Cavernous hemangiomas of the adrenal gland are rare, benign, non-functioning neoplastic tumors. To our knowledge, 55 cases have been reported in the literature to date.We report the first case of a large, non-functioning adrenal cavernous hemangioma that was incidentally found during the preoperative staging workup of a 75 year old woman with left breast adenocarcinoma. Imaging with US, CT scan and MRI showed a heterogeneous 8 cm mass with non-specific radiological features that was located on the left adrenal gland. The mass was surgically excised and pathology revealed an adrenal hemangioma with areas of extramedullar hemopoiesis.Although adrenal hemangiomas are rare and their preoperative diagnosis is difficult, they should always be included in the differential diagnosis of adrenal neoplasms. Adrenals are an infrequent location for benign vascular tumors like cavernous hemangiomas-such tumors are most commonly situated on the skin or in the liver. Their clinical presentation is usually vague, with non-specific abdominal pain being the predominant symptom. Frequently, they are discovered as incidentalomas either during imaging or in autopsies. Since 1955, when Johnson and Jeppesen described the first adrenal cavernous hemangioma, only 55 cases have been reported in the literature [A 75 year old female patient with breast cancer was admitted to our hospital for surgical treatment. Her preoperative staging workup with an abdominal ultrasound, revealed a heterogeneous solid lesion of the left adrenal gland. Clinical examination and laboratory tests, including adrenal hormonal levels , were normal. Abdominal CT scan showed a well-defined, heterogeneous, retroperitoneal mass with speckled calcifications that measured 8 cm and was located on the left adrenal gland. After bolus IV injection of contrast medium the tumor showed irregular enhancement. On subsequent MRI, the tumor demonstrated hyperintensity on both T1- and T2-weighted images with fat component and irregular peripheral enhancement (Figure During the same operation, the patient underwent a left adrenalectomy through a left subcostal incision followed by modified radical left mastectomy. Her postoperative course was uneventful and she was discharged five days later.On gross examination, the adrenal tumor appeared as a red tan mass measuring 8 cm × 6 cm × 4 cm. Focal red-purple hemorrhagic and cystic areas were present, along with diffuse calcifications. Normal adrenal gland parenchyma was noted on the surface of the mass Figure .Microscopically, dilated, blood filled vascular spaces were observed. The spaces were lined by a single layer of thin endothelial cells with collagenous walls Figure . InteresThe histological diagnosis was that of an adrenal cavernous hemangioma with coexistence of extramedullar hemopoiesis and no signs of malignancy.The pathology report on the breast specimen showed a grade II infiltrating tubular adenocarcinoma, measuring 5 cm in greatest diameter. None of the 13 excised lymph nodes had signs of malignancy.The evolution of radiological imaging in the last 20 years resulted in increased detection rate of clinically inapparent adrenal masses, also known as adrenal incidentalomas. It is estimated that adrenal masses are an accidental finding in 1–5% of all abdominal CT scans performed. Adrenal hemangiomas, however, are extremely rare, and their differential diagnosis preoperatively is rather challenging.Adrenal hemangiomas are most usually cavernous, unilateral lesions of the adrenal glands that appear in the sixth or seventh decade of life, with a 2:1 female to male ratio . Their sDistinguishing a large adrenal hemangioma from other lesions of the adrenal glands, and especially from malignant tumors, can be very difficult. In most cases the final diagnosis is made by histopathology after surgical resection. However, there are some radiological features that, although not entirely specific, should raise the suspicion of adrenal haemangioma. CT scans usually display a characteristic peripheral patchy enhancement with progression to the centre of the tumor that is a common finding . SpeckleMRI has been proven to be the best diagnostic tool so far. The most characteristic finding is the peripheral spotty and centripetal enhancement on dynamic studies. Marked hyperintensity on T2-weighted images in combination with focal hyperintensity in T1-weighted images, indicate areas of calcification and haemorrhage that are associated with adrenal hemangiomas ,18. AngiThe surgical indication for excision of the tumor is the size. Adrenal incidentalomas larger than 6 cm in diameter must be excised because the risk of adrenal cancer is 35% to 98%. For lesions measuring 4 cm to 6 cm, other imaging features, history of extra-adrenal malignancy, patient's preference, age and comorbitities should be taken into consideration. Adrenalectomy and follow-up with imaging are both acceptable in such cases [Adrenalectomy can be performed laparoscopically for lesions measuring less than 6 cm ,19. LargWe presented a rare coexistence of an adrenal cavernous hemangioma with extramedullar hemopoietic tissue in a woman treated for breast cancer. Although rare, adrenal haemangioma should be included in the differential diagnosis of adrenal neoplasms. The main indication for surgical removal of an adrenal mass is its size. However, the risks of haemorrhage, necrosis and thrombosis necessitate surgical excision in most of the cases, especially for tumors more than 3 cm.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.NA was responsible for critical revision of scientific content. MK drafted the manuscript. AIY participated in the design of the manuscript and helped to draft the manuscript. VKS contributed substantially to manuscript conception and design. TT assisted in the preparation of the manuscript.ND participated in the acquisition of data and preparation of the manuscript. VS was the surgeon, approved the final version of the manuscript for publication. AKP performed histopathological and immunohistochemical analysis and contributed substantially to pathology content. All authors read and approved the final manuscript.


Hepatoblastoma (HB) is a rare malignant tumour of the liver and usually occurs in the first three years of life. Most of these tumours arise in the embryo; hence it seems to be unusual that hepatoblastoma occurs in adults and is an exceptional cause of primary malignant liver tumour in adult patients. The diagnosis is often overlooked, and patients might be diagnosed at late stages of the disease at risk of increased mortality. In this paper we report a case of a 30-year-old man with mixed hepatoblastoma and abdominal pain, hepatomegaly and fever. The patient under went noninvasive diagnostic methods: duplex scanning, Computed Tomography (CT), and Magnetic Resonance imaging (MRI). In our experience, despite the important role of histological sample provided by biopsy in defining the diagnosis, very important was the role of MRI, more than ultrasonography and enhanced CT. The MRI detects tumor features as size, margins, and ratio with neighboring organs in order to get the best surgical approach. Epithelial carcinomas of liver are among the most frequent malignant tumours that occur in adults . Far morHepatoblastoma (HB) is a rare malignant tumour of the liver and usually occurs in the first three years of life . Most ofHowever, the existence of HB in adult patients has been refuted by some authors, with nonspecific initial symptoms and difficulty in discerning abnormalities in laboratory data of the patients , 7. ConsIn this paper we report a case of mixed hepatoblastoma, diagnosed in our department of diagnostic imaging, in a young adult patient with abdominal pain, hepatomegaly, and fever.A 30-year-old man had been suffering from right hypochondriac pain and fever. He had no history of the disease and had not received blood transfusion. Furthermore, there was not family history of liver disease. Physical examination revealed that the liver was palpable 5 cm below the right costal margin. Laboratory data showed the following values: aspartate aminotransferase (AST) 23 IU/L; alanine aminotransferase (ALT) 18 IU/L; total cholesterol 154 mg/dL; negative hepatitis B surface antigen, antihepatitis B antibody, and antihepatitis C antibody; alpha-fetoprotein (AFP) 45 ng/mL and total bilirubina 1,71 mg/dL, PCR 54, 50 mg/L (0–5), VES 55 mm/h (2–25). There was no evidence of liver cirrhosis. Blood cultures and screening for tuberculosis were negative.The patient under went noninvasive diagnostic methods: duplex scanning, Computed tomography (CT) and Magnetic Resonance imaging (MRI).The Duplex scanning showed hepatomegaly  with presence of heterogeneous mass in the right lobe. The mass was hyperechoic with some calcifications and few anechoic foci, secondary to haemorrhage and necrotic processes .The color Doppler ultrasonography (US-CD) demonstrated normal patency of sovrahepatic vessels.Subsequently the patient was submitted to Computed Tomography (CT) of abdomen . Images were acquired without and with an intravenously contrast media , and scans were obtained with threephases from the injection of contrast media. The CT confirmed a mass that occupied almost the whole right lobe of the liver with lower attenuation than surrounding liver on nonenhanced scan. After the injection of the contrast media, it was assessed that the lesion was predominantly of lower attenuation with some calcifications and small slit like or round and lower density areas, corresponding to haemorrhage or necrosis. Contrast enhancement was low and heterogeneous .Magnetic Resonance imaging (MRI) with contrast media demonstrated a 23 × 14 × 13 cm mass in the right lobe. This mass presented a well-defined capsule. T1-weighted images showed heterogeneous global intensity with areas with intensity signal similar to haemoglobin metabolites, areas with signal intensity similar to calcifications and area with signal intensity similar to fat. On T2-weighted images the lesion was hyperintense but heterogeneous because of hypointense areas corresponding to calcifications and bands corresponding to area of lobulation and fibrosis. Furthermore right kidney was displaced posteriorly by the mass with its vascular pedicle .Compressive effects of the mass on sovrahepatic veins were observed without occlusion, while gallbladder was displaced but not infiltrated. Related to MRI features was supposed a hepatoblastic nature of the lesion.Subsequent diagnostic imaging phase, a bioptic sample was obtained with a CT-guided biopsy with 18 G tru-cut needle. Histopathologic examination revealed the presence of mesenchymal and epithelial cells in bioptic specimen and confirmed the diagnosis of a mixed hepatoblastoma .After results of biopsy, surgical intervention was performed in exploratory to resect the tumour. A right trisegmentectomy was performed with tumour grossly resected with microscopic residual disease. The surgical diagnosis confirmed the diagnosis of a mixed hepatoblastoma. No postprocedural complications were observed. He is receiving the VI cycles of systemic chemotherapy whit adriamycin and cisplatin which results in good health.HB is the most common primary malignant liver neoplasm in children , 9. Apprβ-catenin accumulation implies an oncogene alteration of the wnt/β-catenin pathway. Furthermore, nuclear p53 accumulation indicates that p53 mutation is also involved in the molecular pathogenesis of the malignancy [The etiology of HB has been elusive. Present investigations of the cytogenetic and molecular genetic abnormalities in HB revealed involvement of chromosomal loci on 1q, 2 (or 2q), 4q, 8 (or 8q), and 20. Loss of heterozygosity imprinting at locus 11p 15.5 also suggests a common genetic basis for HB . The detlignancy , 16, 17 However, cirrhosis of the liver has been seen in association with adult HB in 30% of cases , 18. In This would imply that these tumours may have a different pathogenetic pathway in adults compared to children. Ishank and Glunz classified hepatoblastomas into two groups: epithelial type and mixed epithelial and mesenchymal type .The epithelial type consists of fetal and embryonic cells presenting alone or in combination; in the epithelio-mesenchymal mixed type, mesenchymal elements are present along with the epithelial component . For a lthrive, loss of weight, and a rapidly enlarging upper abdominal mass. The serum AFP level is almost invariably high [In adults, the morbidity of HB is extremely rare, and the initial symptoms are nonspecific so that the diagnosis is often overlooked. The usual presentation is failure to bly high , such asThe initial diagnosis of HB is mainly based on imaging. Proper diagnosis, staging, and treatment of HB require accurate imaging studies. Ultrasound (US) is a noninvasive modality that is particularly useful in the evaluation of infants. HB is seen as a hyperechoic, solid, intrahepatic mass on US . Other sThe complete surgical resection is the cornerstone of treatment for patients with HB and is the only chance of an optimal clinical result. Despite this, the improvements in survival that have occurred over the last three decades have been the function of standardized chemotherapy that reduces tumor size and enables complete tumor excision, even permitting cure in the presence of initially unresectable or metastatic disease . ChemothIn our experience, despite the important role of histological sample provided by biopsy in defining diagnosis, very important is the role of MRI, more than ultrasonography and enhanced CT, in depicting tumor features as size, margins, and ratio with neighboring organs in order to the best surgical approach.


Primary sacral tumors are rare and experience related to accompanying effects of these tumors is therefore limited to observations on a small number of patients.In this case report we present a patient with a history of primary sacral chondrosarcoma, an infection of an implanted spinal stabilization device and discuss the challenges that resulted from a colonic fistula associated with large, life threatening abscesses as late complications of radiotherapy.In patients with sacral tumors enterocutaneous fistulas after free musculotaneous free flaps transfer are rare and can occur in the setting of surgical damage followed by radiotherapy or advanced disease. They are associated with prolonged morbidity and high mortality. Identification of high-risk patients and management of fistulas at an early stage may delay the need for subsequent therapy and decrease morbidity. Primary sacral tumors are rare and experience related to accompanying effects of these tumors is therefore limited to observations on a small number of patients ,2. TheseSacral neoplasms cause mild but noticeable symptoms at an early stage. In these cases it is essential to achieve the right diagnosis in time for wide excision margins. A radical surgical approach with partial or total sacrectomy, including sacrifice of sacral roots and spinal-pelvic fixation, is technically challenging and may jeopardize axial stability. Surgical approaches are therefore often limited by the size of the tumor and additionally dictated by the proximity to vital structures. As a consequence a resection in sano is feasible only up to a certain size of the tumor.By the time of diagnosis sacral tumors are often too large for achieving adequate margins. Although chondrosarcomas are reported to have low radio-sensitivity, local control is sometimes achieved through radiation in patients who have not been radically resected -9. NonetIn this case report we present a patient with a history of primary sacral chondrosarcoma, an infection of the implanted spinal stabilization device and discuss the challenges that resulted from a colonic fistula associated with large, life threatening abscesses as late complications of radiotherapy.A 57-year-old man with a history of chondrosarcoma of the Os sacrum was treated 1985 by a R1 resection at another institution. To support stabilization, implantation of an Universal Spine System  was followed by osteosynthesis of L4/pelvis and additional spongiosaplasty with fibula chips. Post-operatively neutron irradiation was started due to intralesional surgical margins. Ten years later (1995), the patient developed a fulminant osteomyelitis ending up with soft tissue defect of 38 × 26 cm. Following radical debridement with removal of both iliac crests, the fibula chips and the USS system. After extended wound treatment a new USS system with fixation at the arcus root of L4, L5 and the lateral mass of the sacrum was implanted for stabilization of the sacrum. The large lumbal soft tissue defect with exposed vertebrae and hardware was covered with a free flap. For vascular supply the complete saphenous vein graft (76 cm long) was served as an arterio-venous loop and was anastomosed end-to-side to the superficial femoral artery as previously described  and a collateral infiltrate of the psoas muscle. Additionally extended osseous destructions of the sacrum were documented. To differentiate between postoperative defects, the previously diagnosed sequestrating chronic osteomyelitis or a possible relapse of his chondrosarcoma was hardly possible Fig . After a®, Fresenius Kabi AG, Bad Homburg, Germany). Additionally the sacral and praesternal abscesses were once more debrided in the operating room. The right ureter was adherent to the abscess formation and was mobilized. Postoperatively the patient was referred to an intensive care unit. The bilateral septic inflammations of the carpal joints were successfully treated with high dose antibiotics after surgical excision and drainage (Imipenem and Metronidazol). After multiple irrigations of the large wounds and decreasing inflammation, granulation tissue developed. In order to further minimize the dead space of the large wounds, microdeformational wound therapy by means of Vacuum Assisted Closure  was applied to the bilateral sacral wounds. Despite four weeks of intensive care his latissimus dorsi free flap was saved and the patient was discharged with wounds showing no signs of infection and a tendency towards good granulation ®, Fresenius Kabi AG, Bad Homburg, Germany)Polymeric biguanide-hydrochloride Vacuum Assisted Closure (V.A.C.-TherapyIn patients with sacral tumors enterocutaneous fistulas after free musculotaneous free flaps transfer are rare. They occur in the setting of surgical damage followed by radiotherapy or advanced disease and are associated with prolonged morbidity and high mortality. Identification of high-risk patients and management of fistulas at an early stage may delay the need for subsequent therapy and decrease morbidity .As in our case, ulceration of the gut and development of a fistula is based on changes in the collagen tissues and particularly in vascular tissue of the gut . The bowcoli bacteremia and multiple extra-intestinal manifestations.In summary, we have described a 57-yr-old sacral chondrosarcoma patient with a transsacral colon fistula complicated by E. 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.All authors hereby disclose any commercial associations which might pose or create a conflict of interest with information presented in this manuscript. All authors declare that they have no competing interests.LS documented and prepared most of the draft. MS documented and prepared most of the draft. SL Literature research, revision of bibliography. GM Edited the manuscript and helped in preparing the draft. TAS Documented and prepared part of the draft. HUS Edited the manuscript, revision of bibliography and helped in preparing the draft. All authors read and approved final manuscript.


Synchronous midgut carcinoids with gastrointestinal adenocarcinoma are a rare but recognised association.The patient, a 74 year old woman, underwent anterior resection for a low rectal adenocarcinoma. Intra-operatively 3 serosal deposits of tumour were noted in the distal ileum. Histology revealed these to be ileal carcinoids.During resection of a gastrointestinal tumour, a thorough inspection of the abdominal cavity should be undertaken to investigate the possibility of metastatic secondaries or a synchronous tumour as is reported in this case. The natural history of midgut carcinoid tumours is to progress slowly, arising from neuro-endocrine cells that line the tract. They often present with metastasis at diagnosis and occur most frequently in the ileum 52%) and the appendix (22%) . The inc% and theThe patient was referred to by her G.P. to surgical outpatients with a 9 month history of diarrhoea with tenesmus. Physical exam was normal, including a soft non-tender abdomen. Serum full blood count, urea & electrolytes, liver function tests and coagulation screen were all within normal limits. Elective colonoscopy revealed a large villous tumour in the lower rectum, which proved to be a moderately differentiated adenocarcinoma histologically. Staging abdomino-pelvic CT & MRI scans confirmed a 6.5 × 4 cm T3N1 irregular low rectal mass lesion extending inferiorly with involvement of the muscularis and serosa figure .Neo adjuvant chemo-radiotherapy was given to downstage the tumour and repeat MRI scan showed marked reduction of the mass lesion with no evidence of lymphadenopathy.At laparotomy a mobile tumour was noted in the upper rectum following mobilisation. In addition three serosal deposits of tumour were noted in the distal ileum. An anterior resection was performed and 25 cm of ileum was also resectedThe patient made an uncomplicated recovery and was discharged on 15 post operative day.Two nodules of carcinoid were confirmed in the small bowel segment using immuno-staining. They both invaded to serosal level. In the third, tumour was lost on deeper sectioning but it's H&E appearance supported carcinoid. Two tiny mesenteric nodes taken were clear. The 12 cm segment of rectum showed radiation change but no residual primary mucosal lesion, demonstrating complete pathological response with 12 negative lymph nodes.In 1888, Otto Lubarsch, a pathologist based in Berlin first described carcinoid lesions in detail. He reported the autopsy findings of a patient with multiple carcinoid tumours involving the ileum. Carcinoid syndrome and it's classic associated symptoms was reported two years later by Ransom. He described a patient with diarrhoea and wheezing secondary to an ileal carcinoid which had a distant metastasis to the liver [The annual age-adjusted incidence of small intestine carcinoids is reported as increasing, from 1.09 per 100,000 in 1973 to 5.25 per 100,000 in 2004 .Five year survival for resected isolated carcinoid is determined by site, with appendiceal carcinoids having a better 5 year survival prognosis (> 95%) than small intestine carcinoids (70–80%) . Recent Synchronous carcinoids with non-carcinoid neoplasms in the G.I. tract were first noted by Pearson and Fitzgerald in 1949 . A studyHypotheses put forward to explain this association include the secretion of active neuroendocrine peptides such as gastrin and cholecystokinin . Both haIn the case of a synchronous carcinoid with adenocarcinoma, management is directed towards the carcinoma, since the finding of carcinoid is incidental and so it is usually at an early stage. During resection of the colorectal tumour, a thorough inspection of the abdominal cavity should be undertaken to investigate the possibility of metastatic secondaries or a synchronous tumour as is reported in this case. Because of their slow growing natural history, the discovery of an asymptomatic gastrointestinal carcinoid during the operative treatment of another malignancy usually requires resection alone without additional treatment and will have little effect on the prognosis of the individual .Written consent was obtained from the patient for publication of this case reportThe authors declare that they have no competing interests.SM was involved in data acquisition and interpretation, writing initial drafts and subsequent revisions and undertook review of literature on topic. JOD made a substantial contribution regarding conception of report, was the editor of multiple drafts of case report, made many suggestions regarding format of report for inclusion of intellectual content and undertook review of literature on topic. PG made a substantial contribution regarding concept of case report, was the supervisor of work done and drafts of case report prepared by first and second authors. All authors read and approved final approval of version to be published given.


Renal cell carcinoma is the most common form of malignant renal tumour and is extremely lethal. About 25% of the patients develop metastasis at the time of diagnosis, and in many cases during the course of the disease, affecting the lung, lymphatic ganglions, liver, and bone, with skin metastases being quite rare.A 73-year-old patient, who had undergone surgery for adenocarcinoma in the left kidney 10 years previously, visited the dermatological service due to the appearance of recent, rapidly-developing lesion at the back of his neck. It was decided to remove it surgically. The histological study confirmed clear cell carcinoma that was probably of renal origin. A computed tomography scan was performed on the thorax and abdomen, and lesions were observed that were compatible with metastasis in the right kidney and left lung. Treatment with a multikinase angiogenesis inhibitor (sunitib) was started.Due to the late development of the skin metastases and those in other regions that worsen the prognosis, these patients must be subjected to long-term clinical observation. Urologist should pay attention to cutaneous lesion appearing in these patients as in many times they look like benign lesion. Renal Cell Carcinoma (RCC) currently accounts for 90% of all renal tumours, and is the most lethal of urological tumours. Its frequency has increased by 2.5% per annum. Thanks to the increase in image diagnosis  in recent years, these tumours are easier to diagnose at an early stage of the disease. However, it continues to be a highly lethal tumour in the event of the disease being widely disseminated at the time of diagnosis.Approximately 25% of patients diagnosed for RCC are shown to have metastasis at the time of the diagnosis, and these are more frequent in the following order: lungs, lymphatic ganglions, bone, liver, contralateral kidney, adrenal and ipsilateral glands, brain and other less frequent locations such as the skin ,2.A case study is presented consisting of a male patient aged 73 years who had undergone a left radical nephrectomy 10 years ago for grade 3 Fuhrman’s renal adenocarcinoma. Skin lesion had developed 2 months ago on the patient’s neck, and a histological diagnosis confirmed the presence of clear cells, showing positive for EMA, CEA and CD10 [Caucasian and Spanish patient aged 73 years with minor renal failure on whom a left radical nefrectomy for renal adenocarcinoma had been performed 10 years ago in the Urology Unit. The patient was referred to the Dermatological Unit for an asymptomatic lesion at the back of his neck that had developed 2 months previously. The examination revealed the presence of a tumour with a firm consistency, bluish-red in colour, with a somewhat hyperkeratous surface and maximum diameter of 2.5 cm, which was initially diagnosed as a possible angioma. Surgical treatment was indicated due to the late development of the lesion and its rapid growth. The histopathological study revealed the presence of groups of irregular cells with cytological irregularities (large nuclei and prominent nucleoles) and clear cytoplasm . The IHCIn view of the histological results, it was decided to request a CT scan of the abdomen and pelvis which showed an heterogeneous image with a maximum diameter of 3.7 cm in the middle of the right kidney , suggestDue to the presence of multiple metastases, it was decided to refer the patient to the Cancer Unit for evaluation and the administration of medication with angiogenesis/ multikinase inhibitors.Owing to the clinical and evolutive particularities of the RCC, in certain cases diagnosis of this condition is made at a late stage of the disease when the cancer is widespread and has metastasised in different areas of the body. The development of skin metastases accounting for between 1 and 3% of all metastases. The most usual location of skin metastases in these patients is the scalp and face, and they are usually single lesions that grow rapidly, are bluish-red in colour and sometimes pulsating , differeIn most of the cases published regarding patients with RCC, the development of skin metastases takes place within six months to five years of the initial diagnosis and after performing the nefrectomy ,2,6, excTreatment of metastatic renal adenocarcinoma consists of a combination of surgical treatment  and angiogenesis/multikinase inhibitors (sunitinib or sorafenib). However, treatment of single skin lesions is usually surgical , except In short, we wish to emphasise the peculiar natural history of renal adenocarcinoma, in addition to the need for exhaustive monitoring of these patients even up to ten years after the initial diagnosis, due to the development of late metastases that worsen the clinical prognosis and reduce life expectancy, showing an average survival rate after diagnosing metastasis, of between 7 and 32 months .


The reported case illustrates an instance of colonic adenocarcinoma presenting as an isolated tumour 3 1/2 years after open surgery. The presentation was in some respects unique as it was complicated by an incisional hernia and occurred in the anterior abdominal wall. A literature review was performed.An 83 year old lady initially underwent an extended right open hemicolectomy for a mid-transverse colonic adenocarcinoma (T4N2M0). No adjacent structures were involved. After adjuvant chemotherapy, she was kept under regular surveillance. A CT scan and colonoscopy at one year were normal. At 18 months investigations including an ultrasound scan of the liver and a radioisotope bone scan were all negative. Over three and half years later the patient presented with an incisional hernia. Repeat CT scan and tumour markers were reported as negative. At operation, a mass was found within the anterior abdominal wall complicating the incisional hernia. This mass was widely resected and a laparotomy performed. Histology confirmed an adenocarcinoma of colonic origin extending to one of the lateral margins. A post-operative PET scan confirmed the absence of intra-abdominal pathology.The literature regarding recurrence of colonic tumours after open surgery reports low incidences of this occurring within abdominal incisions. The literature indicates prognosis is poor, but the numbers are small and distinction is often not made between isolated recurrence and those with other sites of tumour recurrence. In order to avoid missing isolated wound implantation, careful consideration should be given to those who present with new pathology related to previous cancer surgery incisions, both clinically and radiologically. The prognosis associated with colorectal cancer has significantly improved due to advances in early diagnosis and therapeutic techniques. The post-operative follow-up of such patients remain an integral part of management due to the potential for recurrent disease. The prevalence of loco-regional recurrence or metastatic disease, especially to the liver and lung, is well recognised and hence forms the main focus of follow-up imaging investigation.The question of wound recurrences after laparotomy has been infrequently addressed in the literature ,2, in coCT imaging is an effective modality in diagnosing recurrences; however it may be limited in cases where isolated wound recurrences following open surgery co-exist with other benign pathologies. The case report relates to a patient presenting with an anterior abdominal wall hernia 3 1/2 years after open surgery, who was found to have an incidental anterior abdominal wound tumour at operation, despite a pre-operative CT scan reported as normal.An 83 year old lady initially underwent via a midline vertical incision, an extended right hemicolectomy in 2003. She had presented with weight loss with no previous medical or surgical history. Functionally she was independent and self-caring. Pre-operative radiology (including a staging CT scan) showed a mid-transverse colonic lesion. Colonoscopy revealed no other intra-colonic lesions and tumour markers were normal.At operation, there was no invasion into other structures or the anterior abdominal wall. Histology demonstrated a T4 N2 Mx adenocarcinoma in the transverse colon. The serosa had been breached but the tumour had been completely excised. The apical node was clear but 4 out of 11 nodes were involved. The case was discussed pre- and post-operatively in the Gastro-intestinal (GI) multi-disciplinary meeting (MDM) and staged as T4 N2 M0 (Dukes C1). Adjuvant chemotherapy was offered to the patient, who subsequently underwent a weekly course of bolus 5FU & Folinic acid. This was well tolerated with only grade I nausea and mild hair loss and was completed at six months post-operation.The patient was seen regularly in clinic on a three monthly basis. At one year, the surveillance CT scan  was unremarkable as was colonoscopy. At 18 months, the patient complained of lower back pain in April 2005. In view of her history a chest X-ray, tumour markers and ultrasound scan of the liver were ordered. These were all negative. A radioisotope bone scan was performed. The scan showed only lumbro-sacral arthritis and her pain resolved with simple analgesia.®  was orderedAt three and a half years post-surgery, she reported some mild abdominal discomfort and distension. She attributed this to her incisional hernias, at the site of the midline scar. These had progressively worsened in size as had her symptoms. On examination, she was found to have two incisional hernias which lay 20 mm above and 20 mm below her umbilicus and were 30 mm and 40 mm respectively in diameter. A contrast enhanced staging CT of the chest, abdomen and pelvis was performed. A midline ventral hernia was noted on transverse slices of the CT image but no focal lesion was reported. The anastomotic site appeared normal with no recurrent growth or lymphadenopathy otherwise seen. Tumour markers were not elevated . An incisional hernia repair was subsequently arranged and a specialised mesh was ordered. The provisional plan was to place the mesh behind the anterior abdominal wall (anterior to the peritoneum). As there were two large defects which were closely related, a 20 cm × 15 cm Bard Composix-MeshAt operation in 2007, a further midline incision was performed. Following division of skin and subcutaneous tissue the anterior abdominal wall was visualised. The two incisional hernia sacs were each identified and freed from their attachments to the anterior abdominal wall allowing pre-peritoneal access. At this point it became apparent, that the tissue in between the two incisional hernias was not dense scar tissue. On palpation a hard mass measuring 20 mm × 20 mm in diameter was found situated within the anterior abdominal wall. This was not attached to peritoneum. Thus it appeared as if it may be an isolated recurrence Figure . The mas®. This was attached with 3/0 Prolene to parietal peritoneum using continuous sutures as a modified sub-lay technique. The rectus sheath was approximated but not apposed with 1/0 nylon to allow a tension free repair. A vacuum drain was placed superficial to the anterior rectus sheath. Closure was with interrupted subcutaneous 3/0 Vicryl sutures and clips to skin. The post-operative course was uncomplicated.As defect following the wide excision was closed using the Bard Composix-MeshThe mass which measured 40 mm × 40 mm × 30 mm. Histologically, it consisted of fibro-connective tissue infiltrated by a moderately differentiated adenocarcinoma. The tumour cells were seen to involve one of the lateral surgical margins. There was no superior or inferior extension of the tumour. Subsequent immuno-histochemistry was positive for CK20 and CDX2 and negative for CK7  was not appreciated by the consultant radiologist when reporting on the CT scan. The complexity of the incisional hernia with its components lying above and below the tumour also contributed to the difficulty in picking up the lesion Figure . This waIn the case report, there was no clinical evidence of tumour within the wound pre-operatively. A combined PET/CT scan was found by Goshen et al  to be exGiven the involvement of the surgical margins, the options available were either radical re-excision or radiotherapy. Hughes et al  describeThe case reported illustrates an instance of colonic adenocarcinoma recurring as an isolated tumour after open surgery. Its presentation was unique as it was complicated by an incisional hernia and presented in the anterior abdominal wall. Tumour markers were negative and there was no intra-abdominal pathology. Wound implantation in an incisional scar after open surgery is rare, particularly when it is isolated and presentation is more than two years after the original surgery.The literature indicates prognosis is poor, but the numbers are small and distinction is often not made between isolated incisional wound implantation and those with other sites of tumour recurrence or co-existent intra-abdominal malignancy. Further studies on this would shape current practice.There were a number of factors which arose in this case including the CT scan report, which may have been altered by a higher index of suspicion. In order to avoid missing isolated wound implantation, careful consideration should be given to those who present with new pathology related to previous cancer surgery incisions, both clinically and radiologically.CEA: Carcinoembryonic Antigen; CT: Computerized Tomography; GI: Gastro-intestinal; MDM: Multi-Disciplinary Meeting; PET: Positron Emission Tomography.The author(s) declare that they have no competing interests.Each author performed an independent literature search. AC, and LL operated upon the patient initially, critically appraised the literature and conceived the case report; HJ reviewed the literature and revised the final manuscript; FH reviewed the literature and helped in drafting the manuscript. All authors read and approved the final manuscript.


Pseudo-Meigs' syndrome is a rare pathological entity characterized by the presence of a pelvic mass other than an ovarian fibroma. The mass is associated with ascites with or without hydrothorax.We describe the case of a 41-year-old Caucasian woman with a large uterine leiomyoma associated with massive ascites. A magnetic resonance imaging scan showed a large subserosal leiomyoma with multiple areas of cystic degeneration.To the best of our knowledge, this is the first reported case of pseudo-Meigs' syndrome caused by a uterine leiomyoma and diagnosed using magnetic resonance imaging. The pathophysiology of this syndrome and the role of magnetic resonance imaging are emphasized in this case report. Pseudo-Meigs' syndrome is a rare pathological entity characterized by the presence of a pelvic mass other than an ovarian fibroma. The mass is associated with ascites with or without hydrothorax.We report the case of a patient with pseudo-Meigs' syndrome caused by a uterine leiomyoma. A magnetic resonance imaging (MRI) scan provided a detailed description of the tumor. Our patient's MRI results correlated well with histological findings and helped us make a diagnosis, which enabled the avoidance of a hysterectomy. To the best of our knowledge, currently fewer than 35 cases of pseudo-Meigs' syndrome have been reported in the literature and our case of pseudo-Meigs' syndrome is the first to be diagnosed using MRI.A 41-year-old Caucasian woman presented at our hospital with a 12-month history of abdominal swelling, discomfort, urinary frequency and incontinence. She had regular menstrual cycles and had never been pregnant. Her clinical examination revealed a marked distension of her abdomen and a large palpable mass in her central pelvis. Gynecological vaginal ultrasound (US) showed abundant ascites in her pelvis, as well as a solid, smoothly outlined mass with heterogenous echogenicity. The mass seemed to extend from her pelvic cavity to her abdomen on the midline above the uterus. Her uterus and ovaries could not be identified separately from the pelvic mass. The mass was considered to be of adnexal origin. Laboratory test results of our patient showed the following values: serum carbohydrate antigen (CA)-125 level at 436.7 U/mL , fetoprotein (FP) at 2.8 ng/mL,  beta-human chorionic gonadotropin (β-HCG) at 5.0 mIU/mL , and carcinoembryonic antigen (CEA) 1.07 ng/mL . An MRI scan was requested to further evaluate our patient and to determine the exact nature of her mass.The results of our patient's MRI scan revealed massive ascites and a heterogeneous ovoid pelvic mass measuring 13 × 16 cm. The mass had a broad connection to the uterus. It was located subserosally and extended superiorly from the posterior body and fundus of her uterus. The uterus was displaced inferiorly. She was noted to have a normal endometrial stripe, a normal junctional zone, and normal ovaries. The mass was heterogeneous and produced a predominantly low to intermediate signal intensity on T2-weighted images relative to that of the outer myometrium. Several small foci with very high signal intensities were also seen (Figure Our patient underwent an exploratory laparotomy. A large firm mass, which originated from the uterine fundus, was seen. Multiple lobulated projections were also seen on the superior border of the mass Figure . The masPseudo-Meigs' syndrome is characterized by the presence of ascites. It is also often characterized by pleural effusion caused by a pelvic tumor other than an ovarian fibroma. Tumors associated with pseudo-Meigs' syndrome are usually found in women's genitalia. The most commonly described tumor type is a leiomyoma, which is usually found in the uterus or the broad ligament . Other rUterine leiomyomas are the most commonly reported cause of pseudo-Meigs' syndrome. They usually manifest as increased abdominal distension caused by a progressively enlarging pelvic mass and ascites. Respiratory insufficiency caused by pleural effusion is also often encountered .It is speculated that the presence of ascites results from mechanical irritation of the peritoneum and the leakage of intratumoral fluid from the degenerated leiomyoma. As leiomyomas enlarge, they may outgrow their blood supply, thus resulting in various types of degeneration.Cystic degeneration is considered to be a sequela of edema and is observed in about 4% of reported cases of leiomyomas. Cystic spaces appear as round, well-demarcated areas with signal intensities that have the characteristics of fluid, namely low on T1-weighted images and high on T2-weighted images with no enhancement [Pleural effusions, which are commonly right-sided, result from transdiaphragmatic transport of ascitic fluid . AlthougLaboratory tests usually reveal an elevated serum CA-125 level caused by peritoneal irritation . Our patAn MRI scan enables the detection and characterization of leiomyomas, as well as their differentiation from other types of adnexal masses. If MRI can demonstrate the continuity of an adnexal mass to adjacent myometrium then a diagnosis of leiomyoma can be established. The ability of MRI to visualize normal ovaries, even in the presence of an enlarged, myomatous uterus, may aid in determining the origin of the pelvic masses by excluding a diagnosis of ovarian neoplasm .Ovarian fibromas and Brenner tumors are benign ovarian neoplasms that have a large fibrous component. These neoplasms can have signal intensities similar to those of pedunculated leiomyomas. MRI can show fibromas and Brenner tumors surrounded by ovarian stroma and follicles, thus establishing the ovarian origin of the mass and excluding a diagnosis of leiomyoma .MRI has been shown to be more sensitive than US in detecting leiomyomas. An accurate assessment of an enlarged, myomatous uterus is not consistently possible with US because of its limited field of view.Resection of the tumor leads to the resolution of the ascites and pleural effusion, therefore a thorough knowledge of the pseudo-Meigs' syndrome is important. Although the concomitant existence of a pelvic mass, ascites and pleural effusion is highly indicative of malignancy, hysterectomy and bilateral salpingo-oophorectomy can be avoided.Pseudo-Meigs' syndrome is a well-recognized pathological entity characterized by the coexistence of a pelvic mass, other than an ovarian fibroma, with ascites and hydrothorax. MRI is highly sensitive in detecting the relationship of a tumor with its adjacent structures and in providing sufficient correlation with histological findings. MRI thus renders a correct diagnosis possible, so that aggressive surgical treatment can be avoided.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.DC and EP analyzed and interpreted our patient data and were major contributors in writing the manuscript. AD analyzed our patient data and contributed in writing the manuscript. All authors read and approved the final manuscript.


The association of pseudo-Meigs' syndrome, elevation of CA 125 to the struma ovarii is a rare condition. So far only nine cases have been reported in English literature through MEDLINE search. Here we report a 46-year-old case of the struma ovarii, presented with ascites, hydrothorax, right ovarian mass and elevated serum CA 125 level. These findings were misdiagnosed for an ovarian malignancy at the first impression. Immediate resolution of the ascites, hydrothorax and normalization of the serum CA 125 level were followed by ovarian mass removal. Struma ovarii could be a rare cause of ascites, hydrothorax, ovarian mass and elevated CA 125. This rare condition should be considered in the differential diagnosis in patents with ascites and pleural effusions but with negative cytology. Struma ovarii is a rare ovarian neoplasm derived from germ cells in a mature teratoma. This tumor is generally benign, although malignant transformation has been reported . The preThe present case is a 46-year-old, female, gravida 1, para 1, who was admitted to a local hospital, complaining of fatigue, anorexia, and abdominal swelling. Her medical history included nothing special. Physical examination revealed a palpable mass in the lower abdomen. A thoracoabdominal CT scan showed marked pleural effusion and a heterogeneous mass, large ascites with many nodosity images in the pelvic wall and considered as malignant tumor of ovary.She was then transferred to our hospital for further treatment in September, 2009. The patient's serum CA 125 level was 1230.9 U/mL, while CEA (2.6 ng/ml), AFP (14.2 ng/ml), CA 199 (14.8 U/ml), and CA 153 (7.8 U/ml) levels were within the normal range. Abdominal ultrasonography showed a heterogeneous, multiloculated mass, with a moderate amount of ascites, and subsequent transvaginal ultrasonography revealed a large complex pelvic mass, 16 cm largest dimension, of probable adnexal origin with low blood resistance flow within the tumor. The uterus was normal in size. Abdominal paracentesis yielded 2 liters of yellow serous fluid consistent with an exudative process. Microscopy and cytology revealed only reactive mesothelial cells without malignant cells.The patient was arranged for an exploratory laparotomy. Six liters of straw-colored ascites was evacuated. The uterus was in normal size and the left ovary measured 3 × 2 × 2 cm with a normal appearance. A 20 × 18 × 15 cm complex, multicystic mass, without evidence of external excrescences, had replaced the right ovary. There was no evidence of intraperitoneal  spread of disease or retroperitoneal adenopathy. And right salpingo-oophorectomy was performed. A frozen section of the right ovarian mass was interpreted as struma ovarii. As strongly insisted by the patient and her family member, a subsequent hysterectomy and left salpingo-oophorectomy were performed according to the informed consent.Post operative thyroid function test including serum levels of TT3 (1.78 nmo1/L), TT4 (82.5 nmo1/L), FT3 (8.2 pmol/L), FT4 (30.5 pmol/L) and TSH (2.3 mU/ml) were performed on day two, which were within normal limits. The level of CA 125 was decreased to 817 U/mL. The final pathology revealed right struma ovarii with benign thyroid tissue confined to the ovary  and was first described by Von Klden in 1895 and Gottschalk in 1899 [Despite containing thyroid tissue, only 5% of struma ovarii have features of hyperthyroidism . AscitesIn the literature, very few reports have been published on struma ovarii associated to ascites and elevated CA125 . In bothThe elevation of CA 125 may have been secondary to the presence of ascites; however, its level was much higher than that typically seen with ascites of benign origin. An ovarian mass with ascites and elevated serum CA 125 level in a woman generally suggest a malignancy process. So the present case with the clinic findings of ascites, hydrothorax, markedly elevated serum CA 125 and a large complex pelvic mass in a woman strongly suggest pelvic malignancy before operation. But complete remission of the ascites, hydrothorax, and CA125 was obtained after surgery without any adjuvant therapy.This report emphasizes that there are benign gynecological conditions might show clinical, ultrasonographic and biochemical signs suggestive of malignancy. They rarely should be considered as the benign diseases in the differential diagnosis when the patients presented with ascites, elevated serum CA 125 and pleural effusions, but with negative cytologic examination.CT: computed tomography; TSH: thyroid stimulating hormone; CEA: carcinoembryonic antigen; AFP: alpha-fetoprotein; T3: triiodothyronine; T4: thyroxine; TT3: total T3; TT4: total T4; FT3: free T3; FT4: free T4.The authors declare that they have no competing interests.WJ drafted the manuscript. XL, ZJZ, CJX, XSL are involved in design, acquisition, interpretation and manuscript preparation. All authors had read and approved the final manuscript.WJ, XL, ZLZ, CJX, XSL: Department of Gynecology, Obstetrics and Gynecology Hospital, Fudan University, Shanghai, P. R. China.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.


Cystic lymphangiomas are uncommon tumors that can arise from any part of the body. They can pose a diagnostic and therapeutic challenge. They are more common in infants and children than adults. Broad ligament cystic lymphangioma is extremely rare.A 70-year-old multiparous woman presented with an abdominal mass of 20-year duration. A large cystic swelling was detected on computed tomography scan that was found to arise from the left adnexal region. This 19 kg lesion was found arising from the broad ligament. It was successfully removed. A detailed pathological study, including immunohistochemistry, was required to diagnose the lesion as a cystic lymphangioma.Lymphangiomas should be treated with total surgical excision. Broad ligament lymphangiomas are extremely rare but must be considered as a differential diagnosis of cystic lesions in that region. Cystic lymphangiomas are common in infants and children, but adult cystic lymphangiomas are rare. Although they can occur at any site in the body, cystic lymphangiomas of the broad ligament are extremely rare .A 70-year-old multiparous woman presented with a huge abdominal swelling of 20-year duration. The patient had been unable to walk for 3 months. A computed tomography (CT) scan revealed a huge cystic swelling in the abdomen, possibly arising from the left adnexal region Figure . After pPathological gross findings were those of a very large multiseptate cystic lesion covered with serosa. Microscopy revealed that the cyst wall had bundles of smooth muscle with connective tissue. In addition, the cyst wall was lined internally with epithelium Figure . This tuLymphangiomas are benign tumors of the lymphatic system. They are classified as cavernous, lymphangioma simplex, or cystic lymphangioma . Cystic Lymphangiomas in children are considered to arise from sequestered lymphatic sacs that fail to communicate with the draining lymphatic channels. This is a widely accepted theory. However, the etiology in the adult population is controversial. Some authors believe that the adult manifestations are a result of delayed proliferation of congenital or acquired lymphoid nests after stimuli such as respiratory infection or local trauma . Others Radiographic evaluation with magnetic resonance imaging or CT is invaluable for the diagnosis and determination of the extent of the lesion. In addition, it is essential in defining normal anatomical structures that need to be preserved when surgical excision is performed . AccuratLymphangiomas are treated by surgical excision. Complete excision of the mass with negative surgical margins is the optimal treatment, and the results are excellent . Intra-aEffective immunohistochemical markers specific for lymphatic endothelial cells have been reported including lymphatic vessel endothelial receptor 1, vascular endothelial growth factor receptor 3, and Prox-1. However, the antibodies against these markers are available only for frozen section specimens. More recently, a new monoclonal antibody, D2-40, has become available; this is a specific marker of lymphatic endothelium, since it does not stain vascular endothelium . In thisWe could only find one case of broad ligament cystic lymphangioma reported in the literature . The casAdult cystic lymphangiomas of the broad ligament are very rare benign tumors. Total surgical removal is the treatment of choice. This is a report of one such case diagnosed with the help of the lymphatic marker D2-40, treated successfully with surgery, and recurrence-free 2 years later. Although rare, cystic lymphangiomas must be considered in the differential diagnosis of cystic lesions in the abdomen and pelvis.CT: computed tomography.The authors declare that they have no competing interests.KH contributed to the conception, design, gathering data, and revision of the manuscript draft. SRK contributed to obtaining the data and drafting the manuscript. CSM contributed to obtaining the data, the pathological review, and drafted part 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.


Actinomyces are slow growing, non-spore forming, gram-positive, branching bacilli that thrive in anaerobic and microareophilic conditions. Actinomyces are more commonly associated with oral and cervicofacial infections. Hepatic involvement in infections of the abdomen (known as isolated hepatic actinomycosis) is rare, accounting for only 5% of all cases of actinomycosis.We present the case of a 75-year-old Caucasian woman with a 3-month history of night sweats, fever, chills, abdominal bloating, anorexia, weight-loss, and early satiety. The patient was found to have isolated hepatic actinomycosis infection after undergoing a laparotomy with a biopsy of the liver. The patient has now recovered.Isolated hepatic actinomycosis is a rare and often overlooked etiology for a liver mass. Given its subacute presentation and nondescript symptomatology, physicians should be aware of this differential and the potential pitfalls in diagnosis and management. Actinomyces are slow growing, non-spore forming, gram-positive, branching bacilli that thrive in anaerobic and microareophilic conditions. It is most commonly associated with infection of the oral and cervicofacial region. Abdominal actinomycosis is rare and its pathogenesis is presumed to be hematogenous spread through the portal vein from a mucosal injury or other abdominal focus of infection. Most frequently affected is the ileocecal region. Hepatic involvement has been reported in 15% of those with abdominal infections, and represents 5% of all cases of actinomycosis [In this case report, we report a case of isolated hepatic actinomycosis (IHA) occurring in a 75-year-old female with no predisposing factors. Along with a review of the literature, we will define the risk factors, clinical characteristics, diagnostic methods, and treatment of this infection.A 75-year-old Caucasian woman of northern European descent presented with a 3-month history of night sweats, fevers (101-102°F nightly), chills, abdominal bloating, anorexia with 60 lb weight loss and early satiety. Her past medical history included hypertension, hypothyroidism, gastroesophageal reflux disease, diverticulitis, polymyalgia rheumatica, and a laparoscopic cholecystectomy in 1997. Current medications included HydroDIURIL, Toprol XL, Diovan, Synthroid, Prilosec, Plaquinil, and calcium supplementation. She denied any abdominal pain, nausea, vomiting, diarrhea or constipation and was able to tolerate per os diet despite her early satiety.3/mcL (4.0-10.0) and an absolute neutrophil count of 10.9 103/mcL (1.7-1.6). Hemoglobin level, hematocrit, platelet count and mean corpuscular volume were 8.2 gm/dL (12.0-16.0), 23.9% (36-48), 341 103/mcL (140-450) and 85 FL (82-100) respectively. The following liver chemistries were normal; serum glutamic oxaloacetic transaminase 17 IU/L (15-41), serum glutamic pyruvic transaminase 15 IU/L (10-15), total bilirubin 0.6 mg/dL (0.0-2.0) and direct bilirubin 0.2 mg/dL (0.0-0.8). Prothrombin time and international normalized ratio were 14.9 sec (8.0-12.5) and 1.30 respectively. There was an increase in alkaline phosphotase to 270 IU/L (27-120), tumor marker CA19-9 was 49.6 U/mL (0-35) and alpha fetoprotein was elevated to a level of 7.3 IU/mL (< 5.5). Albumin was low at a level of 1.8 gm/dL (3.2-4.8) as was serum protein 5.0 gm/dL (6.1-7.6). C-reactive protein, a marker of inflammation, was elevated to a level of 10.8 mg/dL (< 0.5).Physical examination revealed an elderly female who did not appear to be in any acute distress. Vital signs: temperature 100.7°F, pulse 54/bpm, respirations 20/min, blood pressure 116/56 mm/Hg, oxygen saturation 98% on room air. Abdominal exam revealed no palpable hepatosplenomegaly, guarding or rebound with normoactive bowel sounds. The rest of her exam was unremarkable. Laboratory studies demonstrated a leukocytosis of 12.8 10The patient underwent imaging with an ultrasound of the liver and a CT of the abdomen. Both demonstrated a lesion in the posterior right lobe of the liver near the dome, measuring 6.9 × 7.4 cm, concerning for abscess and malignancy Figure . This arActinomyces israelii, giving rise to chronic suppurative infections [Actinomyces is a rare cause of intra-abdominal infections. The etiology in the majority of cases involving the liver, are thought to arise directly or hematogenically from an intra-abdominal focus. In rare cases that involve the liver, when a primary focus is not found, the disease process is called primary or isolated hepatic actinomycosis (IHA). There are 13 different species of actinomycosis; 6 of which are associated with human disease . By far the most common human pathogen encountered is fections ,3.Risk factors include previous abdominal/pelvic surgery, abdominal wall trauma, gastrointestinal foreign body, gastrointestinal tract lesions and immunosupression . AlthougDue to the cryptogenic nature of the disease, it takes several weeks to months of symptoms to make the diagnosis with a range of 4 days to 18 months until presentation . The mosBecause imaging studies frequently reveal single or multiple lesions, actinomycosis is often misdiagnosed as a primary or metastatic tumor. The most frequent radiographical finding is a single hypodense mass/abscess 68.4%) . CT ofte% . CT ofDefinitive diagnosis is based on histochemical, macroscopic, and microscopic examination of surgical tissue specimens, which reveal yellow sulfur granules and basophilic filament aggregates respectively . CultureBoth surgical excision and antimicrobial treatment are therapeutically effective. The medical treatment regimen for IHA is the administration of penicillin, tetracycline, or clindamycin . DuratioIn conclusion, IHA is a rare and often overlooked etiology for a liver mass. Given its nonspecific presentation and nondescript symptomatology, physicians should be aware of this differential and the potential pitfalls in diagnosis and management.IHA: Isolated hepatic actinomycosis; IUD: intrauterine contraceptive devices; VATS: video-assisted thoracic surgery.The authors declare that they have no competing interests.All authors contributed in the final approval of the article and to each of the following jobs: drafting of the article, critical revision of the article for important intellectual content, conception and design, analysis and interpretation of the data.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.


Background. Intraparenchymal endometriosis of liver is rare. It may present as liver tumour and the diagnosis is not usually established till after surgery. Case Outline. A 48-year-old postmenopausal woman presented with right upper quadrant pain and a cystic liver mass. Liver function tests and tumour markers  were normal. Radiological imaging  suggested a thick walled cystic mass involving segments IV and VIII with complex intracystic septations. Frozen section at operation suggested a benign cystadenoma. The cyst was enucleated using a CUSA (Cavitron ultrasonic aspirator). The final histology confirmed endometriosis. Discussion. Eleven cases of hepatic endometrioma have been reported and only four in postmenopausal women. Preoperative diagnosis poses a challenge and so far none of the cases have been diagnosed preoperatively. Surgery remains the treatment of choice. Accurate diagnosis at time of operation may avoid extensive liver surgery and its associated morbidity. Hepatic endometriosis is a rare disorder characterized by the presence of ectopic endometrium in the liver. An extensive review of the published literature in English Language showed that only eleven cases have been previously reported. Of the cases reported four have been described in postmenopausal women and six had a previous history of endometriosis. We report a case of hepatic endometriosis in a postmenopausal woman who presented with right upper quadrant pain as her only symptom. Possible methods of pathogenesis are discussed and literature is reviewed. The features of this case and the evidence in literature suggest that hepatic endometrioma should be included in the differential diagnosis for a woman of any age presenting with a hepatic mass, with or without a history of pelvic endometriosis. However, preoperative diagnosis is difficult despite exhaustive investigations in the absence of characteristic clinical and radiological features.ge body habitus precluded any accurate assessment for a mass. The tumour markers  were within normal range as were the liver function tests. An abdominal ultrasound scan had previously shown a 9 × 11 cm cystic mass in segment IV of the liver. The wall appeared thick with complex septae, and there was apparent anterior extension into the extraperitoneal fat. Doppler studies indicated the mass to be completely avascular. Magnetic resonance imaging (MRI) showed an 11 × 13 cm cystic mass in segments IV and VIII, bulging into segments II and III and abutting the left and middle hepatic veins  and had suffered no other gynaecological symptoms since. Her past history included hypertension and mild asthma. Examination revealed slight right upper quadrant tenderness but the patient's lar Figures . The masA laparotomy was performed with the intention of a left trisectionectomy . At operation the initial finding was that of a large cystic tumour in segment IV densely adherent to and infiltrating the abdominal wall and diaphragm. There was no evidence of metastases. The cyst was opened because it seemed unresectable and the interior was found to be thickly trabeculated with a ragged surface exudate. Biopsies were taken from the cyst wall and the septae. Frozen section histology suggested a benign cystadenoma. A nonanatomical resection was performed using a Cavitron Ultrasonic Aspirator (CUSA) and included segment IV and parts of segments II, III, and VIII. The tumour had to be peeled off from the left and middle hepatic veins. Some tumour was left on the diaphragm and pericardium as excision was considered unsafe. The patient made an uneventful recovery and was discharged home ten days postoperation. At followup she is asymptomatic 7 years after surgery with no evidence of recurrent disease.Histological analysis showed an outer wall of fibrofatty tissue abutting large portal tracts with adjacent hepatic parenchyma showing no intrinsic abnormality. The inner wall was lined by fragments of endometrial tissue . DiaphraEndometriosis, first described by Rokitansky in 1860 and is defined as the presence of endometrial tissue at sites other than the lining of the uterus , 3. PartThe pathogenesis of endometriosis is uncertain. The most widely held theories are based upon possible mechanisms which transport endometrial tissue to distant sites in a fashion analogous to metastatic spread of neoplasm , 13. TheA different theory, that of coelomic metaplasia, suggests that peritoneal and submesothelial connective tissue have the potential to change to endometrial tissue . This haRegardless of the pathogenesis, which remains controversial, this lady had a benign hepatic lesion which was not considered in the differential diagnosis. Indeed according to her ultrasound and MRI scans, the working diagnosis was that of a potentially malignant cystic neoplasm. In retrospect it appears that the patient did have a significant gynaecological history (pelvic endometriosis) for which she had undergone a hysterectomy and bilateral salpingoopherectomy. Of the previously reported cases a previous history of endometriosis has been reported , 8–11. Tgery [The ultrasound findings of a thick walled multiseptate cyst around the falciform ligament and high signals on TI and T2 sequence on MRI (suggesting presence of blood) should have raised the suspicion. But these findings are not specific. Inal et al. looked at the previous seven reported cases and concluded that there were no magnetic resonance, computer tomography, or ultrasound characteristics exclusively specific to hepatic endometrioma . This isgery . HoweverHepatic endometrioma is a rare condition and its aetiology and pathogenesis are unclear. This is the twelfth reported case and it seems that no clues to its diagnosis can be gleaned from the history or examination and the imaging studies are not specific to this condition. For these reasons, hepatic endometrioma should be included in the differential diagnosis for a woman of any age presenting with a hepatic mass, with or without a history of pelvic endometriosis. Surgery remains the mainstay of treatment in patients with extensive or aggressive disease. However, in the case of young women fertility issues should be taken into consideration when considering treatment options. Frozen section histology at the time of surgery may help to avoid radical liver resection and its associated morbidity and mortality.


A hemangiopericytoma is a rare, soft-tissue tumor of vascular origin derived from a pericyte of Zimmerman, which is a modified smooth muscle cell that surrounds the small blood vessels. Hemangiopericytomas can occur wherever there are vascular capillaries. However, there are no previous reports of a hemangiopericytoma in the sacrococcygeal space.We describe the first reported case of a hemangiopericytoma found in the sacrococcygeal space. A 47-year-old Japanese woman presented with a palpable tumor on the left side of her anus. Preoperative imaging indicated that the tumor was in the sacrococcygeal space without invasion of other organs. A complete resection was performed via a parasacral incision. The histological and immunohistochemical staining patterns supported the diagnosis of a hemangiopericytoma.A complete resection without piecemeal excision is the best way to treat a hemangiopericytoma. Recognizing the presence of a hemangiopericytoma in the sacrococcygeal space requires appropriate surgery. A hemangiopericytoma is a rare, soft-tissue tumor of vascular origin derived from a pericyte of Zimmerman, which is a modified smooth muscle cell that surrounds the small blood vessels. This type of tumor was first described by Stout and Murray in 1942 ,4, it isA 47-year-old Japanese woman presented with a palpable tumor on the left side of her anus. The tumor was elastic and hard and had a smooth surface. She had no pain or tenderness associated with the lesion, and no other clinical symptoms. Computed tomography (CT) scan showed a mass in the sacrococcygeal space with a smooth surface and no invasion to the rectum Figure . MagnetiThe pre-operative images indicated that the tumor was not derived from the rectum. Because of its vascularity, the pre-operative diagnosis was a soft-tissue tumor such as a solitary fibrous tumor, fibrous histiocytoma, synovial sarcoma, mesenchymal chondrosarcoma, or hemangiosarcoma. A biopsy was avoided due to the risk of needle track seeding. The patient underwent a tumorectomy via parasacral incision, without a rectectomy, because pre-operative examinations including a CT and EUS revealed that the rectum was intact. The tumor was completely removed Figure . The excWe performed an immunohistological analysis to obtain a diagnosis of the type of mesenchymal tumor. The mesenchymal tumor cells in our patient stained positive for CD34 Figure  and vimeLittle has been published about hemangiopericytoma, a rare, soft-tissue tumor. It can occur anywhere vascular capillaries are found. The tumors most commonly occurs in the musculature of the extremities, retroperitoneum, pelvis , head, neck and lungs ,4.The pathological diagnosis of a hemangiopericytoma, in comparison to other mesenchymal tumors such as solitary fibrous tumors, can be difficult . Bcl-2 aet al. reported that hemangiopericytomas showed a poor response to chemotherapy. They treated six patients with pre-operative chemotherapy, and only one of them responded to anthracycline-based chemotherapy [Radiotherapy and chemotherapy are not generally effective for the treatment of a hemangiopericytoma . Some haotherapy . Therefoet al. also reported that 5-year and 10-year survival rates of patients with a hemangiopericytoma were 71% and 54%, respectively. In addition, they noted that the survival rate differed between a curative and a non-curative resection. The 5-year survival rate in patients treated with curative resection and non-curative resection was 79% and 50%, respectively [Spitz ectively . These dThis report presented a rare case of a hemangiopericytoma in the sacrococcygeal space. Many diseases are associated with anal lesions, therefore a thorough differential diagnosis and complete resection without piecemeal excision must always be performed in the management of this type of malignant tumor.CT: computed tomography; FDG-PET: fluorodeoxyglucose positron emission tomography; MRI: magnetic resonance imaging; SUV: standardized uptake value.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.YK initiated the concept of the case report, performed the literature search, and wrote the manuscript. SY performed the pathological investigations, helped in the literature search, and approved the final write up. KT performed the surgery. TH and KM helped revise the manuscript. TT assisted in the surgery and also helped draft the manuscript. YO and TW helped revise the manuscript. JI helped revise the article. HY contributed to the clinical management of the patient and helped revise the manuscript. All authors read and approved the final manuscript.


Extrapulmonary small cell carcinoma (EPSCC) involving the brain is a rare manifestation of an uncommon tumor type.18F]2-fluoro-2-deoxy-D-glucose (FDG) avid lesions at either diagnosis or follow-up.We report a 59 year-old Caucasian female diagnosed with an EPSCC involving the left parietal lobe without detectable extracranial primary tumor followed by serial positron emission tomography/computed tomography (PET/CT) imaging. Histopathological examination at both initial presentation and recurrence revealed small cell carcinoma. Serial PET/CT scans of the entire body failed to reveal any extracranial [Chemotherapy may show a transient response in the treatment of EPSCC. Further studies are needed to help identify optimal treatment strategies. Combination PET/CT technology may be a useful tool to monitor EPSCC and assess for an occult primary malignancy. First described by Duguid and Kennedy in 1930  EPSCC isAn estimated one thousand new cases of EPSCC occur yearly, with an overall incidence between 0.1% and 0.4% of all cancers . ApproxiThe histologic criteria for EPSCC and small cell lung cancer (SCLC) are the same, namely uniform small cells with dense nuclei, inconspicuous nucleoli and sparse cytoplasm . The preStaging criteria for EPSCC is the same as that for SCLC. Limited disease (LD) is defined as a localized tumor with or without regional lymph node involvement; any extension beyond the loco-regional boundaries is defined as extensive disease (ED) ,10.Clinically these tumors represent a rare, heterogeneous group of neoplasms  characteRecent studies have demonstrated that extensive disease, poor performance status and an increased white blood cell count are the major prognostic factors that correlate with mortality ,10.Optimal management is not well characterized because of the rarity of these tumors, and the lack of randomized clinical trials to guide treatment; hence, there are no standard treatment regimens. Most of the data are extrapolated from treatment of small cell carcinoma of the lung. In this report, we present the clinicopathologic features and serial PET/CT evaluation of an EPSCC of the brain.We report a 59 year-old Caucasian female diagnosed with EPSCC of the left parietal lobe without evidence of an extracranial primary tumor. The patient presented with a three-week history of progressive deterioration of right upper extremity coordination and motor strength. A staging PET/CT scan and CT scans of the chest, abdomen, and pelvis, prior to surgery, were performed that revealed a left parietal lobe mass with an intense, FDG-avid rim anteriorly. Neither study showed evidence of metastasis in the lungs or elsewhere in the body. A magnetic resonance image (MRI) of the brain revealed a 4.4 × 4.2 × 4.5 cm left parietal mass.The patient was treated with intravenous steroids and subsequently underwent an MRI-guided sterotatic left parietal craniotomy with complete resection of the tumor one day after admission. Samples of the resection were sent for pathological review. At diagnosis the complete blood count and complete metabolic profile were within normal limits. The patient did not experience either immediate or late post-surgical complications and was discharged to a rehabilitation facility for post-operative recovery and improvement of her performance status for future chemotherapy.Pathologic examination of the parietal lobe resection was consistent with small cell carcinoma figure . The tumTwo months after initial resection surveillance MRI of the brain revealed recurrence of a left parietal tumor. The patient developed profound, progressive neurological deterioration consisting of hemiparesis and expressive aphasia. At this point she underwent a second complete resection of the tumor. Pathology again revealed a small cell carcinoma with an immunoprofile identical to that of the original specimen.The patient experienced profound improvement in her neurological status after the second resection although her ECOG status was 2. She was discharged to a neuro-rehabilitation facility for recovery and optimization of performance status.2 on days 1 through 5 of a 21-day cycle. Overall, the patient tolerated therapy well with only grade 2 nausea and grade 2 anemia. Response to therapy was assessed by monthly MRI scans of the brain. After two months of therapy a reduction in tumor size greater than 50% was observed. The disease eventually progressed and was referred to hospice care 2 months following progression.Four months following the second resection a 1.7 cm left parietal lesion was identified on MRI and chemotherapy with topotecan was initiated. This patient received six cycles of topotecan at the standard dose of 1.5 mg/mOn repeat PET/CT scans, following the second resection and 9 months after diagnosis figure , there wThe prognosis of EPSCC is similar to that of SCLC with fewer than 13% of patients surviving 5 years  althoughExtrapulmonary small cell carcinoma is a rare, aggressive tumor for which there is no standard treatment guidelines . Some auSince EPSCCs have responded well to agents active against SCLC, it was decided to initiate therapy in this patient. Topotecan was chosen because it was felt that the patient would not tolerate a platinum based regimen due to her poor performance status. Topotecan is a semi-synthetic derivative of camptothecin that specifically targets topoisomerase-I. It has shown clinical activity against SCLC . The useImmunohistochemistry can help diagnose EPSCC as these tumors stain positive for chromogranin A and TTF-1. Ordonez reported that TTF-1 lacks specificity to distinguish primary versus metstatic lesions . Other sWhole-body PET imaging with FDG is used in the diagnosis, staging, and follow-up of many cancers with accuracies ranging from 80% to 90% . PET/CT As the staging procedures for SCLC do not differ from those for non-small cell lung cancer (NSCLC) the primary role of PET/CT imaging is to delineate limited disease from extensive disease. There are relatively few indications for PET/CT scanning in SCLC as there is usually extensive disease at presentation. Nonetheless, it has been shown that whole-body PET is superior to conventional staging in the detection of all involved sites, thus it is a highly valuable tool for staging SCLC . FurtherIt is possible that the patient had an occult bronchial primary tumor that was beyond the limits of detection by PET/CT. There was an interval of nine months between the first and final PET/CT scans; this is a considerable amount of time for a bronchial primary to manifest. During this period the tumor recurred twice in the brain and serial PET/CT scans did not reveal extracranial malignancy at diagnosis or at later dates. As such we are of the strong opinion that the tumor did arise outside the lung.Our patient's response to therapy yielded a transient clinical response without profound toxicity. Further studies are needed to help identify optimal treatment strategies in this rare tumor type. Dual-modality PET/CT technology may be a useful tool to monitor EPSCC and may help in our understanding of this rare entity.18F]2-fluoro-2-deoxy-D-glucose; GFAP: glial fibrillary acidic protein; LD: limited disease; MRI: magnetic resonance imaging; NSCLC: non-small cell lung cancer; PET/CT: positron emission tomography/computed tomography; SCLC: small cell lung carcinoma; TTF-1: thyroid transcription factor-1CAM 5.2: anti-cytokeratin; ED: extensive disease; EPSCC: extrapulmonary small cell carcinoma; FDG: [The authors declare that they have no competing interests.CH, NN, MO, NH, AP conception and design, acquisition of data, analysis and interpretation of data, have been involved in drafting the manuscript, revising it critically for important intellectual content and have given final approval of the version to be published.


Lumbar hernia is a rare abdominal wall defect and clinical suspicion is necessary for diagnosis.We report the case of a 40-year-old Caucasian woman with a superior lumbar hernia (Grynfeltt hernia) initially misdiagnosed as a recurrent lipoma. The correct diagnosis was made intra-operatively and the hernia was repaired using synthetic mesh. The patient was free of recurrence at 4 months after the operation.A lumbar or flank mass should always raise suspicion of a lumbar hernia. Ultrasound and computed tomography may confirm the diagnosis. Adequate surgical treatment should be planned on the basis of etiology and hernia size. Both open and laparoscopic techniques can be used with good results. Lumbar hernias are rare defects involving two weak areas of the posterolateral abdominal wall: the superior lumbar triangle of Grynfeltt, which is the most common site, and the inferior lumbar triangle of Petit. In large congenital or postsurgical hernias the defect wall can affect the entire lumbar region. Lower-back pain is the most common symptom although small hernias may be asymptomatic except for a palpable mass. Misdiagnosis with subcutaneous lipoma is possible. Adequate surgical treatment depends largely on the type and size of the hernia and both open and laparoscopic techniques can be used with good results.A 40-year-old Caucasian woman presented with a diagnosis of a recurrent lipoma of the lumbar region. She had a palpable mass and mild occasional pain at the same site where she had been submitted elsewhere to ambulatory excision of a lipoma. An ultrasound examination confirmed the presence of a subcutaneous lipoma. The surgeon who treated the patient omitted a careful physical examination and an excision of the suspected recurrent lipoma was planned under local anesthesia in our day surgery unit.During the intervention, the aspect of the mass was clearly atypical of a lipoma and the suspicion of a lumbar hernia emerged Figure . The herHer postoperative course was uneventful and she was discharged on the same day. Follow-up at 4 months did not show recurrence.Lumbar hernias are rare and a recent review reported approximately 300 cases . They arLumbar hernia may occur in two weak areas of the posterolateral abdominal wall: the superior lumbar triangle of Grynfeltt, which is the more common site, and the inferior lumbar triangle of Petit. In large hernias the defect wall can affect all of the lumbar region ,4.Symptomatology frequently consists of only lower back pain. Small hernias may be asymptomatic except for a palpable mass. In less than 10% of cases, the onset is acute with bowel obstruction ,6.Anamnesis is helpful for diagnosis in post-traumatic or postsurgical lumbar hernias while in spontaneous adult hernias, misdiagnosis may occur ,7.Clinical suspicion is fundamental to guide imaging diagnosis because extraperitoneal fat herniated through a wall defect may mimic a lipoma. Computed tomography (CT) or magnetic resonance imaging (MRI) in patients with a suspected hernia can confirm the diagnosis adding information on parietal defect size, hernia content and muscular trophism .Adequate surgical treatment depends largely on the type and size of the hernia. A single surgeon cannot gain great experience in this pathology but knowledge gained in treatment of other abdominal wall hernias helps in proper planning of surgery. Both open and laparoscopic techniques can be used with good results .In small defects as present in our patient, the anterior approach is easy and effective; we used a plug with an anchor that provides stability positioned through the fascial defect in the preperitoneal space, and the body and rim can be sutured to muscles. Anterior repair is appropriate for repairing recurrent or large defects with a double mesh or a gluteus aponeurosis flap . LaparosAlthough a rare pathology, knowledge of lumbar hernia is important to avoid misdiagnosis. In particular, a lumbar or flank mass should always raise suspicion of a lumbar hernia. Ultrasound and CT may confirm the diagnosis. Appropriate surgical treatment should be planned on the basis of etiology and hernia size.CT: computed tomography; MRI: magnetic resonance imaging.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.GPM and GA performed the operation. GPM was a major contributor in writing the article and reviewed the relative literature. All authors read and approved the final manuscript.


Surgical resection remains the gold standard for the treatment of localized adenocarcinoma of the extrahepatic bile ducts. Yet, treatment of loco-regional recurrences is not well defined.We present an unusual case of distal adenocarcinoma of the extrahepatic bile ducts that was treated with surgery and relapsed two years later with a solitary recurrence on the tract of a previous Redon drain. In addition, a review of the literature on management of loco regional relapses is presented.The ideal management of these patients still remains undefined. Decisions are made based on clinical parameters from retrospective series, such as tumor grade, surgical margins or lymph node involvement. Prospective studies, that include molecular and genetic markers, are needed to improve patient selection and outcomes on this population. Biliary tract neoplasms are relatively infrequent malignancies. Adenocarcinoma of the extrahepatic bile duct comprises around half of all bile duct neoplasms. Surgical resection remains the gold standard for this disease although adjuvant chemotherapy or radiation should be considered in selected cases. Treatment protocols for loco-regional recurrences are not well defined and include surgery, chemotherapy, radiation, or best supportive care. We present an unusual case of adenocarcinoma of the extrahepatic bile duct treated with surgery that relapsed two years later with a solitary recurrence on the tract of previous Redon drain.th right rib.A 62 year-old Caucasian male presented with a seven-day history of painless jaundice with no other accompanying symptoms. His past medical history was remarkable for high blood pressure, hypercholesterolemia, coronary artery disease, tuberculosis, abdominal aortic aneurysm and benign essential tremor. At admission, physical examination revealed unremarkable findings aside from jaundice. Imaging tests showed a hilar-hepatobilliary mass consistent with the diagnosis of adenocarcinoma of the extrahepatic bile ducts (extrahepatic cholangiocarcinoma). On February 2003, the patient underwent a percutaneous biliary internal decompression followed by a laparotomy and curative resection of the tumour by cephalic duodenopancreatectomy. The resection was performed successfully and histological examination revealed an adenocarcinoma of the extrahepatic bile duct with no lymph node involvement and clear microscopic margins (pT2 pN0 as per AJCC classification). The patPathology analysis revealed a tumour implant through the drainage course where the Redon tube was placed in the first liver procedure Fig . No tumoth 2008 due to refractory septic shock.After the successful resection of this unusual recurrence, the patient received six courses of adjuvant chemotherapy  with good tolerance. On DeceAdenocarcinoma of the extrahepatic bile duct are relatively infrequent malignancies representing less than 5% of all solid tumors in adults and accounting for approximately 0.5% of all cancer deaths. AdenocaSurgery provides the only possibility for cure in these neoplasms. Distal cholangiocarcinomas have the highest resectability rates, while proximal tumors have the lowest (particularly perihiliar neoplasms) -6. In onThe main clinical criteria for resectability are: absence of retropancreatic and paraceliac nodal metastases, no distance liver metastases or disseminated disease, absence of invasion of the portal vein or main hepatic artery , and absence of extrahepatic adjacent organ invasion .Patients with positive margins after resection or positive regional lymph nodes should be considered for adjuvant 5FU-based chemotherapy as well as radiation. Yet, no randomized trials have been conducted that support a standard regimen. Individuals with negative margins after surgery and negative regional lymph node involvement can either be observed or treated with adjuvant strategies .No clear guidelines exist for follow-up after surgery in this particular tumor type. Physical exam with routine laboratory tests every 3 to 4 months for the first 3 years post-surgery and then at longer intervals of 6 months until year 5 seems a reasonable approach. The role of CA 19-9 level in surveillance is not clear, but persistently rising levels often precede radiological evidence of recurrence by a number of months. Therefore this marker has been routinely incorporated in follow-up schemas. Which imaging tests to be performed is a topic that has not been specifically addressed in prospective trials, although CT scans of the abdomen every 6 months for 2 to 3 years after surgery is probably the most common approach in routine practice. However, as referred in the case presented, in several occasions CT and abdominal ultrasound are not sufficient to detect loco-regional relapses, which could be easily determined on MRI and PET.While recurrence is mostly loco-regional in the majority of proximal tumors, distal cholangiocarcinomas recur frequently at distant sites including the liver, peritoneum, and lung,15. LikeThe case presented illustrates an example of an unusual pattern of relapse for adenocarcinoma of the extrahepatic bile duct on a draining sinus from a previous surgical procedure. Moreover it shows how the ideal management of these patients still remains undefined. No prospective data exist to set definitive recommendations about the optimum treatment after a curative resection of adenocarcinoma of the extrahepatic bile ducts. Currently, decisions are made based on different clinical parameters that have been established as prognostic factors in retrospective series, such as tumor grade, surgical margins, or lymph node involvement.Surgery is generally not indicated for recurrent bile duct adenocarcinoma due largely to the location of recurrence, technical difficulty, frequent distant metastases and aggressiveness. However, in patients with prolonged relapse-free interval and favorable location, should be an option to consider.More recently, several molecular markers have been explored as possible determinants of invasiveness and relapse. Expression of EGFR, HER2 and VEGF has been correlated with disease recurrence and could be incorporated into the decision-making process of deciding adjuvant treatment in this patient population . AnotherThe current management of patients with relapsed adenocarcinoma of the extrahepatic bile duct after resection of the primary tumor remains poorly defined and is based only on small retrospective series. Further prospective studies are required in order to design the most appropriate treatment strategy on this population. In addition to the clinical knowledge, it may be beneficial to incorporate molecular and genetic markers into the treatment decision algorithm of this neoplasm.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.JRP and ID conceived the idea for the manuscript, conducted a literature search, and drafted the manuscript. EDV and YQ performed surgery, obtained specimen images and critically revised the manuscript. FPR provided and reviewed pathological images FB obtained radiological images used in the manuscript. MH critically revised the manuscript. All authors read and approved the final manuscript.


Jejunogastric intussusception (JGI) is a rare but serious complication of previous gastrectomy or gastrojejunostomy, and a delayed diagnosis can lead to catastrophe. It can present as hematemesis, and an endoscopist aware of the condition can diagnose it early. We present a case of JGI presenting as hematemesis and diagnosed as tumor bleed on endoscopy. Diagnosis of JGI was confirmed on laparotomy, gangrenous efferent limb was resected and a fresh gastrojejunostomy performed. Jejunogastric intussusception (JGI) is a rare complication of partial gastrectomy or gastrojejunostomy with no medical treatment, and it cThe aim of our report is to highlight the possibility of JGI as a differential diagnosis of tumor bleed in a gastric surgery patient.th postoperative day. Histopathologic examination of the resected bowel showed features of necrosis. He is on our regular follow up and is asymptomatic since the last 6 months.A 65-year-old male patient presented to our emergency department with epigastric pain and vomiting for 2 days and hematemesis for 1 day. He had vomited almost 1.5–2 l of blood over the last 24 hours (the vomited blood was preserved by the attendants in a bowl) before presentation. There was history of diarrhea with melina, a sample of which was presented by the attendants in casualty. Previous records revealed that a retro colic gastrojejunostomy with truncal vagotomy was performed 15 years back for peptic ulcer disease. On examination, the patient was pale with a pulse rate of 96 beats per minute (bpm) and blood pressure (BP) of 100/60. A tender, firm epigastric mass was palpable. Laboratory investigations revealed a hemoglobin (HB) of 7.8 g/dl, total leukocyte count (TLC) of 15,000 with 90% neutrophills. Coagulogram, platelet count and serum creatinine were normal. Two large bore intravenous access lines were established, nasogastric tube inserted, and the patient was catheterized. The patient was being resuscitated by crystalloids and blood transfusions to gain time for upper gastrointestinal (GI) endoscopy. X-ray abdomen was done during the process of resuscitation, which was grossly normal. USG abdomen revealed stomach full of echogenic material (blood). After stabilization, the patient was taken for upper GI endoscopy which revealed findings suggestive of a bleeding gastric tumor with stomach full of clots. The bleeding was not controlled by endoscopic measures, and the patient was taken for emergency laparotomy. On laparotomy, efferent limb of jejunum was intussuscepting into the stomach . GastrotAt the Mayo Clinic in a period of 72 years (1907–1980) years, only 16 well documented cases of JGI have been recognized. JejunojeVarious factors have been incriminated in the etiology of JGI, such as hyperacidity, long afferent loop, jejunal spasm with abnormal motility, increased intra-abdominal pressure, increased motility of efferent loop, adhesions leading to intussusception of a more mobile segment into fixed segment, widening of upper jejunum, vomiting, pregnancy, labor and other causes of increased intra-abdominal pressure, dilated atonic stomach and retrograde peristalsis. RetrograA patient with chronic JGI may present with recurrent episodes of abdominal discomfort which may be associated with nausea that is exacerbated by food, and usually subsides after a couple of hours. In many et al., in a similar case, tried endoscopic reduction of the intussuscepting limb but did not succeed.[et al. reported four cases of acute type II JGI. They were able to reduce the intussusceptum in three cases and in situ resection of the gangrenous jejunum was performed in one case. They recommended in situ resection as the method of choice in case gangrenous intussusceptum is found.[in situ and performed another gastrojejunostomy utilizing the gastrotomy incision. Future recurrence of JGI can be prevented by anchoring the involved jejunal segment to either the neighboring jejunal limb or to the transverse mesocolon.[Emergency surgery is the only answer in acute JGI, and various options include reduction of the limb, resection, take-down of the anastomosis, and revision of the anastomosis. Stefano  succeed. Jain et is found. We resecesocolon.We conclude that JGI with gangrenous, bleeding intussusceptum can mimic a bleeding gastric tumor, and high index of suspicion regarding JGI is needed in an operated case of gastrojejunostomy or gastrectomy, with a palpable epigastric mass.