Transomental herniation is a rare but recognised clinical condition, which usually presents as an emergency with bowel obstruction. It accounts for 1-4% of intra-abdominal herniations. We reviewed 3 patients found to have a transomental defect during elective diagnostic laparoscopy performed for chronic abdominal pain. To our knowledge, there is no case series reported in the literature on transomental defect in the non-emergency situation.A retrospective case note analysis of 3 patients, found to have transomental defect during elective diagnostic laparoscopy, was undertaken. Data were gathered with respect to clinical presentation, investigations performed, transomental defect size and outcome of surgery. All patients were followed up for 6 months post-operatively. Three females (age range 18-35 years) were referred with a 3-10 year history of chronic intermittent abdominal pain, often postprandial. Blood tests, radiological investigations  and endoscopy were all normal. In each case, diagnostic laparoscopy revealed the presence of a peripheral defect in the greater omentum, but no actual small bowel herniation. No other pathology was found. These defects were resected, which subsequently led to complete resolution of the patients’ symptoms.Chronic abdominal pain of unknown aetiology with normal radiological findings may be caused by intermittent obstruction due to small bowel herniation through a transomental defect. This should be considered during elective diagnostic laparoscopy, in the absence of any other obvious pathology. The omentum should be thoroughly inspected as a discrete entity and any such defects should be closed or resected. Transomental hernias are rare accounting for 1-4% of all internal hernias . The majThe aim of this study was to document the number of patients where transomental defects (TOD) were found in patients undergoing elective diagnostic laparoscopy for chronic abdominal pain and whether the correction of the defect led to resolution of the symptoms.A retrospective analysis of 3 patients with TOD found at diagnostic laparoscopy was undertaken. Our initial case led to the finding of a large congenital TOD, which led to the hypothesis that it may have been responsible for intermittent transomental herniation of likely small bowel and thereby explaining the patient’s symptoms. The correction of the defect led to complete resolution of the symptoms immediately after surgery and this was confirmed at her 6 months follow-up. This led us to scrutinise the greater omentum more closely during similar cases, leading to two further cases with similar findings.An 18-year-old British Caucasian female presented to the gastroenterology team with a 5-year history of intermittent abdominal pain. She has always been a very active individual both in sports and education, but was affected by sudden onset central cramping abdominal pain, often postprandial followed by episodes of diarrhoea. The pain would resolve spontaneously. Neither the frequency nor the duration was predictable.Her haematological & biochemical results were normal. Radiological investigations in the form of ultrasound, magnetic resonance cholangiopancreatography (MRCP) and small bowel follow through were also within normal limits. The patient’s only past medical history included an open appendicectomy at the age of 5 years for a non-perforated inflamed appendicitis.The general surgical team performed a diagnostic laparoscopy in hope of finding a diagnosis. This revealed a large central 5 cm × 6 cm greater omental defect on the inferior border . The omeA 35-year-old British Caucasian female was managed under the care of gastroenterology, paediatrics & gynaecology with a 10-year history of chronic right iliac fossa pain. Her endoscopic examinations (OGD & colonoscopy) and radiological investigations  were normal. Her only significant past medical history included a diagnostic laparoscopy by the gynaecologist when she was 25 for mid-cycle pain with associated dyspareunia. There were no positive findings at that laparoscopy and her gynaecological symptoms settled, but she continued to have persistent right iliac fossa pain, which was colicky in nature with associated nausea and vomiting and settle spontaneously with pain free episodes.Repeat diagnostic laparoscopy revealed a macroscopically normal looking appendix and an omental adhesion forming a 3 × 4 cm defect , 3. The A 28-year-old British Caucasian female presented with a 3-year history of chronic abdominal pain. The pain was described as being postprandial but with no correlation with food types. It was mainly central in site but often generalised. OGD & colonoscopy with biopsies, CT abdomen and pelvis, small bowel follow through and ultrasound abdomen and pelvis were normal. Diagnostic laparoscopy revealed the presences of a 2 cm × 3 cm greater omental defect on the left margin , 4. The TOD are often recognised when patients present with acute intestinal obstruction mainly involving the small bowel but also segments of the large bowel such as sigmoid and caecum. These can sometimes be confirmed using pre-operative radiology but often at the time of emergency surgery.In our small but yet interesting case series, the patients were younger than the usual age of greater than 50 years [The aetiology of these defects varies from congenital, trauma or inflammatory causes . HoweverThe radiological investigations for such defects are difficult, as seen in our patients who had multiple negative investigations prior to laparoscopy. All our patients were found to have a defect in the greater omentum, whist the other type of omental herniations through the lesser omentum, in comparison, is more often detected pre-operatively with CT or barium studies -5. RadioWe wish to highlight the importance of looking for such defects in the setting of chronic abdominal pain, which has not been previously reported. The omentum is a viscus in its own right, and hence, at diagnostic laparoscopy, it should be inspected as a discrete entity, especially in the absence of any other pathology. It is also advisable that the greater omentum should be dealt carefully during other laparoscopic procedures to minimise trauma and creation of TOD, which can lead to bowel herniation and subsequent obstruction. Our review of the literature shows mainly cases from the radiological aspect during acute situations with patients presenting with bowel obstruction. To our knowledge, there is no literature discussing the role of the transomental defect in an elective setting prior to these patients possibly being admitted in the future as an emergency with transomental herniations.


Background. 10% of ovarian fibromatous tumours typically exhibit increased cellularity, mitotic activity, and less frequently nuclear atypia. Therefore, the classification within the group of fibromatous tumours may represent some difficulties, thus, one or several of these features should appear. Case. We introduce the clinical and pathologic features based on one case of recurrence of a mitotically active cellular ovarian fibroma (MACF) in the pararectal fossa. This recurrence took place six years after primary surgery. Macroscopically, the tumour was firm, fibrous, well delimited, yellow-white without gross necrosis. On microscopic examination, it was composed of a densely cellular proliferation of fibrolastic-like cells with bland nuclear features and arranged in a fascicular pattern. There was no sign of significant atypia or necrosis. Conclusion. Recently, this case is the first report of a recurrence of MACF, following primary surgery with no tumoral rupture or surgical difficulty. The clinical outcome of ovarian cellular fibromas (CFs) and MACFs is typically uneventful. This case, however, strongly suggests maintaining a long-term clinical follow-up even though the principal tumour was surgically treated without tumour rupture or in the absence of adherence or any surgical difficulty. A 65-year-old woman lookedfor assistance to the gynecological consultation due to recent pelvic pain. Fiveyears earlier, she went through a total hysterectomy with bilateralsalpingoophorectomy for a benign ovarian mass described as an ovarian fibromawhich eventually happened to be 10 cm long.The gynecological exam did not show any mass,the vaginal cuff was soft and not tender. A vaginal ultrasound and a CT scan highlighteda pelvic mass of 10 cm in the pouch of Douglas and revealed no evidence of adenopathy. A laparoscopic exploration of abdominalcavity was performed. This pointed out a large mass in the Douglas while no other lesion inside the cavity was seen. Subsequently, the mass wasresected and properly extracted in an endobag. Due to its very low position inthe pelvis and to the hemorrhage, the resection could not be completed.Macroscopically, the tumour (12 × 10 × 9 cm) was firm, fibrous, well delimited,yellow-white without gross necrosis. On microscopic examination, it was made ofa densely cellular proliferation of fibrolastic-like cells with bland nuclearfeatures and arranged in a fascicular pattern. No significant atypia ornecrosis could be observed. The tumoral cells were highly immunoreactive forvimentin and focally for alpha-inhibin,actin and CD99. Desmin, h-caldesmon,CD10, HMB-45, andc-kit were negative. The mitotic index was estimated at 4 mitoses/10 HPFs and the ki-67 indexat 9%. After reviewing the slides concerning the primitive ovarian tumour,these presented similar morphological and immunohistochemical features.Based on the above, it washence decided to diagnose a recurrence of mitotically active cellular fibromaof the ovary (MACF). Six months later, the patient was diagnosed with a newrecurrence which mass was 8 cm long and situated in the right pararectalregion. The patient went though an MRI which later confirmed the recurrence ofthe mass. A laparotomy was then performed with a large resection of thepararectal recurrence . The patWe present the clinical andpathologic features over one case of recurrence of a mitotically activecellular ovarian fibroma. This specific type of histological characteristic israre. Ovarian stromal tumourscomposed of a pure proliferation of fibroblastic cells usually consist offibromas, cellular fibromas (CFs), and hardly ever of fibrosarcomas. Themajority of these neoplasms are benign fibromas with appreciable intercellularcollagen, nonstriking cellularity, bland nuclei, and rare to absent mitoticfigures (MFs).About 10% of fibromatoustumours exhibit increased cellularity, mitotic activity, and less frequentlynuclear atypia. It is then difficult to classify a case within the group offibromatous tumours when one or several of these features exist.In 1981, Prat and Scullysuggested histological criteria for the distinction of CFs from fibrosarcomasbased on a study including the analysis of 17 different cases [More recently, a study fromIrving et al. reviewed 75 cases of ovarian CFs . They alNo data about follow-upwere highlighted for patients presenting CFs though these happened to bementioned with regard to patients presenting MACFs. Finally, this study did notrefer to any recurrence for patients with MACFs.Since the first studypublished by Prat and Scully in 1981, a number of ovarian fibrosarcomas with abenign clinical course have been reported in the literature in which thediagnosis was based primarily or exclusively on a mitotic rate of 4 or moreMFs/10 HPFs –11. In tAccording to thisclassification, review of literature shows that only 2 cases of recurrence ofMACFs have been reported . Both paOur case shows a recurrence5 years following primary surgery. The first surgical intervention wasperformed without any problem hence no adhesiolysis was necessary and theovarian tumour was extracted without rupture. Our report is the first topresent a late recurrence of MACF after primary surgery which was previouslyperformed with no tumoral rupture or surgical difficulty.The clinical outcome ofovarian CFs and MACFs is typically uneventful, but this report stronglysuggests long-term clinical follow-up even if primary tumour was surgicallytreated without rupture or in absence of adherence or surgical difficulty.


Synchronous gynecological tumors are rare; it is even rarer to find the rarest of gynecological tumors that of the fallopian tube, together with a histological sub-type as rare as verrucous cervix.We report a synchronic fallopian tube adenocarcinoma and a verrucous cervical cancer. A 85-year-old woman with postmenopausal genital hemorrhage, endometrial biopsy was reported as squamous metaplasia, an exploratory laparotomy was performed finding a tubal tumor diagnosed as adenocarcinoma, a staging procedure was performed. Final staging revealed IB1 cervical carcinoma and IA G3 fallopian tube carcinoma. Adjuvant treatment with chemotherapy was not accepted by the patient. The patient has remained in follow-up, and at 9 months, there has been no documented evidence of recurrent disease.Reasons for our presentation of this work are: first, due to the rarity of these, and second, because of the usefulness of possessing a case report for establishing a norm for later behavior with respect to treatment of these patients. Over the past years, an increase has been detected in the incidence of synchronous tumors, due mainly to the increment in life expectancy of the population and the development of ever more specific diagnostic methods that discover tumors that were not perceived previously , as wellThe most common presentation is the combination of endometrium and ovarian neoplasms in the case of ovary, this is related with incessant ovulation, consequent estrogen production, and continuous stimulation of the endometrium, with the consequent formation of neoplasms characteristic of this site .Malignant neoplasms of cervix, vagina, and vulva, in which the presence of the human papilloma virus (HPV) plays a very important role, comprise another example of synchronous tumors in which if the lesion is voluminous, it may not be possible to differentiate one from the other .Association of the common risk factors in infrequent gynecological tumors such as those of fallopian tube, which represents < 2% of cases  is nearlCancer of the cervix is frequent neoplasm. The most frequently identified histologies are squamous, adenocarcarcinoma, and adenosquamous, these representing > 95% of cases, there are several rare histological variants of squamous carcinoma, such as verrucous, which is found at a much higher frequency in other sites such as oral cavity, skin, and larynx .In this histological variant, the role of HPV has also been implicated as etiological agent. However, reports that exist in this respect have not been able to determine any HPV type in particular .If synchronous gynecological tumors are rare, it is even rarer to find the rarest of gynecological tumors that of the fallopian tube, together with a histological sub-type as rare as verrucous cervix. We present a case of a patient with a primary carcinoma of the fallopian tube synchronous with a cervical carcinoma, verrucous type.A 84 year female was referred to the Instituto Nacional de Cancerlogía de México for abnormal postmenopausal genital hemorrhage. Family cancer history was negative. The patient reported no personal antecedents of importance related with the condition. On physical gynecological examination, there was no evidence of macroscopic lesions of vulva, vagina, or cervix; uterus was in 8-cm anteversion, and adnexa were not palpable. Colposcopy was performed, and this was reported as unsatisfactory due to an important atrophic cervical epithelium without evidence of acetowhite lesions and with normal vascular pattern.A pelvic Ultrasound (US) was conducted reporting a 10-mm endometrial thickening. Fractionated endometrial biopsy showed condiloma. An exploratory laparotomy and total abdominal hysterectomy with bilateral salpingo-ophorectomy with frozen section study was planned. During the procedure, there was evidence of right adnexal tumor of 6 × 4 cm. A frozen section evaluation reported serous just carcinoma of right fallopian tube limited to fallopian tube; in cervix, a lesion in endocervical canal was identified, which was diagnosed as benign in the intraoperative pathological consultation; in uterus, there was no macroscopically identified endometrial lesion. Therefore, fallopian tube-staging surgery was completed.Final staging was IB1 cervical carcinoma and IA G3 fallopian tube carcinoma according to FIGO staging system. The patient declined any adjuvant treatments, either chemotherapy or radiotherapy or both. The patient remained free of disease after nine months of close follow up.Grossly the cervix showed a lesion in the endocervical canal very near the inferior segment that measured 2 × 1.1 cm, which partially obliterated the canal and infiltrated the cervical stroma on the left side. Right fallopian tube was dilated, and the lumen was occupied by a papillary-like neoplasm localized in the middle third of the fallopian tube, which extended to the external third  using the BigDye Terminator v3.1Positive PCR products were purified and subsequently sequenced in a programmable thermal cycler  by using one of the PCR oligonucleotides as a sequencing primer(GP5+). The obtained sequence was compared with the GenBank database  by using the BLAST program resulting in that the sample was positive for type 11 HPV, no high risk HPV DNA was possible to identify .At a specialized hospital such as the Instituto Nacional de Cancerología de México, the finding of synchronous tumors, despite their rarity (6%), is expected. Among rare gynecological tumors, tumors of the fallopian tube represents 2% -3,7,9,10The great majority of fallopian tube tumors are diagnosed after surgery by anatomopathological evaluation based on criteria established by Hu and modified by Sedlis as follows : 1) If bOverall survival for cancer of the fallopian tube is 50–60% at 5 years; the majority of recurrences presents during the first 2–3 years, and are nearly all extrapelvic ,11. InitVerrucous carcinoma of the cervix represents less than 1% of cases , the relTreatment of choice is surgical, including treatment for recurrences in which total pelvic exenteration has to be considered; this is because invasion is due to a greater degree to local extension than to the lymph gland pathway. In general, this tumor type entertains a good prognosis when surgery is feasible. Radiotherapy should be avoided given that the tumor is radio-resistant and in addition, radiotherapy induced anaplastic changes that lead to regional and distant metastases as a consequence ,7,9.Recently there are some case reports of cervical and tubal carcinomas, one of them  was assoSeveral reports show relation between abnormalities on cervical smears and tubal carcinoma, these abnormalities are characteristically glandular rather than squamous and is important to mention that no association with human papilloma virus induced abnormalities had been shown .We can conclude that synchronous gynecological tumors are rare, most frequent association of fallopian tube tumors is with endometrium, although there are reports that support their relationship with breast cancer. Verrucous carcinoma is very rare in cervix, entertains a good prognosis, and is a type of cervical cancer in which radiotherapy results in damage rather than in benefit. Reasons for our presentation of this work are: first, due to the rarity of these, and second, because of the usefulness of possessing a case report for establishing a norm for later behavior with respect to treatment of these patients.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 authors declare that they have no competing interests.DCL was involved in the design and writing of the manuscript. DPM was involved in pathologic evaluation and writing of the manuscript. AT was involved in literature and case review. RMM was involved HPV detection typing and genetic sequence. LC was involved in manuscript completion and critical review. All authors read and approved the final manuscript.


Well-differentiated papillary mesothelioma of the peritoneum (WDPMP) is a rare subtype of epitheloid mesothelioma, which is usually seen in young women. WDPMP is generally considered of low malignant potential, however the long-term nature of the tumor remains poorly defined.We describe the long-term follow-up of a 60-year-old woman of West African descent who has survived 24 years with WDPMP after receiving extensive local and systemic adjuvant chemotherapy. Her clinical course has included three exploratory laparotomies with intraperitoneal and intravenous chemotherapy over two decades. Her course was complicated by anthracycline-induced cardiomyopathy, for which she underwent an orthotopic heart transplant. Our patient is alive with stable radiological evidence of peritoneal disease, and continues to suffer from chronic abdominal pain.No consensus exists regarding optimal treatment strategies for WDPMP. However, given the low malignant potential of the tumor, careful consideration should be made before proceeding with aggressive interventions. Further, long-term follow-up reports are required to fully characterize this tumor. Mesothelioma is an uncommon neoplasm which originates from the mesothelial lining of the pleura, pericardium, peritoneum, and tunica vaginalis ,2. MaligWe report the case of a 60-year-old woman of West African descent, with no history of asbestos exposure, who originally presented 24 years ago to another institution with acute abdominal pain. At that time, she underwent an exploratory laparotomy and was found to have nodules diffusely covering the peritoneum. A total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed for suspected ovarian carcinoma, and biopsies were taken of the peritoneal nodules. The pathology from this original surgery was interpreted as low-grade papillary mesothelioma. She then received six adjuvant cycles of intravenous cyclophosphamide, doxorubicin and cisplatin. She underwent a second-look laparotomy six months later, and still had gross disease visible in the peritoneum. Post-operatively she received three additional cycles of intraperitoneal cisplatin and intravenous sodium thiosulfate. She subsequently received maintenance therapy with alternating courses of tamoxifen and megace alternating every two weeks.She presented four years later with obstructive gastrointestinal symptoms and was again found on laparotomy to have diffuse peritoneal studding. Pathology from this surgery was interpreted again to be papillary mesothelioma. As a result, she began six cycles of carboplatin and cyclophosphamide chemotherapy for suspected progressive disease. Several months later, she presented with complaints of shortness of breath, orthopnea, and worsening lower extremity edema. A multi-gated acquisition scan (MUGA) revealed an ejection fraction of 14% and enlarged cardiac silhouette on chest X-ray, and she was clinically diagnosed as having anthracycline-induced cardiomyopathy. Medical therapy was initiated at that time for congestive heart failure.Two years later, she was found on a computed topography (CT) scan to have an interval increase in loculated subhepatic fluid collection and a lobular soft tissue mass in the right subphrenic region. She then received three cycles of VP-16 and ifosfamide. She remained well until 2000, when she underwent an orthotopic heart transplant. Upon subsequent reimaging of her abdomen the next year, she was found to have continued slow progression of the tumor and was started on single-agent paclitaxel followed by cyclophosphamide for two months. She was then referred to our institution in late 2001 with stable disease on abdominal CT and a presumed diagnosis of malignant peritoneal mesothelioma refractory to therapy. Over the following year, she was maintained on combination capecitabine and gemcitabine therapy and had stable disease as assessed by CT scans. However, in early 2003 she was found to have declining renal function and was forced to stop chemotherapy.She was observed closely until 2004 and had little change in her overall tumor burden, but had recurrent ascites requiring drainage by paracentesis on multiple occasions. Because of doubts about the true nature of her peritoneal tumor, a further biopsy of her tumor was performed in 2004, with the final interpretation demonstrating a low-grade papillary mesothelioma of the peritoneum : Malignant peritoneal mesothelioma; (WDPMP): Well-differentiated papillary mesothelioma of the peritoneum; (MUGA): Multi-gated acquisition scan; (CT): Computed topography; (IPHP): Intraperitoneal hyperthermic chemoperfusion;The authors declare that they have no competing interests.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 journal's Editor-in-Chief.JC researched and composed the literature review and the patient history. PH was a major contributor to the patient history and to critical revision of the manuscript. Both authors read and approved the final manuscript.


While biliary cystadenoma and biliary cystadenocarcinoma involving the liver are not uncommon, biliary cystadenocarcinoma of the gall bladder is an extremely rare lesion and can be very difficult to diagnose.A 50-year-old Indian woman presented with pain and swelling in the right hypochondrium. An ultrasonography revealed a cystic lesion arising from the gallbladder fossa. This lesion was initially managed with aspiration and antibiotics by the treating physician. The patient was referred for surgical management because the abscess was not resolved through conservative treatment. A diagnosis of an infected nonparasitic cyst was made and deroofing of the cyst was performed. A histopathological examination of the excised cyst wall showed cystadenocarcinoma. The patient subsequently underwent a successful surgical excision of the lesion.Infective lesions of the liver are common in developing countries and are usually managed through aspiration and antibiotics. Cystadenocarcinoma of the gallbladder needs to be considered in the differential diagnosis of cystic lesions arising from the gallbladder fossa. A high index of suspicion and cytological examination from the wall of such complex lesions will help in the timely management of such lesions. Extra hepatic biliary cystadenocarcinomas are uncommon and most often arise from the hepatic ducts. Cystadenocarcinomas arising from the gallbladder are extremely rare -3. TheseA 50-year-old Indian woman with diabetes and hypertension presented with right upper abdominal discomfort, loss of appetite and low grade fever. She had no history of jaundice. Examination revealed hepatomegaly and tenderness in the right hypochondrium. An abdominal sonography revealed an 11 × 7.5 × 11.2 cm, predominantly anechoic, thick-walled lesion arising from the inferior surface of the liver with a few low-level internal echoes and a homogenous echogenic area in the superolateral portion of the lesion. The gallbladder could not be visualized separately and the other abdominal viscera were normal. Her liver function tests were normal and serological tests for amoebiasis and hydatid disease were negative. Her chest X-ray was normal. A diagnosis of liver abscess was made and aspiration of the lesion revealed turbid fluid. However, the lesion did not resolve with repeated aspirations and antibiotics. A contrast enhanced computerized tomography (CECT) scan revealed a well-defined, lobulated cystic lesion with a solid component in the superior part of the lesion. Figure Cystic lesions originating from the gallbladder fossa can be either from the inferior surface of the liver or from the gallbladder and the distinction can sometimes be difficult. In this case, the lesion arose from the gallbladder fossa and the gallbladder was not visualised separately on imaging. It was presumed that the gallbladder was either compressed or displaced by the cystic lesion.Cystic lesions of hepatic origin, like simple cysts, parasitic cysts and abscesses, are more frequent than those arising from the gallbladder.A primary hydatid cyst, lymphangioma and an epithelial cyst associated with adenocarcinoma of the gallbladder are some of the cystic lesions of the gallbladder reported in the literature -7. Thereet al. reported such a problem in managing hepatic biliary cystadenomas and carcinomas, one of which was initially treated as a liver abscess [et al. observed enhanced mural nodules on computed tomographies in all cases of biliary cystadenocarcinoma, but not in patients with cystadenomas [Because biliary cystadenomas and cystadenocarcinomas are rare, they are not easy to diagnose preoperatively and cannot be accurately distinguished from each other merely by imaging. Simple cysts can usually be differentiated from cystic neoplasms by imaging. Thin, regular, non-enhancing wall and septations are features of simple cysts. However, complications like infection and hemorrhaging make the distinction more difficult. Cystic neoplasms can be misinterpreted as infective lesions of the liver, especially in areas where such lesions are more common, as in the present case. Yu  abscess . While s abscess ,10. Pojcadenomas . Thick aGuided fine needle aspiration cytology (FNAC) from the irregular area of the wall or the mural nodule prior to the first surgery in our patient may have avoided delays. However, definite diagnoses of FNAC of cystic lesions can be difficult owing to poor cellularity, degenerative changes in the cells and reactive changes. Sampling errors are also a problem in these predominantly cystic lesions. Fluid cytology was not found to be useful in detecting malignancy in biliary cystic neoplasms .Elevated levels of carcinoembryonic antigen and CA 19-9 in the cyst fluid were reported . These tTwo distinct types of biliary cystadenocarcinomas of the liver are described. The less aggressive tumors with 'ovarian-like stroma' occur in women and the more aggressive type without such stroma are seen in men . BecauseThe gallbladder histology in our case showed a thickened cyst wall lined by a single layer of tall columnar non-mucin secreting cells, focal nuclear pleomorphism and multilayering. Invagination into the underlying subepithelium was also seen. The underlying stroma showed dense fibrosis and congested blood vessels. Following radical excision, a subsequent biopsy of the gallbladder showed a biliary cystadenoma component with a solid area of well-differentiated adenocarcinoma suggesting the malignant transformation of a pre-existing benign lesion.The tumor in this case could be dissected off the colon and duodenum and we performed a cholecystectomy with a partial resection of the liver. A multivisceral resection was required in the case described by Waldmann .This case is reported to emphasize that cystadenocarcinoma of the gall bladder needs to be considered in the differential diagnoses of cystic lesions arising from gall bladder fossa. Image guided FNAC from suspicious areas of the wall of the cystic lesions or mural nodules may help in the diagnosis and immediate treatment, as they can be mistaken for infective lesions, in areas where such lesions are common.CECT: contrast enhanced computerized tomography; ERCP: endoscopic retrograde cholangiopancreaticography; FNAC: fine needle aspiration cytology.The authors declare that they have no competing interests.SCS and GS collected and analyzed the patient's data. SCS and GS prepared the manuscript. DB performed the histological examination. VB interpreted the CT scan images. All the authors read and 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


Gastritis Cystica Profunda is a well recognized entity which may occur several years after previous gastric surgery. This is a premalignant condition and may lead on to carcinoma of the stomach.We report a case of a 50-year-old man with epigastric pain and haematemesis. 28 years ago he had undergone partial gastrectomy and gastroenterostomy for benign gastric ulcer. An Upper gastrointestinal endoscopy showed a possible bleeding vessel on the anterior wall lesser curve of the stomach. The lesion was injected with adrenaline 1 in 100,000. In spite of the intervention he continued to have haemetemesis with significant haemodynamic impairment. At exploratory laparotomy, an oedematous ridge on the posterior wall with a bleeding point on the posterior gastric wall. Histology showed features consistent with gastritis cystica profunda. He made an excellent post-operative recovery.We suggest that patients who are diagnosed with gastritis cystica profunda should be regularly followed up as this is a premalignant condition. Gastritis Cystica Profunda is a well recognized entity occurring several years after previous gastric surgery . It is sA 50-year-old man presented as an emergency with epigastric pain and haematemesis. He was continuously vomiting out copious amount of fresh blood. He had a history of heavy alcohol intake and used to consume 10 units of alcohol per day and smoked 20 cigarettes/day. The patient had undergone partial gastrectomy and gastroenterostomy for benign gastric ulcer 28 years ago.General examination revealed anaemia with no jaundice, clubbing or lymphadenopathy. The pulse rate was 108 beats per minute and blood pressure was 80/50 mm Hg. His laboratory tests revealed Hb 8.9 g/dL and MCV 80.2. Chest and abdominal x-rays were normal. He was transfused four units of packed red blood cells. An urgent upper gastrointestinal endoscopy revealed large amount of fresh blood and clots in the fundus of stomach. A possible bleeding vessel was identified on the anterior wall lesser curve high in the fundus. The lesion was injected with adrenaline 1 in 100,000. In spite of the intervention he continued to have haemetemesis with significant haemodynamic impairment. The patient underwent an exploratory laparotomy which revealed an oedematous ridge on the posterior wall with a bleeding point on the posterior gastric wall.This was overrun with a Vicryl 2/0 stitch which controlled the bleeding. A biopsy of the oedematous ridge was obtained.Histology of the oedematous ridge revealed partly duodenal mucosa and partly a mixture of body and antral-type gastric mucosa, the latter showing areas of intestinal metaplasia. There was widespread disruption of the muscularis mucosa. The sub mucosa contained numerous glandular structures, lined mainly by antral-type gastric mucosa, showing marked cystic dilatation. These findings were consistent with gastritis cystica profunda Fig .The patient was discharged from hospital in ten days without any post-operative complications. Arrangements were made for him to be followed up as an outpatient. He was also advised to stop smoking and consuming alcohol and was referred to support groups.2 blockers [Gastritis cystica profunda (GCP) is a condition characterized by benign, cystic down growth of gastric glands into the sub mucosa of the stomach . This isblockers . This inLittler and Gleibermann proposed that mucosal prolapse and subsequent inflammation play a role in development of GCP . HistoloStage1: with cystic glands limited to mucosal layer .Stage 2: with gastric glands spreading into the sub mucosa (gastritis cystica profunda).Gastritis cystica superficialis shows wide cystic glands superficial to muscularis mucosae lined by both columnar and flattened mucous producing epithelium with basophilic cytoplasm . The chaet al. reported that the interval from gastric surgery to the formation of GCP ranged from 3 to 40 years (mean 16.2) and the risk increase with time [In their series of 18 patients with GCP, Franzin We suggest that patients who are diagnosed with gastritis cystica profunda should be regularly followed up as this is a premalignant condition.The authors declare that they have no competing interests.VI helped in acquisition of data and preparation of the first draft, IHM was responsible for conception of the idea, overall preparation and revision of the manuscript, PJM was responsible for management of the patient and revising the manuscript for important intellectual content. All authors read and 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.


Papillary ependymoma is a rare tumor that may be located along the ventricular walls or within the spinal cord. We report the case of a 54-year-old patient with a papillary ependymoma WHO grade II arising at the entrance of the aqueduct. The tumor caused hydrocephalus. The tumor was completely removed via a right-sided endoscopic approach with restoration of the aqueduct. The free cerebrospinal fluid passage through the aqueduct was not only visualized by endoscopy but also controlled by intraoperative high-field magnetic resonance imaging. Therefore, an additional endoscopic third ventriculostomy was unneccessary. Tumor related obstructive hydrocephalus is a frequent event. In most cases however, the cerebro spinal fluid (CSF) pathway is compressed by an intraparenchymal lesion itself or by the surrounding edema . In the minority of cases, tumors arise within the ventricular walls  . In thisA 54-year-old man presented to our clinic with progressive headache, visual disturbance, and, gait impairment since 9 months. Neurological examination revealed marked ataxia, no further neurological deficits. Magnetic resonance imaging (MRI) of the brain revealed obstructive hydrocephalus  caused b The patient underwent tumor resection via a rigid neuroendoscope. Neuronavigation was used as an aid for exact burrhole placement. We sought to access the aqueduct as well as, possibly, the floor of the third ventricle for third ventriculostomy. The burrhole was placed right frontal approximately 5 cm in front of the coronal suture. Intraoperatively, the tumor was found to be smooth and moderately vascularized . The lesHistopathological examination showed a papillary tumor with increased cellularity. The chromatin of the round nuclei was fine granular and the cytoplasm was eosinopilic. There were small perivascular anuclear zones with fibrillary processes. Mitoses were absent . ImmunohPostoperatively, the patient developed transient headache which was treated by CSF diversion via the ventricular drainage and an additional lumbar drainage. Hypothetically, we sought to relief transient hydrocephalus due to poor resorption in long-standing hydrocephalus. Herein, the symptoms resolved over few days and the drains could be removed. He showed also diplopia which was intermittend at first but then remained stable over the follow-up period of 15 months. Postoperative MRI revealed no spinal drop metastases.  Papillary ependymoma is a rare variant of ependymoma. The differential diagnosis includes choroid plexus papilloma, papillary meningioma and metastatic papillary carcinoma . Ependym Reviewing the literature, we found 4 articles fulfilling our request  which were published between 1963 and 1996. In 1996, Park et al. reported two cases of large papillary ependymoma in the left lateral ventricle and the pineal region, both cases were associated with hydrocephalus . An abst In our case, a small papillary ependymoma was located at the caudal entry of the aqueduct and caused hydrocephalus. The patient was treated by endoscopic tumor removal with restoration of the aqueduct. Intraoperative MRI was used to document macroscopically total tumor resection and restoration of the aqueduct. In such cases with small tumors arising along the ventricular walls, macroscopically total tumor removal should be achieved. In case of failed or questionable restoration of the aqueduct, endoscopic third ventriculostomy (ETV) should be performed. Herein, intraoperative MRI can be helpful in the decision whether ETV should be performed or not. Notable, patients should be followed postoperatively for possible secondary occlusion of the aqueduct due to adhesions. In this case, ETV might be necessary afterwards. Insertion of a ventricular-peritoneal shunt is no longer an alternative as first treatment in these patients , 3. We c In conclusion, papillary ependymoma is a rare tumor that arises along the CSF-pathway and may cause hydrocephalus. MRI is the key diagnostic tool to define the cause of hydrocephalus. Surgery, either open microsurgical resection or endoscopic resection can erase the tumor. In small tumors, endoscopy is the treatment of choice. If the aqueduct can not be restored or restoration is questionable, endoscopic third ventriculostomy should be performed. Neuronavigation can be helpful in these case to estimate the exact angle of endoscopy entrance. Histological examination of the surgical specimens is the key to diagnosis. Even if the tumor correnponds to WHO grade II, metastasis to other organs has been reported.


The majority of patients with small bowel diverticula are asymptomatic, however, associated complications include inflammation, intestinal obstruction, perforation and gastrointestinal haemorrhage. Bladder divertulae are uncommon and can herniate into the femoral or inguinal canal as well as the scrotum. We report the case of an elderly lady who underwent laparotomy for an incarcerated femoral hernia and was found to have the catheter balloon stuck into a bladder diverticulum in the femoral canal and coexisting small bowel diverticular perforation. A slender 87-year-old Caucasian lady presented with a five day history of colicky generalised abdominal pain and not opening her bowels. She was nauseated and vomited on a few occasions. She had no history of bleeding per rectum or change in bowel habit but she reported a one and a half stone weight loss over twelve months. She also described symptoms of urinary frequency. Her past medical history consisted of angina, valvular disease and an abdominal hysterectomy and salpingo-oophorectomy 20 years ago for post-menopausal bleeding with no evidence of malignancy. She was a non-smoker and consumed approximately seven units of alcohol per week. On clinical examination, there was tenderness in the right iliac fossa with generalised abdominal distension, bowel sounds were present and there was no guarding or rebound tenderness. The white cell count was normal at 7.9 × 109/L and the C-reactive protein titre was 37 mg/L. A plain abdominal radiograph displayed proximal small bowel loops and air in the large bowel (Figure Small bowel obstruction has many causes including adhesions, hernias, malignancy and inflammatory bowel disease. A common cause of small bowel obstruction and bowel strangulation is femoral herniation of the small bowel . HerniasDue to the rarity of small bowel diverticular complications, this is not the first diagnosis entertained for a patient presenting with obstruction, bleeding or an acute abdomen. This can lead to some delay in diagnosis and definitive treatment. The most useful imaging technique for diagnosing small bowel diverticulosis is CT scanning, however the disease often only presents at laparotomy . Small bHerniation of the urinary bladder wall is uncommon. The incidence of groin hernia containing the urinary bladder has been shown to be around 0.36%, although higher incidences have been reported . Often sA high index of suspicion of the content of groin hernia is required to avoid iatrogenic injury to abdominal organs. Although unusual contents of groin hernias, such as the bladder, can be picked up by imaging, such as excretory urography, the likelihood is that the contents are only delineated at surgery . From reWritten informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of consent is available for review by the Editor-in chief of this journal.The authors declare that they have no competing interests.AH, MM and SP all performed the initial clinical assessment of the patient and surgical procedure. KR collected patient data and researched the literature for discussion. KR and AH formulated a draft report. All authors contributed in the final report.


A 3-year-old girl presented with left exotropia. Funduscopy demonstrated a retinocytoma associated with five discrete retinoblastomas in the left eye and three discrete retinoblastomas in her right eye. The clinical manifestations and fundus imaging findings are described. Retinocytoma is a rare retinal tumor that is generally believed to be a benign variant of retinoblastoma.–6 The re12It is important to recognize retinocytoma clinically and differentiate it from active retinoblastoma as it usually requires close observation rather than active treatment.Simultaneous occurrence of retinocytoma and retinoblastoma is very rare78 and mo7A 3-year-old girl was referred to the oncology service because of left exotropia associated with an intraocular mass. Familial history was not contributory. Examination under anesthesia revealed an elevated semitranslucent mass measuring about 9 × 8 mm in basal dimensions and 3 mm in thickness superior to the left fovea, surrounded by a margin of chorioretinal atrophy and retinal pigment epithelial alteration . Some caThe patient was referred for systemic chemotherapy with a vincristine-etoposide-carboplatin protocol for six cycles. Examinations under anesthesia were performed in between the chemotherapy cycles every 6-8 weeks. In addition, cryotherapy was performed for more peripheral retinoblastoma tumors and posteriorly located malignant tumors were treated using transpupillary thermotherapy. Overall, two sessions of cryotherapy were performed for both eyes as well as three sessions of TTT for the left eye and two sessions for the right eye. At the last examination under anesthesia , 1 year RB1 gene mutation and carries similar genetic implications as germinal retinoblastoma.[11Retinocytoma is considered to be a rare benign phenotypic lastoma.15 In famiastoma.117 Histoloastoma.118Retinocytoma and retinoblastoma have been reported in different forms and association; in the same family,27 in a c212129Although all four diagnostic features of retinocytoma may be present in 10% of the patients, all were


A 72-year-old man presented with a two month history of rectal bleeding. Colonoscopy demonstrated synchronous lesions at 3 cm and 40 cm with histological analysis confirming synchronous adenocarcinomata. He developed bilobar hepatic metastases while undergoing neoadjuvant chemoradiotherapy. Treatment was complicated by Fournier's gangrene of the right hemiscrotum which required surgical debridement. Eight months later he re-presented with an ulcerating lesion on the right hemiscrotum. An en-bloc resection of the ulcerating scrotal lesion and underlying testis was performed. Immunohistological analysis revealed metastatic adenocarcinoma of large bowel origin. Colorectal metastasis to the urogenital tract is rare and here we report a case of rectal carcinoma metastasizing to scrotal skin. Colorectal cancer is the second commonest non-cutaneous cancer diagnosed in the Republic of Ireland, with over 1800 new cases annually. At presentation, approximately 35% of patients have advanced disease . MetastaA 72-year-old Caucasian man presented in January 2006 with a two-week history of symptomatic hypochromic, microcytic anaemia of 8 g/dL. He gave a short history of rectal bleeding and tenesmus. Colonoscopy demonstrated an exophytic, circumferential, near-obstructing lesion 3 cm from the anal verge with a synchronous exophytic lesion in the sigmoid colon. Biopsies confirmed the presence of synchronous poorly-differentiated adenocarcinomata of these sites.Radiological staging with endo-anal ultrasound demonstrated a 2.2 cm diameter mass lesion in the rectum extending beyond the rectal wall. Magnetic resonance imaging (MRI) of anorectum confirmed a locally advanced neoplasm with suspicion of involvement of one mesorectal lymph node- Staging pT3N1Mx. Computed tomography (CT) of abdomen and pelvis confirmed a locally-invasive distal rectal neoplasm with no evidence of distant metastases.Due to the development of obstructive symptoms, the patient required a laparoscopic defunctioning loop ileostomy prior to commencing neoadjuvant chemoradiotherapy. Two weeks post-operatively he developed Fournier's gangrene of the right hemiscrotum which was successfully treated by surgical debridement, delaying the commencement of neoadjuvant therapy.Pre-operative chemoradiotherapy consisted of 5,040 centiGray (cGy) delivered in fractions of 180 cGy per day, five days per week, and 5 Fluoro-uracil given in a 120 hour continuous intravenous infusion at a dose of 1,000 mg per square metre of body surface area per day during the first and fifth weeks of radiotherapy. He was scheduled for low anteriorresection six weeks following the completion of chemoradiotherapy.During week five of treatment, July 2006, he was noted to have deranged liver enzymes. Ultrasound scan of abdomen noted suspicious bilobar liver lesions and tri-phasic CT of liver was consistent with the development of significant bilobar liver metastases.He was subsequently treated with the FOLFIRI regimen of palliative chemotherapy . Baseline CT of abdomen and pelvis in November 2006 showed progression of hepatic metastases.In December 2006 he presented acutely with a painless necrotic ulcer affecting the skin of the anterior right hemiscrotum. Ultrasound of scrotum demonstrated a heterogenous solid vascular lesion of the scrotal skin thought to originate in the upper pole of the right testis figure .He underwent radical en-bloc resection of the scrotal skin and underlying testis due to intra-operative suspicion of testicular involvement figure . HistoloImmunohistochemical staining of tumour tissue was positive for carcinoembryonic antigen (CEA) figure  and cytoCutaneous colorectal metastases are rare and signify advanced disease. Metastases in incision sites typically occur in the incision made at time of cancer resection surgery rather than in a pre-existing scar . AbdominWritten informed consent was obtained from the patient's next or kin 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.DM reviewed the case history and prepared the case report. SM reviewed the literature and prepared the final draft. KB, SM, and DM were involved in the surgical care of the patient. RR interpreted the radiological images. IT performed the histological examination of the scrotal specimen and immunohistochemical staining and analysis. PD was involved in oncological care. All authors read and approved the final manuscript.


A 21-year-old man, who had been treated for congenital dilatation of the bile duct 13 years previously, presented with anacute abdomen. The physical examination suggested peritonitis, and an emergent laparotomy was performed. A perforationwas foundin the jejunum approximately 100 cm distal to the ligament of Treitz, followed by resection of a 60-cm jejunalsegment. No tumorous lesions were found during the operation, and the resected jejunal segment showed only focalmyxomatous thickening of the serosa. Despite intensive therapy, he died of uncontrollable septic shock 2 days after theoperation. Unexpectedly, however, histological examination revealed a liposarcoma, showing an unclassifiable histology.From the distribution of the lesion and the histological findings, it is thought that a primary lesion was somewhere else,covered by severe adhesions due to the previous operation, and that the tumor cells spreading from it could have caused thejejunal perforation through vascular involvement. Although extremely rare, liposarcomas in the abdomen can cause intestinalperforation. It is important for both clinicians andpathologists to carefully investigate the cause of an unusual clinicalpresentation such as intestinal perforation.


Primary leiomyoma of the lung is a rare benign tumor that usually presents as a solitary lesion predominantly in young females. Fewer than 100 cases have been reported. Common symptoms include fever, chronic cough, hemoptysis, chest pain, shortness of breath, and pneumonias.A 34-year-old, non-smoker female who presented with recurrent pneumonias. She was found to have a primary leiomyoma of the right middle lobe. This was treated by right middle lobectomy. 6-month follow up showed patient doing well without evidence of residual disease on computerized tomography.Primary pulmonary leiomyoma is a rare tumor distinct from benign metastasizing leiomyoma. Histologic features include absence of mitotic count, low cellularity, lack of cytologic atypia and pleomorphism. Treatment is by conservative surgical resection and carries a favorable prognosis. Primary leiomyoma of the lung is a rare benign tumor of mesodermal origin. It accounts for about 2% of all benign lung tumors . Since iThis case is a 34-year-old Caucasian female nonsmoker who presented with recurrent pneumonias of her right middle lobe. She experienced recurrent fevers, cough, and occasional pleuritic pain for approximately one year. She had no gynecologic issues and her pelvic exam was normal. Chest radiograph showed a bandlike density in the area of the right middle lobe. Computerized tomography (CT) of the chest showed pleural thickening of the inferior half of the right middle fissure with adjacent subsegmental atelectasis of the lateral segment of the right middle lobe with some air bronchograms and bronchiectasis seen within it Figure . BronchoShe was taken to the operating room for a right middle lobectomy that required opening of the right middle lobe bronchus with hand-sewn bronchial closure and mediastinal lymph node sampling. The resected specimen contained a well-circumscribed mass contained within the bronchus attached by a stalk, deep to the actual bronchial margin. Pathology revealed a leiomyoma with acute and chronic inflammation and interstitial fibrosis in the remaining lung tissue Figure . The tumPrimary leiomyoma of the lung is a solitary lesion predominantly in females presenting in the young to middle aged, with a mean age of 35 years . HistoloAnother tumor of the lung with features similar to a leiomyoma is the benign metastasizing leiomyoma. While this tumor is also a benign appearing smooth muscle tumor with an absence of atypia and mitotic count, benign metastasizing leiomyoma frequently presents as multiple lung lesions, commonly in a patient with a history of hysterectomy for benign uterine fibroids .Treatment for all suspected leiomyomas of the lung whether symptomatic or not should be surgical resection for definitive diagnosis. In treating this entity, the surgical philosophy is to conserve tissue. Because of distal bronchiectasis and chronic inflammatory changes, lobectomy and pneumonectomy are common. For more peripheral tumor locations, a less radical resection is appropriate. Bronchoscopic resection is also possible, however the tumor is usually too extensive for this treatment .The lesion must be distinguished from leiomyosarcoma. In contrast to leiomyoma, features of leiomyosarcoma include a well-differentiated smooth muscle lesion with an average mitotic count of 1/10 hpf, with or without increased cellularity or pleomorphism . PrognosBenign pulmonary leiomyoma are rare tumors that are treated with conservative surgical resection. They are distinct from benign metastasizing leiomyoma and leiomyosarcoma. Following resection, they are associated with excellent prognosis.CT: computerized tomography; SMA: smooth muscle actin.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.MV reviewed the case material and literature and was primary author. AK was the primary editor of this report. LP provided pathologic images and information on stains. PG was the primary pathologist analyzing the specimen. ML was the surgeon treating the patient. All authors read and approved the final manuscript.


We present three cases of this rare tumor, describe the functional imaging results, and review the available literature.Mucoepidermoid carcinoma of the tracheobronchial tree is rare. Such tumors usually present with signs and symptoms of bronchial obstruction. Histologically, they can be classified as high-grade or low-grade tumors. Experience of imaging these tumors with 18F-FDG PET-CT and 68Gallium 1,4,7,10-Tetraazacyclododecane-NI,NII,NIII,NIIII,- tetra acetic acid (D) - Phel1-Tyr3-octreotide positron emission tomography-computed tomography (68Ga-DOTATOC PET-CT). 18F-FDG PET-CT revealed mild uptake in all three patients, whereas 68Ga-DOTATOC PET-CT revealed no significant uptake in any patient, making carcinoid tumor unlikely. Results of histopathological examination were consistent with low-grade mucoepidermoid carcinoma in all patients.Three Caucasian patients, two men (21 and 24 years of age) and one woman (14 years of age), with bronchial masses were evaluated by us. All three patients were symptomatic, and underwent a thorough clinical examination, bronchoscopy and biopsy, plain computed tomography, 18F-FDG PET-CT may have a prognostic relevance by predicting the histopathologic differentiation of the tumor.Our study reveals that functional imaging may be helpful in the initial investigation of patients with mucoepidermoid carcinoma.  However, the literature on PET-CT findings in MECs is scanty. We present three cases of this rare tumor with the FDG uptake patterns, and review the available literature.Mucoepidermoid carcinoma (MEC) of the tracheal-bronchial tree is rare, comprising only 0.1% to 0.2% of primary lung malignancies [This was an investigative protocol, which was approved by the ethics committee of our institution, and informed consent was gained for each patient.18F-FDG PET-CT and 68Gallium 1,4,7,10-Tetraazacyclododecane-NI,NII,NIII,NIIII,- tetra acetic acid (D) - Phel1-Tyr3-octreotide (8Ga-DOTATOC) PET-CT using a dedicated PET-CT scanner  to assess the diagnostic value of these methods in evaluating bronchial tumors.All patients presenting with tracheal-bronchial tumors underwent We present three such cases, for which the histologic diagnosis was MEC, and retrospectively analyze their PET-CT findings and histopathologic grading. Their clinical parameters and other details are given in Table 18F-FDG PET-CT scan  of the chest revealed a mass measuring 27 × 16 mm and occluding the left main bronchus. On bronchoscopy, the mass was seen to be occluding the left main bronchus. Results of a bronchoscopic biopsy were inconclusive. The patient underwent a 18F-FDG PET-CT revealed slight uptake (SUVmax 3.2) in the tumor, whereas 68Ga-DOTATOC PET-CT revealed no significant uptake. Histological results of a biopsy taken from the mass were suggestive of low-grade MEC.A 21-year-old Caucasian man presented with a one-year history of cough and dyspnea on exertion. CECT of the chest revealed a 10 × 10 mm mass in the right main bronchus. Bronchoscopic examination revealed a mass in the right main bronchus, which bled on contact. 18F-FDG PET-CT revealed mild uptake in the tumor (SUVmax 3.9), whereas 68Ga-DOTATOC PET-CT revealed no significant uptake. The patient underwent a right pneumonectomy with resection of the carina. Histopathologic examination of the operative specimen revealed features of low-grade MEC.A 24-year-old Caucasian man presented with a one-year history of cough and haemoptysis. On clinical examination, decreased air entry was detected on the right side of the chest. CECT revealed a mass of 35 × 38 mm in the right main bronchus with collapse of the right lung. Bronchoscopic examination revealed a mass starting at the level of the carina, occluding the right main bronchus completely. Histopathological examination of a bronchoscopic biopsy of the mass was suggestive of a neuroendocrine tumor. MEC is an uncommon lesion accounting for under 1% of primary malignant bronchial tumors. Although generally indolent, local invasion and lymph node metastases may occur. The tumor generally affects patients aged over 30 years. Patients usually present with cough, haemoptysis, wheezing and recurrent pneumonia due to bronchial obstruction, but 9% to 28% of cases may be asymptomatic [Histologically, MECs are composed of varying mixtures of mucus-secreting, columnar and goblet cells. They are classified as high-grade or low-grade based on histologic appearance . The histopathologic grading also reflects the prognosis of these tumors .On conventional radiology, MECs usually appear as oval or lobulated, slightly enhanced, endobronchial masses with occasional punctuate calcification. There may be post-obstructive pneumonia and/or peripheral atelectasis .18F-FDG PET-CT scan varies from zero to 6.2 for low-grade MECS and from 2.86 to 23.4 for high-grade MECs. We found uniformly slight uptake (low SUV) on 18F-FDG PET-CT in all three cases, all of which had a histopathologic diagnosis of low-grade MEC -Phel1-Tyr3-octreotide positron emission tomography-computed tomography; CECT: contrast enhanced computed tomography; MEC: mucoepidermoid carcinoma, SUVmax: standardized uptake value.Written informed consent was obtained from patients two, three and from patient one's father 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.TJ conceived the study and made a major contribution in the compilation, analysis, literature review and formatting of the manuscript, AK had a major contribution in the analysis and editing, RK helped in the study design and data acquisition, RD contributed in data analysis, MM helped in the review of the literature. All the authors have read the final manuscript and have approved it.


Intra-abdominal fibromatosis, usually located at the mesenteric level, is a locally invasive tumor of fibrous origin, with no ability to metastasize, but a tendency to recur. Certain non-typical cases of intra-abdominal fibromatosis with involvement of the bowel wall can be misdiagnosed because of their different biological behavior.We describe the case of a 64-year-old Caucasian man presenting with mesenteric fibromatosis and involvement of the bowel wall, who was treated surgically. The macroscopic and microscopic appearance of the lesion mimicked a gastrointestinal stromal tumor, a tumor with potential malignant behavior.It is essential to make an early and correct diagnosis in such equivocal cases, so that the appropriate treatment can be chosen and suitable patients admitted to clinical trials if appropriate. New and reliable criteria for discriminating between intra-abdominal fibromatosis and gastrointestinal stromal tumor should be proposed and established because novel sophisticated therapeutic strategies have been introduced in the international literature. Mesenteric desmoids account for less than 10% of sporadic desmoid tumors and are particularly common in patients with familial adenomatous polyposis (FAP) . PatientGastrointestinal stromal tumors (GISTs) on the other hand originate from gastrointestinal pacemaker Cajal cells, which are the primary effectors controlling gut motility . GISTs mThe enigmatic biology and anatomic location of intra-abdominal fibromatosis (IAF) highlight the need to discriminate between these two diseases. IAF is benign and exclusively locally aggressive, whereas GISTs present with a risk of aggressive clinical behavior, depending on location, diameter and number of mitoses; thus GISTs are potentially malignant and may lead to distant metastases. The fact that IAF and GISTs have different biological behaviors makes treatment recommendations difficult despite the recent introduction of new therapeutic strategies. A significant factor limiting any attempts at generalizing management strategies is the small number of cases available for analysis, reflecting the relative rarity of the disease.A 64-year-old Caucasian man was admitted to our hospital with a ten-year-history of a mild diffuse abdominal pain associated with anorexia. He reported no noticeable weight loss or other symptoms. His family history was unremarkable and he had no history of previous abdominal surgery.On physical examination, we noted a mass in the left upper quadrant of the abdomen, which was mobile. A CT scan of the abdomen revealed a homogeneous, non-enhancing mass, 70 × 100 mm in size, in the mesenteric region near the small bowel, with a consistency suggesting thick mucinous or proteinaceous material, which possibly represented an intestinal wall tumor. There were no other relevant findings.At laparotomy, a solid mass measuring 80 × 100 × 120 mm was identified at the root of the jejunal mesentery, infiltrating the adipose tissue and bowel wall, and in close association with the superior mesenteric and the middle colic vessels  requiring long-term parenteral nutrition . AdditioThe location of a desmoid tumor within the mesentery of the small bowel may complicate the management of patients with FAP and interfere with surgical strategy, preventing proctectomy or ilial pouch-anal anastomosis (IPAA), at least in some patients . It may It is generally easy to diagnose primary IAF in its classic presentation as a mesenteric mass, because of its distinctive gross and microscopic features . HoweverHistological features characteristic of GIST include the presence of spindle or epithelioid cells with variable architecture, mitotic activity and nuclear atypia, and myxoid or hyalinized stroma. Necrosis and hemorrhage can also be seen. By contrast, IAFs are characterized by a spatially homogeneous proliferation of wavy spindle cells without atypia, associated with collagen deposition  and an infiltrative border. These features are sufficiently characteristic of mesenteric fibromatosis to allow distinction from GIST on the basis of routinely stained sections in most cases. In the few equivocal cases, immunohistochemical analysis can provide more precise diagnosis.The distinction between these neoplasms is very important because they have different biological behaviors, and there are important clinical implications for the patient. Complete excision of the tumor with a margin of uninvolved tissue (when this is possible) is the most effective treatment yet described, and it may be followed by other therapies if GIST is diagnosed. After the more radical (free margin) resections required for GISTs, the five-year survival rate is approximately 45%, whereas for metastatic disease it drops to 20% . The tumThe optimum treatment protocol for desmoids tumors has not yet been established and, in many cases, a multidisciplinary approach including surgery, chemotherapy, and radiation therapy is required. The rarity of cases in even major oncological centers has traditionally limited the ability to study this disease. The notion that a specific genotype can predict the development of an aggressive desmoid tumor in a given patient could prove to be valuable in allowing appropriate patient selection for early therapy or even a chemopreventive strategy. Several novel pharmacologic and biologic treatment approaches are actively being developed, although long-term follow-up is needed for their substantiation.The aim of this report is to stress the importance of correctly characterizing a tumor localized in the bowel wall and infiltrating the mesentery to plan the appropriate treatment because tumor diagnoses based on immunohistochemical staining or traditional histologic criteria alone are not specific enough.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.The authors declare that they have no competing interests.CIF was the patient's surgeon, and was involved in drafting the manuscript and critically revising it for important intellectual content. EPM, CNS, BGS, PPP and CKF have made contributions to conception and design. CNS contributed to the analysis and interpretation of data and wrote the paper. All authors read and approved the final manuscript. All authors contributed equally to the final draft of the manuscript. CIF has given the final approval of the version to be published.


Gastrointestinal tract small cell carcinoma is an infrequent and aggressive neoplasm that represents 0.1–1% of gastrointestinal malignancies. Very few cases of small cell esophageal carcinoma arising in Barrett's esophagus have been reported in the literature. An extremely rare case of primary small cell carcinoma of the distal third of the esophagus arising from dysplastic Barrett's esophagus is herein presented.th postoperative day. Histopathology revealed a primary small cell carcinoma of the distal third of the esophagus arising from dysplastic Barrett's esophagus. The patient received another 3 month course of postoperative chemotherapy with the same agents and remained free of disease at 12 month review.A 62-year-old man with gastroesophageal reflux history presented with epigastric pain, epigastric fullness, dysphagia, anorexia, and weight loss. Esophagogastroscopy revealed an ulceroproliferative, intraluminar mass in the distal esophagus obstructing the esophageal lumen. Biopsy showed small cell esophageal carcinoma. Contrast-enhanced chest and abdominal computed tomography demonstrated a large tumor of the distal third of the esophagus without any lymphadenopathy or distant metastasis. Preoperative chemotherapy with cisplatine and etoposide for 3 months resulted in a significant reduction of the tumor. After en block esophagectomy with two field lymph node dissection, proximal gastrectomy, and cervical esophagogastric anastomosis, the patient was discharged on the 14Although small cell esophageal carcinoma is rare and its association with dysplastic Barrett's esophagus is extremely infrequent, the high carcinogenic risk of Barrett's epithelium should be kept in mind. Prognosis is quite unfavorable; a better prognosis might be possible with early diagnosis and treatment strategies incorporating chemotherapy along with oncological radical surgery and/or radiotherapy as part of a multimodality approach. Since treatment protocols are not well established due to the rarity of the neoplasm, multi-institutional studies are needed to obtain sufficiently large populations for investigation and optimization of therapy of the disease. Gastrointestinal tract small cell carcinoma is an infrequent and aggressive malignancy. It represents 0.1–1% of gastrointestinal malignancies, with the esophagus being the most common primary site . Small cSince McKeown's first description in 1952 , few casA 62-year-old man with a history of gastroesophageal reflux disease presented with a one month history of epigastric pain, epigastric fullness, dysphagia, anorexia, and weight loss of 9 kgs. Clinical examination and blood tests were normal apart from elevated carcinoembryonic antigen . Chest x-ray, electrocardiogram, echocardiography, and abdominal ultrasound (US) were normal. Esophagogastroscopy revealed an ulceroproliferative, intraluminar mass in the distal third of the esophagus obstructing the esophageal lumen. Biopsy showed small cell esophageal carcinoma. Due to esophageal lumen obstruction, endoscopic US was not feasible. Contrast-enhanced chest and abdominal computed tomography (CT) demonstrated a large tumor of the distal third of the esophagus without any lymphadenopathy or distant metastasis Figure .th postoperative day.The patient received preoperative chemotherapy with cisplatine and etoposide for 3 months with a significant reduction of the tumor size Figure . En blocHistopathology revealed a tumor (2.5 × 1.5 cm), 2 cm above the gastroesophageal junction, that was a small cell esophageal carcinoma infiltrating the submucosal and, focally, the muscular layer Figure . The marA diagnosis of primary small cell carcinoma of the distal third of the esophagus arising from dysplastic Barrett's esophagus was made. The patient received another 3 month course of postoperative chemotherapy with the same agents (cisplatine and etoposide) and was free of disease at review after 12 months.Small cell esophageal carcinoma is a rare neoplasm ,2. AlthoSmall cell esophageal carcinoma is an exceptional finding in patients with Barrett's esophagus -7. The pThe most common symptoms are dysphagia, anorexia, weight loss, chest pain, and regurgitation ,2,6. TheAlthough treatment protocols are not well established because of the paucity of cases and the lack of large studies, chemotherapy remains the treatment of choice given the systemic nature of the disease ,2. SinceThe reported patient presented with a large tumor of the distal esophagus. Our initial treatment consisted of a 3 month course of preoperative chemotherapy with cisplatine and etoposide that resulted in a significant reduction of the neoplasm. Given the response to chemotherapy, and since no metastatic disease was identified in the post-chemotherapy investigation, radical surgical resection, including en block esophagectomy with two field lymph node dissection and proximal gastrectomy, was then performed. Based on the promising results of preoperative chemotherapy, another 3 month course of postoperative chemotherapy with the same agents was administered. Although conclusions regarding treatment of such a rare clinical entity cannot be drawn from a case report, the effects of our treatment strategy seem encouraging since our patient remained free of disease at review at 12 months.Small cell esophageal carcinoma is rare and its association with dysplastic Barrett's esophagus is extremely infrequent. The high carcinogenic risk of Barrett's epithelium, though, should be kept in mind. Prognosis is quite unfavorable and treatment protocols are not well established. A better prognosis might be possible with early diagnosis and treatment strategies incorporating chemotherapy along with oncological radical surgery and/or radiotherapy as part of a multimodality approach. Our treatment strategy of preoperative chemotherapy followed by radical surgical resection and postoperative chemotherapy in the reported patient may have yielded promising results. Multi-institutional studies are needed to obtain sufficiently large populations for investigation and optimization of therapy of the disease.The author(s) declare that they have no competing interests.HM contributed to manuscript conception, research, acquisition of data, drafting and writing of the manuscript. DT carried out the operation and contributed to acquisition of consent and critical review of the manuscript. KGT assisted in the operation, contributed to organising and drafting of the manuscript, and critically revised the manuscript. AL contributed to manuscript conception, research, acquisition of data, drafting and writing of the manuscript. MP contributed to manuscript conception, research, acquisition of data, drafting and writing of the manuscript. AB carried out the histopathologic evaluation and contributed to writing of the manuscript. PP contributed to the preoperative and postoperative management of the patient and to critical review of the manuscript. KF contributed to organising and drafting of the manuscript, and critically revised the manuscript. SK assisted in the operation and contributed to critical review of the manuscript.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.


Malignant degeneration in association with orthopaedic implants is a known but rare complication. To our knowledge, no case of osseous malignant fibrous histiocytoma after anterior cruciate ligament reconstruction is reported in the literature.We report a 29-year-old male Turkish patient who presented with severe pain in the operated knee joint 40 months after arthroscopic anterior cruciate ligament reconstruction. X-ray and MR imaging showed a large destructive tumor in the medial femoral condyle. Biopsy determined a malignant fibrous histiocytoma. After neoadjuvant chemotherapy, wide tumor resection and distal femur reconstruction with a silver-coated non-cemented tumor knee joint prosthesis was performed. Adjuvant chemotherapy was continued according to the EURAMOS 1 protocol.Though secondary malignant degeneration after orthopaedic implants or prostheses is not very likely, the attending physician should take this into consideration, especially if symptoms worsen severely over a short period of time. The continuous increase in recreational sports leads to a continuously increasing number of capsule and ligament injuries of the knee joint as well. About 20% of the knee injuries are accompanied by anterior cruciate ligament (ACL) ruptures . Reconstth decade of life; men are affected more often than women [a known complication. Secondary malignant fibrous histiocytoma has been desribed at sites of pre-existing bone lesions [The primary malignant fibrous histiocytoma (MFH) has been described for the first time as a soft tissue tumor in 1964 by O'Brien and Stout . MFH occan women . The devan women  is a rar lesions , after i lesions .In the case history presented below, we report the manifestation of an osseous MFH at the distal femur 40 months after arthroscopic anterior cruciate ligament reconstruction using autologous semitendinosus tendon. To our knowledge, no case of an osseous MFH after anterior cruciate ligament reconstruction is reported in the literature.In January 2006, a 26-year-old male had sustained an isolated rupture of the right cruciate ligament during a soccer game. This was diagnosed by a stability investigation with a positive Lachman and Pivot shift test and was confirmed by MRI. The knee joint was without signs of irritation. ROM was 0°-140°. Ten days later, arthroscopic ACL reconstruction was performed using a quadruple autologous ipsilateral semitendinosus tendon graft. The fixation elements used were a titanium Endobutton  on the femoral side and a titanium Suture Disc  on the tibial side, respectively. The peri- and postsurgical course was free of complications. 10 months after ligament reconstruction, the patient could resume sports activity and was very satisfied with the operative outcome. 40 months after surgery, the patient presented with strongly increasing pain in the medial femoral condyle of the operated knee joint. Clinical investigation showed a stable knee without signs of soft tissue irritation. Non-contrast radiology showed a space-occupying lesion of 5.6 × 5 cm at the medial femoral condyle, mainly located in the metaphysis Figure  and 1b. MRI with contrast agent (Gadolinium) revealed a bone tumor of 6.2 × 5 × 4.2 cm. The bone tumor had infiltrated the medial retinaculum and the vastus medialis muscle Figure  and 2b. An en bloc-resection of the distal femur 12 cm proximal to the knee joint including adjacent joint capsule and tumor-infiltrated soft tissue was performed. Resection was performed 1.5 cm below the tibial plateau. For subsequent distal femur reconstruction, a silver-coated non-cemented modular knee joint prosthesis  was used Figure.  and 4b. Osseous malignant fibrous histiocytoma has been described for the first time in 1972 by Feldman and Norman . Its defIn the literature, only 2 cases of malignant degeneration after ACL have been described so far. Sirveaux et al.  reportedIf a former trauma can favour MFH development remains unclear. Bader et al.  reportedAs the five-year survival rate is 10 to 30% if the MFH is treated only locally, neoadjuvant and adjuvant chemotherapy was performed analogous to the treatment of osteosarcoma in several studies . The mulMFH: malignant fibrous histiocytoma; MRI: magnetic resonance imaging; EURAMOS: The European and American Osteosarcome Study Group; MUTARS: Modular Universal Tumor And Revision System; Bq: Becquerel; Tc: Technetium; HDP: hydroxymethylene diphosphonate; HE: haematoxylin-eosin; ROM: range of motion; ACL: anterior cruciate ligamentThe authors declare that they have no competing interests.TE and JS were the major contributors in writing this manuscript. PR was the referent pathologist for this case. TJH diagnosed, investigated, followed up and managed the patient. TE, SFW and MDS performed the surgery on the patient. All authors read and 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.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2407/10/264/prepub


We report a case of a granular cell tumor colliding with intramucosal adenocarcinoma of the esophagus. A 58-year-old white was found to have a 5 mm nodule in the distal esophagus detected by upper gastrointestinal endoscopy performed as part of the workup of long standing reflux. Endoscopic biopsies revealed intramucosal adenocarcinoma arising in the setting of Barrett’s esophagus. The adenocarcinoma infiltrated a granular cell tumor also present at the nodular site. Endoscopic mucosal resection using Duette band ligation and hot snare electrocautery was performed. Margins were negative for both tumors, and endoscopic surveillance for recurrence is planned. Granular cell tumors (GCTs) are relatively uncommon, usually benign neoplasms, which may arise anywhere in the body . About 3A 58-year-old white man was found to have a 5 mm nodule in the distal esophagus 1 cm proximal to the Z-line when he underwent upper endoscopy during the workup of long standing reflux esophagitis. Biopsies from the lesion showed a granular cell tumor and Barrett’s esophagus without dysplasia. On follow up endoscopy with biopsies, adenocarcinoma was diagnosed arising in the setting of Barrett’s esophagus and colliding with a granular cell tumor -5. The pOn final pathology, the adenocarcinoma was confined to the mucosa, and margins were negative for both adenocarcinoma and granular cell tumor. Repeat endoscopy 8 weeks following mucosal resection revealed no evidence of a visible lesion at the site of the resected nodule. Biopsies from the esophagogastric junction revealed intestinal metaplasia without dysplasia. He will continue his outpatient surveillance at the referring institution, with our recommendation being endoscopy at 6 month intervals for 2 years, with interval increase to 2 year surveillance if no dysplasia is detected during the initial screening period.Granular cell tumors are uncommon tumors, and only 1%-8% of them originate in the gastrointestinal tract. Esophageal GCTs account for about one third of all gastrointestinal GCTs . In a reEsophgeal GCTs are usually discovered incidentally during upper gastrointestinal endoscopy performed for other reasons. If symptoms are present; they are unrelated to tumor development in 50%-90% of patients ,6. EndosThere is an interesting but unexplained association between GCTs and other malignant neoplasms. Strong et al. reported that the incidence of this association was 9% . The coeThe optimal treatment for GCTs remains controversial, however, the current treatment options are as follows: a conservative approach with regular endoscopic follow-up for tumors <10 mm in diameter without evidence of malignant change , and surEMR was recently reported to be an effective treatment in lesions similar in size to this case . EUS wasThe risk of regional metastatic disease with intramucosal cancer is on the order of 7-10% and this approaches the risk of major surgical morbidity and mortality in many centers ,21. Ther


Primary malignant pericardial mesothelioma is a very rare pericardial tumor of unknown etiology.A 61-year-old Caucasian woman was admitted to our hospital complaining of exertional dyspnea due to a large pericardial effusion. Intrapericardial fluid volume declined after repeated pericardiocentesis, but the patient progressively developed a hemodynamically relevant pericardial constriction. Pericardiectomy revealed a pericardial mesothelioma. Subsequently, four cycles of chemotherapy  were administered. The patient remained asymptomatic, and there was no recurrence of the tumor after three years.Pericardial mesothelioma should be considered and managed appropriately in non-responders to pericardiocentesis, and in patients who develop constrictive pericarditis late in their clinical course. Primary malignant pericardial mesothelioma is a very rare pericardial tumor of unknown etiology.A 61-year-old Caucasian woman was admitted to our hospital complaining of exertional dyspnea (NYHA III) and chest pain. Transthoracic echocardiography demonstrated a large pericardial effusion. Pericardiocentesis revealed 1500 ml of an acellular, sterile pericardial effusion and symptoms were markedly relieved.The patient was re-admitted three months later, and transthoracic echocardiography showed a recurrent large pericardial effusion with partly organized fibrinous structures inside the effusion. There were no signs of cardiac tamponade, but there was a thickened right ventricular pericardium  scan demonstrated an intrapericardial accumulation of the tracer, indicating a local infection or a tumor Figure  [1].The patient's level of intrapericardial fluid declined after repeated pericardiocentesis, and cytology of the pericardial fluid revealed signs of chronic infection, but no malignant mesothelial cells. Subsequently, the patient developed a hemodynamically relevant pericardial constriction (SD5). Therefore, a partial pericardiectomy was performed, and histological examination Figures  and 4b rThis was considered to be a PMPM because no signs of a pleural mesothelioma were found. Despite the above-mentioned findings of the magnetic resonance imaging (MRI) scan of the chest, FDG-PET, echocardiography and pericardiocentesis, we suspected PMPM but could not definitively declare a preoperative diagnosis of PMPM.Subsequently, four cycles of chemotherapy with pemetrexed and cisplatin  were administered, and remission was achieved -5]. The. The5]. Diagnosis of pericardial diseases can be challenging and often requires a multimodal imaging approach including echocardiography, MRI, CT and FDG-PET scans ,7. The mThere is some recent evidence that asbestos may have a harmful effect on pericardial serosa. However, there has not yet been any definite proven association between asbestos exposure and pericardial disease ,8-10]. . 10]. InPMPM is often discovered late during a patient's clinical course or at autopsy. Frequent clinical diagnoses refer mainly to acute pericarditis, constrictive pericarditis, and cardiac tamponade and sometimes to various types of coronary heart disease.Surgical resection remains the main treatment modality in PMPM. The prognosis of this disease remains extremely poor due to its late presentation, inability of complete tumor eradication by surgery and the poor response of PMPM to radiotherapy or chemotherapy. A median survival time from the onset of symptoms is six months -10]. Re. Re10]. PMPM should be considered and managed appropriately in non-responders to pericardiocentesis or pericardial window for treatment of pericardial effusion or tamponade, and in patients who develop constrictive pericarditis late in their clinical course.d-glucose; FDG-PET: F-18 fluorodeoxyglucose positron emission tomography; MRI: magnetic resonance imaging; PMPM: primary malignant pericardial mesothelioma.CT: computer tomography; FDG: 2-fluoro-2-deoxy-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 authors declare that they have no competing interests.TB, LF, CL, AM, GP, HJT, DH and CP analyzed and interpreted the patient data regarding the cardiologic disease, therapy and the echocardiographic diagnostic. TB was a major contributor in writing the manuscript. JK analyzed and interpreted the magnetic resonance imaging; OL analyzed and interpreted the FDG-PET. AT and KMM performed the histological examination of the tumor. All authors read and approved the final manuscript.Movie 1 in Movie 2 in Movie 3 in Movie 4 in Movie 5 in Click here for fileClick here for fileClick here for fileClick here for fileClick here for file


Staphylococcus aureus remains the commonest pathogen. We report an extremely rare case of PPA caused by Streptococcus milleri. Only one case has been reported in literature so far.Primary Psoas abscess (PPA) is an infrequent clinical entity with obscure pathogenesis and vague clinical presentation. High index of clinical suspicion is required for the diagnosis of psoas abscess. We also emphasises the importance of bacteriological confirmation of microorganism involved, although  Staphylococcus aureus (SA). We report an extremely rare case of PPA caused by Streptococcus milleri (SM). Only one case has been reported in literature as far as our knowledge [Primary psoas abscess (PPA) is extremely rare clinical entity, which potentially carries high mortality and morbidity if diagnosis is delayed. A PPA has no obvious focus of infection, with the commonest pathogen being nowledge . It carr9/l. Plain abdominal radiograph showed a soft tissue shadow on right upper quadrant. Ultrasonography (USS) guided aspiration was undertaken and 250 ml of thick brown blood stained pus was drained and sent for microscopy and culture. Drain was kept in situ. Microbiology reported SM as causative organism sensitive to penicillin and erythromycin. Patient was started on intravenous 1.8 g qds benzyl penicillin for 4 weeks. Computed Tomography (CT) and barium enema was performed to rule out bowel origin  declare that they have no competing interests.All authors were involved in the care of the patient. NBB drafted the manuscript. AMSA, SJ critically reviewed and improved the manuscript. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for participation in this research.A copy of the written consent is available for review by the Editor-in-Chief of this journal.


We report a case of extraskeletal osteosarcoma (ESOS) and autopsy findings. A 35-year-old man presented with an ossified tumor in the right thigh and lung metastasis. The lung tumors continued to develop despite multiagent chemotherapy and caused death within 8 months. Autopsy revealed many secondary lesions in the lungs, especially in the left lung. Histopathologically, the primary tumor and one of the secondary tumors showed proliferation of spindle-shaped tumor cells focally forming lace-like osteoid material. Therefore, we made a definite diagnosis of ESOS. Extraskeletal osteosarcoma (ESOS) is a rare malignant mesenchymal neoplasm in soft tissues but not directly attached to the skeletal system. It accounts for less than 4% of all osteosarcomas and approximately 1.2% of all soft tissue sarcomas . Here, wA 35-year-old man presented with a swelling in his right thigh, which he had first noticed one month before. His medical history was unremarkable. On physical examination, a well-circumscribed, elastic-firm soft tissue mass was palpated. The mass was attached to deep fascia and was not mobile. Its maximum width was about 16 cm. The skin over the swelling was slightly reddish and showed no signs or either ulceration or necrosis, but was varicosed. Laboratory tests revealed slight increase in the white blood cell count (8800) and alkaline leukocyte phosphatase (ALP) (330), but no other abnormalities.Plain radiography of the right thigh showed a large soft tissue mass with marked calcification in a radial pattern . ComputePlain chest radiography revealed 8 nodular densities in the left lung and 6 in the right, which were considered to be metastasis . The larRegarding the findings described above, the biopsy site of the tumor in the right thigh was chosen to contain calcification and well-enhanced areas with gadolinium both of which were considered as signs of tumor aggressiveness. Indeed, the biopsy site chosen included the antero-lateral and distal sections of the tumor. The deep portion of the tumor was excised along with the peripheral portion so as to obtain both calcification and noncalcification areas.An open biopsy specimen had malignant osteoid in lace-like formations of long or short spindle cells with osteogenesis and chondrogenesis. Hypercellularity and cellular pleomorphism were observed. The number of mitotic figures was seen about five mitosis/10 high-power fields (HPF). Immunohistochemically, the tumor showed reactivity with antibodies against vimentin, desmin, and CD68. The MIB-1 labeling index was 30–40%, and S-100, PAS, SMA, EMA, and keratin showed no reactivity. Thus, the tumor was diagnosed as ESOS.Multiple metastases in both lungs were considered as inoperable. Moreover, from the aspect of pain control, there is little point in excising the primary tumor because the patient did not feel any pain. Therefore chemotherapy was primarily chosen as palliative treatment. The chemotherapy regimen referred to Kanazawa University protocol  was modi2, doxorubicin 250 mg/m2 and ifosfamide 16 g/m2, and mesna 9.6 g/m2, respectively. Complications including elevation of liver enzymes, hemorrhagic cystitis, auditory disorder, and pancytopenia were observed. In cases with chemotherapy induced severe pancytopenia, the dose of doxorubicin was reduced in the subsequent cycle. The doses of the antitumor agents were as follows: first and second cycles: cisplatin 460 mg, doxorubicin 114 mg and caffeine 14.4 g; third cycle: doxorubicin 106 mg, ifosfamide 17.5 g and mesna 10.5 g. Total doses administered were cisplatin 520 mg/mResponse to chemotherapy was evaluated after three cycles in 2 months. According to RECIST , the tumor in the thigh was regarded as a stable disease (SD) and the pulmonary lesion as a progressive disease (PD). There were no indications for surgery, and the patient and his family desired more chemotherapy. Therefore, one more chemotherapy cycle  was given, and the response was the same as the previous cycles. Nevertheless, the pulmonary lesion maintained its momentum conclusively occupying the left lung  and causAn autopsy was performed. The tumor was white and about 15 × 10 × 10 cm in diameter. The underlying bone periosteum was not involved. There were numerous pulmonary metastatic lesions, especially in the left lung. Both lungs were almost collapsed. No other metastasis was found. Histopathological sections of the thigh tumor and one of the pulmonary lesions showed proliferation of spindle-shaped tumor cells focally forming a lace-like osteoid material. The other pulmonary metastasis sites did not have any osteoid material . We madeESOS is a relatively rare, high-grade malignant soft tissue neoplasm. The 5-year survival rate is as low as 25% to 37% . It occu Mechanical injury has been considered as a causative agent . As withMicroscopically, unlike in myositis ossificans where the most mature portion is located at the periphery, in ESOS, there is often a “reverse zoning phenomenon”, that is, central deposition of osteoid material and atypical spindle cell proliferation at the periphery. As with this case, reverse zoning phenomenon was observed. ESOS is generally a high-grade sarcoma and seems to be relatively chemoresistant, whereas osteosarcoma of bone is generally considered to be a relatively chemosensitive form of sarcoma . FurtherAutopsy revealed metastasis only in lungs, and no metastasis was found elsewhere. Sordillo et al. reported that distant metastasis was found in 36 of 48 patients with ESOS, and the metastasis was to the lungs in all 36 cases . TherefoWe have reported a case of extraskeletal osteosarcoma and autopsy findings. It had already metastasized to the lungs at the first visit to our hospital and did not metastasize to any other locations. The lung tumors continue to develop despite multi-agent chemotherapy, causing death within 8 months. One of the secondary tumors contained osteoid material.