaints of cough, shortness of breath and sputum production for the last few years. There was a history of one episode of hemoptysis. She had no history of fever, weight loss or loss of appetite.Clinical examination was unremarkable.Chest radiographs Figures  and 2 weWhat are the radiological findings on these images?What is the most likely diagnosis and the differential diagnosis of this appearance?On the CT scan, AP and lateral chest X-rays, about 9 cm in diameter oval-shaped solid mass with well-defined borders is seen, and it contains three flicks of calcification. The mass is situated in the left middle lung zone, extending medially into the left lung hilum. Posteriorly, the mass is found abutting the posterior chest wall without causing rib destruction; however, the adjacent rib has slightly increased density. The mass has homogenous density with minimal contrast enhancement. The rest of the lungs were clear, and no mediastinal lymphadenopathy was seen.Benign: Lung sequestration, bronchogenic cyst (CXR), hydatid cyst (CXR), chondroma, aspergiloma, neurofibroma, etc.Malignant: Ewing's sarcoma, PNET, chondrosarcoma, neurofibrosarcoma, malignant fibrous histocytoma, lymphoma, etc.Diagnosis: Thoracic Ewing's SarcomaEwing's sarcoma is the most common primary skeletal tumor of the thoracic cage. Fifteen The radiology finding in the chest is a chest wall mass with rib destruction (82%), but mixed lytic-sclerotic (9%) and even sclerotic only (9%). However, Ewing's sarcoma occasionally manifests as a large intrathoracic mass with only a small component of bony involvement.Extra-osseous intrathoracic Ewing's sarcoma is very rare, and it is usually seen in young adults and manifests as a well-circumscribed, noncalcified mass, more frequently in the paravertebral location. This tumor has propensity to spread inwards towards the lung hilum and into the spinal canal via the intervertebral foraramina.Involvement of bone marrow, the hallmark of an osseous origin, is typically absent at CT and MRI imaging in these extra-osseous tumors.One-quarter to one-third of the patients with Ewing's sarcoma have metastases at the time of diagnosis, especially those with tumor close to the trunk. The most common sites of spread are lungs and bones; hence chest CT scan, MRI and bone scan are important in the patient work-up.Treatment is initially chemotherapy and usually followed by resection with or without radiotherapy. The overall 5-year survival rate is 50%.Ewing's sarcoma (extra-osseous or rib origin) should be considered in the differential diagnosis of any young patient who presents with a solid intrathoracic mass.