Teratomas of the sacrococcygeal area are usually diagnosed in infancy and are rarely seen in adults.We report the case of a 36-year-old Greek woman experiencing chronic constipation due to a benign presacral teratoma. Imaging examinations showed a pelvic mass without evidence of malignancy. An ovoid tumour with a maximum dimension of 6 cm was surgically removed. A histologic examination revealed a mature cystic teratoma. Two years after surgery, the patient is well, with no evidence of recurrence and no constipation.Sacrococcygeal teratomas are rare in adults. A high index of suspicion is important in making an early diagnosis. Rectal examination and radiologic evaluation are also valuable. Teratomas are tumours composed of various cell types representing more than one germ layer. Teratomas derived from germ cells occur in the gonads, whereas teratomas derived from embryonal cells are found in other locations. The sacrococcygeal area is the most frequent site of teratomas ) in neonates, infants, and children younger than four years. In adults, tumours at this site are very rare, occurring at a rate of between 1 in 40,000 and 63,000 ,3. We reA 36-year-old Greek woman was admitted to our department complaining of left abdominal and pelvic pain that had developed progressively during the previous six months. She had also experienced constipation for two years, which was temporarily relieved with regular laxative treatments. Recto-sigmoidoscopy revealed extra-luminal pressure. Plain X-ray of the abdomen revealed a mass in the pelvis without extension to the sacrum. Abdominal/pelvic ultrasonography demonstrated a hypoechoic tumor in the lesser pelvis . A computer tomography (CT) scan of the pelvic area showed that the rectum and urinary bladder were displaced anteriorly by a mixed-type mass with soft tissue elements, inhomogeneous fatty tissue and calcifications, extending from the presacral area to below the coccyx Figure . On magnRoutine laboratory tests were within normal ranges. Human chorionic gonadotropin (hCG) and serum tumour markers, including alpha-fetoprotein (aFP), carcinoembryonic antigen (CEA) and carbohydrate antigens  were not elevated.During laparotomy, a 5 cm mass was found posterior to the rectum and anterior to the sacrum, extending between the anal sphincters Figure . The tumThe surgical specimen was an ovoid cystic lesion measuring 6 × 5 × 4.5 cm. On sectioning, two cystic cavities were found, the larger with a maximum dimension of 5 cm and containing sebum, and the smaller one measuring 1.5 cm and containing mucin. On histologic examination, the larger cavity was lined with stratified keratinizing squamous epithelium and the smaller one was lined with pseudostratified mucin-producing columnar epithelium with occasional ciliated cells. The cyst wall was composed of fibro-adipose tissue, smooth muscle fiber and glandular structures with dilated lumens. A diagnosis of mature cystic teratoma was made.SCTs originate either ventral or dorsal to the sacrum, and may grow posteroinferiorly into the gluteal area or anterosuperiorly into the lesser pelvis. In the ventral location, these tumours may grow to a large size as they develop into the retrorectal or presacral space. The clinical presentation of SCTs is mainly attributed to their growth and size, or to dysfunction of adjacent organs due to local pressure or infiltration . When thDifferential diagnosis is mainly from congenital abnormalities and tumours of the presacral area, such as anterior meningocele, rectal duplication, tailgut cyst, neurogenic tumours , osseous , and cysts, soft tissue tumours  as well as uterine, ovarian and metastatic tumours.Cystic teratomas may often be complicated by inflammation and therefore may be associated with soft tissue destruction. Thus, SCTs may be misdiagnosed as high lying pararectal abscesses, fistulas with presacral extension, pilonidal cysts with abscess formation, postinjection granulomas, and osteomyelitis or tuberculosis of sacrum ,7.Concerning the radiological evaluation, multiple dense calcifications are present in 50% of teratomas on plain radiographs. Irregular calcifications are reported to be present in 25% of malignant teratomas , in compThus, the preoperative diagnosis can be assisted significantly by modern imaging techniques such as CT and MRI scanning. CT scanning clearly demonstrates whether the tumour consists of cystic and/or solid components with fluid, fat, soft tissues, and calcifications. MRI can be used to evaluate the presence or absence of bone or nerve infiltration, to define accurately the tumour extent and to delineate soft tissue planes for surgical planning ,9.In the case of our patient, CT and MRI showed calcifications within the tumour and absence of sacral infiltration, as well as clear planes between the lesion and the adjacent organs suggesting that this was a benign tumour.Surgery is the preferred treatment for presacral teratomas if the tumour can be completely extirpated. Posterior , anterior , or combined approaches have been used, depending on the tumour size and location .The anterior approach is typically performed for high lesions without evidence of sacral involvement, as was the case in our patient. Attention must be given to the middle sacral artery, which is frequently the major supplying vessel. This approach has the advantage of providing excellent exposure of important pelvic structures, such as the iliac vessels and ureter .Sacrococcygeal teratomas are rare in adults. A high index of suspicion is an important factor in making an early diagnosis; rectal examination and radiologic evaluation are also valuable.aFP: alpha-fetoprotein; CEA: carcinoembryonic antigen; CT: computer tomography; MRI: magnetic resonance imaging; SCT: sacrococcygeal teratoma.Written informed consent was obtained from the patient for the 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.DP and TSP admitted the patient in the emergency room, analyzed and interpreted the patient's data and drafted the manuscript. AM, VNP and SA were the surgeons and the attending physicians and also revised the manuscript. DT and PH performed the histological examination of the mass. All authors read and approved the final manuscript.


The management of very long tubular bowel duplications poses a special challenge to even the most skilled surgeon. In these cases, mucosal stripping is usually employed. We report a novel case of a two-year-old boy, with 120 cm long jejuno-ileal duplication, wherein, bowel salvage was achieved, utilizing the Bianchi principle, originally described for bowel lengthening in cases of short bowel syndrome. Duplications of the alimentary tract are rare. The treatment needs to be tailored for each case because of a wide spectrum of presentations. The salvage of almost 120 cms of small bowel by dissection through one of the mesentric leaves based on the Bianchi technique,[1A two-year-old male child presented with abdominal pain of 15 days duration. There was no history of vomiting or constipation. He had a slightly distended abdomen. A vaguely palpable lump could be felt on abdominal examination. It occupied almost the entire abdomen. The child weighed 10 kg. His haematologic investigations were normal.An erect radiograph of the abdomen showed a well-defined, huge, soft tissue lesion in upper abdomen. There was paucity of gas in the abdomen and pelvis. Two to three different cysts were noted within the abdomen on ultrasonography. Computed tomogram (CT) scan of the abdomen showed a 10×5 cm large, cystic, lesion in the lower abdomen, continuous with the bowel wall .At laparotomy, cystic duplication of the jejunum was seen. A cyst measuring 12×10 cm in size was connected by a small stalk to a larger cyst of about 28×12 cm in size. There was no communication with the native jejunum. The blood vessels of the involved bowel passed both anterior and posterior to the cyst. Enucleation of the cysts was done by opening one leaf of the jejunal mesentery and preserving the vascularity of the bowel, through the posterior arcade. After near complete enucleation of the cystic duplication, another 70 cm long tubular duplication was found in continuity , invadinThe tubular duplication was entering the terminal ileum at its distal end. There were a lot of hypertrophic mucosal folds at the entry point into the ileum. This portion (about 8 cm of bowel) was excised and local end-to-end anastomosis was done. The operative time was five hours. Blood loss of 60 cc was replaced. The dissection was aided by use of bipolar diathermy and operating loupes with 2.5×magnification.The excised mass weighed 750 g and contained viscid mucous. The histopathology showed the intestinal type lining mucosa. The patient had an uneventful post-operative recovery. At 1-year follow-up, the child is healthy and asymptomatic.In our case, there were two large cystic duplications i.e., 12×10 cm and 28×12 cm of the proximal jejunum, connected to each other by a small stalk. This was further continuous with a 70 cm long tubular duplication . The cysIn this situation, excision of the involved bowel would have significantly compromised the bowel length. Hence, we enucleated the two large cystic duplications and separated the long tubular duplication from the native bowel by dissection of the common wall. By coagulating and dividing the overlying blood vessels of the anterior mesenteric leaf based on Bianchi principle, salvage In the Bianchi technique, the entire circumference of the bowel is divided into two halves, each of which is supplied by blood vessel from one mesenteric leaf. Whereas,Though such type of dissection has been utilized for limited lengths of small bowel, preservaHigh degree of technical expertise, patient bowel handling, gentle dissection using bipolar diathermy and magnifying operating loupes and vigilant post-op care salvaged the small bowel in this two-year old child.


A number of non-malignant diseases that share similar morphological features as gastrointestinal stromal tumor (GIST) have been reported. Co-existence of GIST with these other diseases is rarely recognized or reported.We report a case of a 62 year-old man with long-term stable control of metastatic GIST with systemic therapy, presented with an apparent intra-abdominal progression but not supported by imaging with positron emission tomography. Subsequent resection of the intra-abdominal tumor identified a non-malignant fibroid.Differentiating localized progression of GIST from other diseases has important prognostic and therapeutic implications. The potential for co-existence of non-malignant soft tissue neoplasm should always be considered. The finding of gain-of-function mutation of KIT has revolutionized the treatment of advanced gastrointestinal stromal tumor (GIST). This has subsequently led to development of effective systemic therapy utilizing tyrosine kinase inhibitors (TKI). Imatinib is the prototype TKI that was initially reported to achieve a partial response rate of 53.7% and stable disease rate of 27.9%. With thA number of soft tissue neoplasm share many similarities in the morphological and immunophenotypic profiles with GIST. Aggressive fibromatosis (AF) and keloid type fibromatosis scar tissues are distinct soft tissue tumors. AF is a fibroproliferative disease with a propensity for intra-abdominal presentation; it may In this report, we present a case of a man who had been treated with imatinib and achieved long-term stable advanced GIST, but presented with localized proliferation of soft-tissue neoplasm mimicking GIST.A previously healthy, 62 year old man was diagnosed with a gastric antral tumor after investigations for symptomatic anemia. A barium swallow confirmed the presence of tumor causing subacute gastric outlet obstruction. Laparoscopy identified a gastroduodenal tumor and synchronous bilobar liver metastases. No peritoneal disease was identified. The primary tumor was completed excised and a liver biopsy was performed intra-operatively. Histopathology was consistent with metastatic malignant gastrointestinal stromal tumor, with typical spindle cell features on light microscopy. C-KIT was positive and the mitotic rate was 60/50 per high power fields Figure . SubsequPost-operatively, this man recovered well from his surgery and was commenced on imatinib 600 mg daily; he subsequently required dose-reduction to 400 mg daily due to grade 3 neutropenia. He still had residual hepatic metastatic disease which was visible on computerized tomography [CT] scan, and was FDG-avid on positron emission tomography [PET] evaluation. For a period of eighteen months after surgery, imatinib was tolerated well and achieved good disease control. CT and PET imaging during this period revealed regression of size of the liver metastases. After two years of therapy, however, a CT scan revealed an increase in size of a dominant segment VI hepatic metastasis which was treated with radiofrequency ablation. He was then maintained on imatinib at 600 mg daily with subsequent disease control.At four and half years from diagnosis, an asymptomatic infrapyloric mesenteric mass was identified on a surveillance CT which progressively increase in size over the next two months Figure . A PET sAt subsequent laparotomy, the tumor was found to be lying within the peritoneal leaves of the mesocolon extending from the origin of the superior mesenteric vessels to the inferior pancreatico-duodenal vessels. Histopathology showed a tumour mass composed of spindle shaped fibrocytic/fibroblastic like cells amongst intervening collagen Figure  with lowImatinib was continued 600 mg daily, with brief cessation during the peri-operative period, as metastatic GIST remained radiologically stable. No specific adjuvant therapy for the soft-tissue tumor was employed post-operatively.In this patient with metastatic GIST, the development of the mesenteric tumor four years after the institution of imatinib initially suggested disease relapse. Debulking surgery remains a recognized standard practice in the case of local progression where such procedure is associated with prolonged survival with the elimination of imatinib resistance clones. HoweverThis patient underwent surgery with the pre-surgical diagnosis of a localized progression; surgery was aimed to achieve disease control with the elimination of a presumably localized imatinib resistance tumor. Post-surgery, the histopathologic findings revealed a tumor with reduced cellularity and low mitotic activity consistent with the pre-operative non-glucose avid PET findings. Collaborative pathologic review was obtained and excluded diagnosis of a recurrent GIST. However, a definitive uniform diagnosis could not be made. The possible differential diagnoses of this soft-tissue tumor include aggressive fibromatosis (AF) or intra-abdominal keloid type fibrocollagenous scar.Immunohistochemistry was positive for C-KIT, which is unusual in AF or intra-abdominal keloid type fibrocollagenous scar. Mutation analysis that was performed on the mesenteric tumor further clarify that this mass, which was absent of Exon 11 C-KIT mutation, was different from the Exon 11 C-KIT mutation positive of the original resected antral GIST.APC or CTNNB1). Some studies [Histologically, AF lies on a spectrum of disorders characterized by excess proliferation of fibroblast-like spindle cells. These c studies -11 have There is very limited literature on intra-abdominal keloid type fibrocollagenous scar. The scar in this patient was presumably formed from previous surgical laparotomy. There is growing evidence to suggest that Transforming Growth Factor β is impliThe immunophenotypic profiles of GIST, AF and keloid may overlap. Fibromatoses may stain for vimentin, smooth muscle actin, and desmin. In some series, fibromatosis did not stain for CD34 or S-100 protein, while CD34 staining and occasional S-100 protein positivity were seen in GIST,14. It hThe long stable disease control, the absence of glucose avidity of pre-operative PET and the absence of the C-KIT mutation in the mesenteric tumor were all features that might have suggested the possibility of an alternative diagnosis to GIST recurrence. Although surgical resection of this mesenteric mass may remain necessary, a correct diagnosis has important implication for his future systemic therapy. This case report highlights the importance of recognizing the coexistence of other diseases in patients with chronic GIST.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.CL and AH drafted the original manuscript, with subsequent further contributions from KD, GS and DG. KD reported the histopathological findings and supplied the photomicrographs used in this manuscript. GS reported the surgical findingsAll authors read and approved the final manuscript.


Benign metastasising leiomyoma refers to a type of lesion characterised by leiomyomatous alterations without any indication of malignancy. It presents as either a singular nodule or multiple nodules of proliferating smooth muscle cells and is generally found in the lungs of women who have undergone a hysterectomy. The purpose of this case report is to contribute to the knowledge of this rare disease by presenting evidence and experience of a patient case. In particular, this report seeks to investigate the therapeutic approaches in order to understand whether a standard of care can be prescribed and whether the use of prophylaxis therapy with progesterone as a follow-up to surgery serves as a reasonable treatment in certain cases diagnosed as benign metastasising leiomyoma.We present the case of a 52-year-old Caucasian woman who developed a pelvic relapse and a pulmonary localisation of benign metastasising leiomyoma following a hysterectomy for myomatous uterus.Our literature review revealed a single case of the use of chemoprophylaxis as treatment of a benign metastasising leiomyoma. The role of chemoprophylaxis in preventing future recurrences remains unclear. The use of progesterone as an adjuvant therapy for benign metastasising leiomyoma could simply be palliative, with associated psychological benefits, or it could be of therapeutic significance. There are conditions, although rare, in which histologically apparently benign leiomyomas of corpus uteri extend beyond their usual boundaries or are associated with extra-uterine leiomyomas. The term benign metastasising leiomyoma (BML) refers to a type of lesion characterised by leiomyomatous alterations without any indication of malignancy. It presents as single or multiple nodules of proliferating smooth muscle cells, usually in the lungs of women who have undergone a hysterectomy.Controversy exists as to whether lung leiomyomas represent the synchronous or metachronous development of an independent lung lesion; the term more readily describes the metastases of a benign uterine primary tumour, and certainly recent molecular investigators tend to favour this latter definition .The purpose of this report is to contribute to the knowledge of this rare disease by presenting evidence and our experience of a patient case. In particular, this case report seeks to investigate the therapeutic approaches in order to understand whether a standard of care can be prescribed and whether the use of prophylaxis therapy as a follow-up to surgery serves as a reasonable treatment in certain cases diagnosed as BML.A 52-year-old Caucasian woman, gravida IV, para 2, abortus 2, with a 5-year history of uterine leiomyomas, presented in January 1999 with a pelvic mass of 87 mm at its greatest diameter at sonography. In July 2000, the mass had increased in volume, and in January 2001, the patient was admitted to hospital for a total abdominal hysterectomy with a bilateral salpingo-oophorectomy owing to uterine leiomyomas.At laparotomy, the uterus was found to be three times the normal size (14 cm × 14.5 cm). In addition, a mass of about 30 mm in diameter located in the right laterocervical region was resected; this second mass was softer and less resistant to indentation than the leiomyomas inside the uterus. The ovaries appeared to be regular. The histological exam confirmed the presence of multiple leiomyomas of the corpus uteri and a right laterocervical leiomyoma. All of the specimens considered were positive for oestrogen and progesterone receptors. At 2-month follow-up, there was no evidence of disease. More than 10 years before admission, two incomplete abortions had occurred in the first trimester of pregnancy and had been followed by uterine curettages.After 3 years, the patient presented with abdominal bloating and occasional abdominal pain. A pelvic sonographic examination revealed a mass of 12 cm at its greatest point. In December 2004, magnetic resonance imaging was carried out. This revealed the pelvic mass to be solid and non-homogeneous, as evidenced by the presence of haemorrhagic areas, necrosis and vascular structures; the non-homogeneous nature was reinforced following the injection of contrast medium whereby non-homogeneous high enhancement became apparent. The mass was connected to the right pelvic wall via a vascular supply that appeared to originate from the iliac vessels in the obturator region. The rectum and bladder walls did not appear to be affected by the growth of the mass. A positron emission tomography-computed tomography (PET-CT) total body scan was performed, from which a pelvic localisation with low glucose affinity could be concluded. There were no pulmonary masses on pre-operative chest X-ray.In order to remove the mass, a second abdominal intervention was performed, which revealed no evidence of tumour invasion in the pelvis or abdominal viscera. The mass measured 13 cm × 9 cm × 6 cm, was delimited by serosa, and when cut appeared to be myxoid and with oedematous areas inside.Histologically, spindle-shaped smooth muscle cells were present each containing a regular oval nucleus, without mitosis and with a moderate vascularisation and oedematous aspect. The cells were positive for smooth muscle specific actin, and there was a low proliferation index (2% of nucleus being MIB-1 positive). Again, the specimens expressed oestrogen and progesterone receptors Figures . The finIn light of the histological results and the lack of evidence to suggest immediate prescription of radiotherapy or chemotherapy, the decision was taken to withhold treatment and await observation at follow-up.A routine chest X-ray the following year, in May 2005, showed a nodular posterior basal density in the right lung of about 4 cm in diameter. The presence of a single nodular mass was confirmed at CT scan of the thorax ; a low MIB-1 index of 1%; and a positive immunohistochemistry reaction for oestrogen receptors, progesterone receptors, H-caldesmon and desmins. Moreover, testing was negative for keratins, Bcl2, CD10 and CD99.A review of the histological pattern of the original mass and pulmonary mass showed low mitotic indices in both the pelvic mass (2 mitoses per 10 HPFs) and also the pulmonary mass (less than 1 mitosis per 10 HPFs) and that the histology of the masses was similar, typifying a leiomyoma Figures  and 5.One month after the intervention, a course of chemotherapy prophylaxis was commenced using 80 mg per day of megestrol. At the 6-month follow-up, a CT scan of the thorax and abdomen was negative for masses; the 12-month examination was similarly negative.In our literature review, we could only find around 100 documented cases that make reference to BML diagnoses . Even ifIn explaining the histogenesis of these lesions, opinion in the existing literature can be classified into three main streams. For proponents of the first approach, the uterine neoplasm is regarded as a low-grade leiomyosarcoma with malignant potential. The second approach centres on the presence of lung emboli formed of cells that originated from a benign leiomyoma of the uterus. The third and final hypothesis asserts that multi-focal smooth muscle proliferations may be the result of the independent growth of smooth muscle tissue in response to hormonal milieu.Serving to validate the hypothesis that staminal mesenchymal cells from the myometrium are embolised to other organs , our patient, in common with many other documented cases, had a previous history of gynaecological surgery, including curettages and hysterectomy.Lesions are discovered incidentally, although symptoms such as coughing, chest pain and dyspnoea have been noted. Furthermore, lesions produce differing outcomes, some of which lead ultimately to fatality.The differential diagnosis for pulmonary nodules on chest radiography includes benign and malignant primary and metastatic neoplasms, vascular lesions, infectious and noninfectious inflammatory granuloma and collagen vascular disease. In every instance when a locally relapsed disease with a distant metastasis is discovered, all of the observed possibilities must be considered. One important step that brings us to diagnosis involves performing either a biopsy or a surgical resection.Pulmonary specimens for histological analysis have been obtained through various methods, including percutaneous biopsy, transbronchial lung biopsy and lobectomy. The greatest challenge in distinguishing a leiomyoma from a leiomyosarcoma concerns assigning the intermediate combination of diagnostic criteria, in order to decide whether the patient is experiencing a benign or malignant disease.When mesenchymal cell neoplasms are present, the histological differential diagnosis falls between BML, primary pulmonary leiomyoma, leiomyosarcoma, metastatic BML from a source other than female genital internal organs, hamartoma and lymphangioleiomyomatosis.The first therapy proffered in the literature relates to the surgical resection of the pelvic recurrence and, where possible, the pulmonary localisations. Several authors noted a more favourable outcome after bilateral oophorectomy, including a curative aspect of this surgical intervention . Howeverin vitro for decreasing cell numbers and for stopping cell proliferation [in vivo [Tamoxifen has proven to be effective feration , but its[in vivo . Another[in vivo . Aromata[in vivo . Progest[in vivo .The utilisation of long-acting GnRH analogues, which suppress pituitary gonadotrophin biosynthesis by decreasing the number and sensitivity of GnRH receptors, has been documented with favourable results in several reports . The litA number of different classes of growth factors and apoptosis-related factors have been identified as having a high likelihood of affecting leiomyoma growth, vascularity and extracellular matrix deposition: epidermal growth factor, insulin-like growth factors, transforming growth factor-β family, platelet-derived growth factor, angiogenic factors, Bcl-2 protein, tumour necrosis factor-α and p53 protein.The heterogeneity of leiomyoma growth within the same uterus, despite the identical exposure to circulating sex steroid concentrations, suggests the involvement of local growth factors. These are expressed differently in normal smooth muscle and leiomyoma, which indicates that such factors may be involved in paracrine stimulation .These results indicate that future non-surgical treatments for leiomyomas may include compounds that block the actions of specific growth factors involved in the control of uterine smooth muscle cell proliferation and growth.The complexity of the interactions existing between specific growth factors, hormonal composition and leiomyoma behaviour and development forms the basis for understanding how a certain therapy can achieve a positive outcome in one instance, and yet fail to take effect in others. In order to make an informed decision as to which treatment to administer, an improved characterisation of the molecular expression and genetics of leiomyoma is necessary. Such information will likely clarify the reasons why certain therapies appear more efficacious with specific types of leiomyoma . Future A review of BML therapies was carried out with the intention of clarifying the rationale for the use of a drug prophylaxis to prevent recurrences. Following an analysis of the existing literature, it was ascertained that the use of progesterone (medroxyprogesteron) as a prophylactic agent was limited to a single documented case .In our patient, following two recurrences and consequent surgical resections within a short period, the decision was taken to use a chemoprophylaxis agent despite the uncertainty surrounding its role in preventing future recurrences . As a coBML: benign metastasising leiomyoma; CT: computed tomography; HPF: high-power field; MRI: magnetic resonance imaging; PET-CT: positron emission tomography-computed tomography.The authors declare that they have no competing interests.APL, RJL and DM carried out the literature research and drafted the manuscript. PP made pathology contributions. CM and FL carried out the clinical and surgical management and helped in drafting and the critical revision of the 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.


This report calls attention to severe gastrointestinal hemorrhage as a manifestation of small bowel carcinod tumor.We report a case of an adult in his 5th decade presenting with massive melena.On presentation the patient was tachycardic (pulse115/min), and hypotensive (BP 80/55 mmHg). His complete physical examination was unremarkable. Rectal exam showed frank blood. Nasogastric lavage produced greenish fluid with no blood. His hemoglobin was 8.1 gm/dl and hematocrit was 25.1%. Panendoscopy revealed an ileal bleeding mass. Interestingly a colonoscopy done 6 months earlier for anemia workup was normal. Emergent laparotomy revealed numerous smaller intraluminal nodules scattered throughout the small bowel.Histopathology revealed carcinoid tumor. Carcinoid tumors are apudomas that arise from enterochromaffin cells throughout the gut. Small bowel carcinoids are most commonly found in the ileum, within 60 cm of the ileocecal valve . The prest, 2008) reveals only three cases reported for LGI bleed caused by small bowel carcinoid tumor [Any bleeding distal to ligament of Treitz is considered as lower gastrointestinal bleeding. Most cases of lower gastrointestinal bleeding involve the colon 85%), approximately 10% are actually due to upper gastrointestinal bleeding and only 3% to 5% cases are secondary to bleeding from small intestine . While r%, approxid tumor . To our 2 saturation 100% on room air. He was not in acute distress, abdomen was soft, non-tender, non-distended, no masses or organomegaly and normal bowel sounds. Rectal exam revealed frank blood. Significant initial lab findings included a hemoglobin level of 8.1 gm/dl and hematocrit of 25.1%. Aggressive fluid replacement via two wide bore IV lines was instituted accompanied by two units of PRBC and he was admitted to the intensive care unit. After stabilization, an EGD did not show any bleeding source. Colonoscopy showed a bleeding mass [Figure A 44 year old male, presented to the ER with complaints of bleeding per rectum for 1 day. His complaints started with 4 episodes of watery diarrhea with dark blood. He denied any fever, abdominal pain, weight loss, and change in bowel pattern, hematemesis or jaundice. The patient had immigrated to USA 18 months prior from India where he was found to be anemic requiring two blood transfusions over the last four years. Past medical history was significant for anemia only and his home medication was an iron and folate supplement. His anemia was investigated with an esophagogastroduodenoscopy (EGD) and colonoscopy 6 months prior in India, both of which were normal. In the emergency room his vital signs revealed: temperature 98.6°F, pulse 115/min, BP 80/55 mmHg, respiratory rate 32/min and OThe etiology of massive LGI bleed includes diverticulosis, angiodysplasia, polyps, dieulafoy's lesion, infectious or inflammatory bowel disease Table . CarcinoOur patient was unique in that he was younger and did not have any of the typical symptoms. He presented with massive LGI bleed which is unusual for small bowel carcinoid that tends to be slow growing and may remain asymptomatic. Further, as demonstrated in our patient, standard endoscopic examination may miss them. Thus these are difficult to diagnose and as in our patient maybe rarely detected before a surgical procedure. As the carcinoid tumor is highly infiltrating early diagnosis is mandatory. Thus a high index of suspicion and the utilization of other modalities for complete examination of the small bowel such as capsule endoscopy, mesenteric angiography, and radionuclide RBC scan should be considered in patients with LGI hemorrhage in whom EGD and colonoscopy are negative.Carcinoid tumors can cause massive lower GI bleeding. Because of a substantial false-negative rate for lesions on initial endoscopy as demonstrated in our patient, and also because of the fact that small bowel carcinoid tumors are highly infiltrating thus mandating early diagnosis; an aggressive approach should be taken.The authors declare that they have no competing interests.GD drafted the abstract and manuscript, participated in the patient management. MP supervised the abstract and manuscript, supervised patient management. Both authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Inflammatory myofibroblastic pseudotumours of the liver are rare tumour-like lesions that can mimic malignant liver neoplasms. The symptoms and radiological findings of this rare tumour can pose diagnostic difficulties. We describe a 69-year-old gentleman who was admitted to our department with symptoms suggestive of acute cholecystitis. Ultrasonography and computed tomography of the liver raised the possibility of metastatic liver disease. A core biopsy of the liver was performed to confirm the diagnosis of liver metastasis. Unexpectedly it showed no evidence of malignancy but instead revealed an inflammatory myofibroblastic pseudotumour of the liver. This case report highlights the diagnostic dilemma that arose due to the similarity of appearances between the two pathological entities on imaging and this stresses the need for accurate histological diagnosis so as to avoid unnecessary surgical intervention. To the best of our knowledge, only a minority of cases are reported in the literature associating a hepatic inflammatory myofibroblastic pseudotumour with gall stones. An inflammatory myofibroblastic pseudotumour (IMFP) of the liver  is a rare, benign, tumour-like lesion first described in 1953 by Pack and Baker . The symA 69-year-old Caucasian gentleman presented to the surgical department with right upper quadrant pain, nausea, vomiting and a recent weight loss. His medical antecedents included rheumatoid arthritis and bronchiectasis. On examination, the patient was found to have tenderness in the right hypochondrium. An abdominal ultrasound (US) scan revealed a thick-walled gall bladder with multiple calculi and a normal looking liver. Laboratory investigations showed a decreased haemoglobin concentration (11.6 g/dl), an elevated C-reactive protein level (137 mg/l) and an increased erythrocyte sedimentation rate (98 mm/hr). The liver function tests were deranged with a raised alkaline phosphatase (412 u/l), a raised alanine transaminase (53 u/l) and a normal serum bilirubin level. An initial diagnosis of cholecystitis was made and the patient was treated empirically with antibiotics.Whilst waiting for a laparoscopic cholecystectomy, the patient once more presented to the emergency department with recurrent episodes of abdominal pain. A repeat abdominal US scan revealed an ill-defined area in the right lobe of the liver with altered echogenicity. This ambiguous region was considered to represent either metastatic infiltration or an early hepatic abscess. Subsequently, computed tomography (CT) revealed multiple low attenuation lesions in the right lobe of the liver, the largest measuring 5.5 cms Fig . The lesAn IMFP is a benign, tumour-like mass characterized by proliferating fibrous tissue infiltrated by inflammatory cells . This coThe etiology of IMFPs remains unclear although infection, radiation and chemotherapy have been suggested as possible causes . More rIMFPs most often occur in childhood and early adulthood in non-European populations . The malRadiological diagnosis of this tumor can be demanding, sonographically the lesion may be hyperechoic, hypoechoic, or complex in echogenicity. CT imaging may help alleviate the diagnostic dilemma common to patients presenting with IMFPs. Enhancement patterns between hepatocellular carcinoma (HCC) and IMFPs differ significantly, with HCCs exhibiting hyperdensity in the arterial phase and hypodensity of the capsule in the delayed phase. On MRI the lesion demonstrates increased signal intensity on both T-1 weighted and T-2 weighted sequences in relation to a normal liver . These vHistologically, IMFPs tend to appear as densely hyalinized structures with whorl-like patterns of dense collagen bundles infiltrated by multiple inflammatory cells. No features of malignancy are present. There is great histological variation and the features of IMFPs tend to be non-specific. This has led to IMFPs becoming a diagnosis of exclusion in adults .To the best of our knowledge, only a minority of cases associating hepatic IMFP with gall stones are reported in the literature . The exaThe management of an IMFP of the liver has traditionally been surgical but more recently spontaneous regression of the tumour has been reported with the use of antibiotics or non-steroidal anti-inflammatories as was evident in our patient ,12,15. SIn conclusion this case report emphasizes the importance of considering an IMFP as part of the differential diagnosis and highlights that a conservative approach to a patient with an IMFP is an effective treatment option.IMFP: Inflammatory Myofibroblastic Pseudotumour; US: Ultrasound; CT: Computed Tomography; ESR: Erythrocyte Sedimentation Rate; CRP: C - reactive protein; GI tract: Gastrointestinal tract; FNA: Fine Needle Aspiration: HCC; hepatocellular carcinoma.The authors declare that they have no competing interests.PS, IS, AW and TAJ were involved in the acquisition of data and it's analysis. PS, TAJ and IS wrote and edited the manuscript. All authors have read and approved the final manuscript.Written informed consent was obtained for the publication of this article and the accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


The clinical presentation and preoperative diagnosis of this anomaly are discussed.Cystic pancreatic tumors are rare in children and the immature cystic teratoma of the pancreas is even rarer. A review of the world literature shows 18 documented cases involving all the age groups. The preoperative evaluation of this lesion is rather questionable, with definitive diagnosis taking place intraoperatively. We report the 19 Teratomas can be divided into two subtypes: mature and immature. The mature type can be further subdivided into solid type and cystic type, hence, dermoid cyst. Derived from totipotent stem cells, they possess the ability to generate tissues from all the three germ layers: endoderm, mesoderm and ectoderm.A 5-month-old male child (weight: 7 kg.) was admitted to our hospital with a history of abdominal swelling for 1 month. His birth was normal vaginal delivery at 38 weeks of gestation and the postnatal period was unremarkable. A mobile smooth, nontender mass was palpable in periumbilical area during physical examination. Routine blood and urine biochemistry profiles were normal. The serum tumor marker AFP was normal. Abdominal ultrasonography demonstrated a large septate cystic mass (size: 8.0 × 7.0 cm) containing an echogenic solid component (size: 4.5 cm × 3.5 cm) in right lumber/hypochondriac region with a possibility of pancreatic cyst. Pancreas was not clearly observed because of the poor window . CT scanSurgical exploration of the patient revealed a large cystic mass at the head of the pancreas, compressing on 2nd and 3rd part of duodenum . InitialDermoid cysts are thought to arise from the embryonic inclusion of skin at the time of neural groove closure; thereforThe important distinguishing feature of teratoma is the presence of tissues derived from all three germ cell layers within a single lesion. Teratomas are classified as “mature” and “immature” teratomas, which replace the terms “benign” and “malignant” teratomas, respectively. The term immature teratoma is used for tumors containing primitive neuroectodermal, endodermal or mesodermal tissues. Cells isUnlike dermoid cysts present elsewhere in the body, little radiographic evidence is available regarding their pancreatic location. However, by extrapolating the documented findings to the pancreas, it appears equally so that the radiological appearance of these lesions depends on the proportions of the various tissues that are affected by these lesions. Ultrasouet al.[At this point, an excisional biopsy is usually performed. However, the prospect of cytologic diagnosis should not be overlooked. In 1991, Markovsky et al. describeTreatment is surgical extirpation, i.e., simple excision of cyst. In view Intrabdominal teratomas generally present as abdominal masses and entail good prognosis on timely treatment. Pancreatic trauma are extremely rare and are difficult to diagnose preoperatively, the diagnosis is often made in retrospect once the histology of the excised mass is studied.


Nongalenic intradural arteriovenous fistulas, although uncommon, are clinically important. Choosing the appropriate therapeutic approach has been a controversial issue within the last decade.A 15-year-old male was presented with a calcified nongalenic arteriovenous fistula in the left parietal region, supplied by the left middle cerebral artery, and draining into the left lateral sinus. The patient was managed surgically with traditional clipping the feeder artery, along with piecemeal resection of the huge calcified mass. Although endovascular methods may be the treatments of choice in similar cases, in such huge calcified lesion, non-amenable to endovascular occlusion, open surgery seems to be preferred. Arteriovenous fistula (AVF) is a rare entity occurring at different sites of the intracranial structures. The lesion has been described as a direct connection between an artery and a vein without any intervening nidus, dissimilar to true arteriovenous malformation (AVM) . These lIn this report, a case of huge nongalenic AVF with a pattern of wonderful intracerebral calcification is presented.A 15-year-old left handed male was admitted because of a 5-year history of weakness in the right extremities, aggravated within 3 months. The patient was a product of normal gestation and delivery, and other than a history of birth head trauma, he had uneventful medical history and perfect developmental milestones.At admission time, physical examination revealed a right hyperreflexia, hemihyposthesia and hemiparesis, with the estimated force of 4/5 according to Louisiana State University Medical Center grading system [Brain computed tomography (CT) and magnetic resonance imaging (MRI) disclosed a huge (7.8 × 5.3 cm) intra-axial elliptical mass with a calcified margin in the left parietal region, causing a midline shift of 1 centimeter and containing hemorrhagic components of different ages Figure  and 2B. Echocardiography displayed no heart failure or valvular abnormality.Open microsurgery was performed, and through opening the Sylvian fissure the hypertrophic distal segment of MCA was exposed, reaching to a calcified mass in the distal part of the Sylvian fissure. The feeder artery was clipped proximal to its entrance into the mass. Piecemeal resection of the calcified wall Figure  was perfth day displayed the left ACA to fill in a normal pattern, with the MCA stem refilling to the end of Sylvian fissure, where the lesion was previously resected (Figure The patient was sedated for 2 days using midazolam and sodium thiopental. He was conscious following extubation on the third day without neurological deterioration. Control brain CT on the day following the surgery revealed neither remnants nor hematoma figure . Controld Figure  and 2D.Histopathological assessments demonstrated mature vascular structures with clot and atheromatous plaques and encircling calcified layer Figure .The patient returned to school one month later, and no episode of headache or seizure has happened in 2 years follow-up.Arteriovenous fistula is an uncommon but clinically important condition. Theoretically, the fistula consists of an arteriovenous shunt, having a direct passage with no intervening nidus or vessels ,7.Various kinds of classification of AVFs have been established, based on location, draining veins, flow speed, and anatomical structures. Basically, arteriovenous fistulas are categorized to dural and intracerebral lesions. The latter is equivalent to the so-called intra-axial, intradural, cerebral, and pial AVFs. Pial fistulas differ from the dural type as they acquire feeders from cortical arteries. They are further categorized into galenic and nongalenic forms. Nongalenic cerebral AVFs do not involve the persistent embryonic median prosencephalic vein ,1. NongaAlthough AVFs may present in childhood, most cases remain undetected until later life.These entities usually manifest in adulthood with hemorrhage, headache, seizure, elevated venous pressure, mass effect, or cerebral ischemia due to steal phenomenon ,6. The mAt present, four-vessel cerebral angiography is the gold standard diagnostic procedure which can distinguish nongalenic AVFs from AVMs . AngiogrObliteration of the fistula through standard craniotomy, first described by Dandy in 1928, is still advocated by many surgeons. However, for deep seated lesions or fistulas in eloquent regions less invasive endovascular alternatives have been considered recently.In this patient, there are several hints suggesting the fistula to be originated from early childhood or even prenatal period. Although head injury is more associated with dural rather than nongalenic fistula, birth time head trauma may be proposed as a potential predisposing factor in this patient. Left-handedness can imply a potential damage to dominant hemisphere within the first stages of the patient's life, as regards the hypotheses advocating the role of early brain damage in pathological left hand domination .Calcification which is more probable in pial than dural AV lesions, should be considered too . The chaConsidering the calcified mass accompanied by significant midline shift in this case, even if the fistula had been obliterated by endovascular methods alleviating the mass effect would have been impossible. Indeed, the overall risk of endovascular occlusion of AVF in one stage and surgical removal of the calcified mass in another stage would outweigh the risk of a single operation for occluding the accessible feeder artery and removing the mass simultaneously. The calcified shell would physically restrict approach and handling of endovascular devices.Hence, in spite of concerns about the potential drawbacks of different therapies, the most appropriate options should be tailored for each patient considering particular situations.Various therapeutic options have been proposed for nongalenic AVFs. Although endovascular techniques have been shown to be less invasive, such massive calcified lesions which make significant mass effect or are unlikely to dwindle after transarterial occlusion are better managed surgically.Written informed consent was obtained from the patient and his parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.SAFT performed the microsurgery and gave the final approval for the version to be submitted.MZZ made contribution to conception and analyzed the patient data.ZH made contribution in collecting data and drafting the manuscript.ATM was contributor in revising the manuscript.MH was contributor in drafting the manuscript.


The nasal cavity harbors an enormous variety of neoplasms, including epithelial and mesenchymal tumors. Hemangioma is an infrequent mesenchymal tumor of the nasal cavity, mostly arising in the mucosa and rarely in the bones. We describe the case of a 73-year-old woman who was referred to our hospital with a tumor in her left nasal cavity. The tumor originated from the left inferior turbinate. Histological examination subsequent to complete excision revealed that the tumor was an intraosseous cavernous hemangioma. To our knowledge, this is the second case of intraosseous hemangioma of the inferior turbinate reported in the English literature. A wide variety of tumors occur in the nasal cavity. Hemangiomas account for about 20% of all benign neoplasms of the nasal cavity. Hemangioma of the nasal cavity occurs most commonly on the septum (65%), lateral wall 18%), and vestibule (16%) [8%, and vHemangiomas also occur as solitary lesions in bones. These tumors account for only 0.7% of all primary bone tumors . IntraosA 73-year-old woman was referred to our department with a 1-month history of left-sided nasal obstruction. She had no history of epistaxis or facial trauma. Anterior rhinoscopic examination revealed a mass obstructing the left nasal cavity. A hypertrophied inferior turbinate seemed to be occupying the left nasal cavity. The tumor was bony hard and covered with intact mucosa not hypervascularized mucosa . No otheUnder general anesthesia, the patient underwent surgical excision by the Caldwell-Luc procedure. The tumor, inferior turbinate, and medial wall of the maxillary sinus were resected en bloc. Intraoperative hemorrhage was 20 mL. The nasal cavity was packed with gauze. The packing was removed on the fifth postoperative day. The postoperative course was uneventful, and there was no evidence of recurrence at 8-month follow-up. Macroscopically, the tumor was 4 × 5 cm in size and covered with intact mucosa. Microscopically, the tumor composed of bony trabeculae and anastomosing vascular channels of cavernous size. The histological diagnosis was cavernous hemangioma . Hemangiomas are benign tumors originating in the vascular tissues of skin, mucosa, muscles, glands, and bones. Although head and neck lesions are common sites for hemangioma, hemangiomas of the nasal cavity are rare. The most common site for nasal hemangiomas is the nasal septum, followed by the lateral wall and vestibule . SeveralHemangiomas occur not only in soft tissues but also in bones. Intraosseous hemangiomas account for only 0.7% of all primary bone tumors. The most common sites in the head and neck are the skull (53%), mandible (10.7%), nasal bones (9%), and cervical vertebrae (6%). Intraosseous hemangiomas originating in the nasal cavity are extremely rare. Only one case of hemangioma within the turbinate bone has been reported in the English literature . The cause of intraosseous hemangioma is not well understood. Although many patients have a history of local trauma, a causal relationship remains doubtful . In our Diagnosis of intraosseous hemangioma is extremely difficult. It presents as a slowly enlarging, hard mass. It usually does not present signs that suggest a vascular lesion  . RadiogrBased on histopathological examination, hemangiomas can be subdivided into two types, that is, capillary and cavernous types. Although cavernous hemangiomas of the nasal cavity are uncommon, most intraosseous hemangiomas show a cavernous pattern , 10. Therapeutic approaches of intraosseous hemangioma include surgery, radiotherapy, sclerotherapy, and embolization , 11, 12.Although radiological diagnosis of intraosseous hemangioma has been established, clinical and computed tomographic evidence does not always lead to an exact diagnosis. Therefore, surgery should play a definite role in both diagnosis and treatment. In summary, we report a case of intraosseous hemangioma of the inferior turbinate. Intraosseous hemangiomas in unusual sites pose diagnostic difficulties. The possibility of intraosseous hemangioma must be considered when a bony mass is detected in the nasal cavity.


Gestational choriocarcinoma is a highly malignant tumor of trophoblastic cells with a propensity to metastasize to various sites including lungs, vagina, brain, liver, kidney, and gastrointestinal tract, in descending order of frequency. A 29-year-old Caucasian woman presented to the hospital as an emergency with abdominal pain, hypotension, nausea and vomiting at about 6th month of a twin delivery after in vitro fertilization. Our case probably represents the first case of metastatic choriocarcinoma developed after in vitro fertilization, whether associted with or not, which was cured successfully after chemotherapy. Gestational choriocarcinoma is a highly malignant tumor of trophoblastic cells with a propensity to metastasize to various sites including lungs, vagina, brain, liver, kidney, and gastrointestinal tract, in descending order of frequency. The ultimate cause of gestational trophoblastic disease is claimed to be genetic in origin. No environmental etiological factor has been implicated up to now apart from deficient vitamin A precursor carotene in diet . The firWe hereby report the third case diagnosed with spontaneous splenic rupture, a peculiar mode of presentation. Being metastatic at presentation, it is also the first case ever reported in the English literature. We also discuss possible etiopathogenic role of IVF with gestational trophoblastic disease.A 29-year-old Caucasian woman presented to the hospital as an emergency with abdominal pain, hypotension, nausea and vomiting at about 6th month of a twin delivery.Sixteen months previously she had been operated on due to vaginal bleeding. Pathologic examination at that time revealed necrotic chorion villi and chronic endometritis. Thirty-nine months previously she had been started on an in vito fertilization protocol due to the diagnosis of infertility. The regimen consisted of recombinat FSH , human menopausal gonadotropin , human chorionic gonadotropin , leuprolide acetate  and LHRH analog . She was continued on this treatment for two months and stopped when doctors told her husband had azospermia. One month later another 2 months hormonotherapy failed to generate oocytes. Seven months later she was again started on gestoden and ethynyestradiol  together with leuprolide acetate and recombinant FSH. After successful collection of oocytes in vitro fertilization was achieved and she became pregnant with twins. Pregnancy was supported with progestogen tablets up until 10th week of gestation. She had nausea and vomiting starting from the 4th week till 24th week of gestation. She delivered two healthy babies at term. Postpartum bleeding continued for two months and dilatation & curettage were performed due to placental retention and no abnormal pathology was noted at that time. Irregular menses occured thereafter. No abnormality was detected apart from a left sided ovarian cyst. Three months before presentation symptoms of left sided abdominal pain, nausea and vomiting started. Physical examination and laboratory tests were reported to be within normal limits. Proton pump inhibitors and antacids gave no relief.When she presented to the emergency room of the local hospital, rigidity and rebound tenderness were detected on abdominal examination. Abdominal ultrasonography revealed intraabdominal fluid and solid mass in the spleen. Splenectomy was performed with the diagnosis of spontaneous splenic rupture and she recovered uneventfully. The diagnosis of a local pathologist was diffuse large cell non-Hodgkin lymphoma (NHL). She was referred to the oncology clinic of our University Hospital.Paraffin blocks were obtained and transferred to our hematopathology department. Microscopic features were inconsistent with NHL. However, there was a necrotic and hemorrhagic tumor composed of pleomorphic cytokeratin positive epithelial cells with round nuclei and large cytoplasm. These cells were surrounded with cytokeratin(-) and β-HCG(+) sincyciotrophoblastic cells. The definitive pathologic diagnosis was metastatic choriocarcinoma.Computerized tomography (CT) scan of the abdomen and pelvis showed bilateral ovarian cysts, a 2 cm left sided renal lesion, and bilateral inguinal lymph nodes. CT scan of the chest revealed aortopulmonary lymph nodes less than 1 cm in diameter and minor parancymal nodules less than 5 mm in diameter. Cranial magnetic resonance imaging was normal. Plasma beta-human chorionic gonadotropin (β-HCG) level was found be 7991 mIU/mL . She was treated with 4 cycles of BEP regimen  starting from 8 months after antecedent pregnancy. Her β-HCG level dropped to 18,3 mIU/mL after the first cycle and <1 mIU/mL after the second cycle and remained low thereafter. She has no sign of recurrence for the last 25 months of follow-up.Gestational choriocarcinoma is the most malignant form of a group of tumors including complete and partial molar pregnancy, invasive mole, placental site trophoblastic tumor, and choriocarcinoma. They are collectively known as gestational trophoblastic disease. Although choriocarcinoma has a very high propensity to metastasize to various sites including brain it has also a very high cure rate. Nongestational choriocarcinoma also occurs and is usually resistant to therapy . The preEthnic origin, age, diet and oral contraceptives have been implicated as a risk factor for gestational trophoblastic disease (GTD). On the other hand, there are several case reports showing development of GTD after IVF. An attempt to explain the relationship between infertility, its treatment and GTD by Bates et al. did not find a potential association .Franceschi et al. reported that women who used fertility drugs were not at increased risk of developing ovarian cancers compared with women who did not . One yeaIn conclusion, our case probably represents the first case of metastatic choriocarcinoma developed after in vitro fertilization, whether associted with or not, which was cured successfully after chemotherapy.


In this case report, we describe the presentation and treatment of a patient with nasopharyngeal chondrolipoma. Lipomas are common soft tissue tumours, although their incidence in the nasopharynx is very low. A rarer variant of lipoma, chondrolipomas are benign mesenchymal tumours. They are formed by the proliferation of mature adipocytes and contain different amounts of mature cartilaginous tissue; Weiss “Enzinger and Weiss's soft tissue tumours”, 4th ed: Mosby, St Louis; 2001 This represents the second reported case of a nasopharyngeal chondrolipoma. An endonasal approach to excision has not been previously described. There are few reported cases of lipomas arising in the nasopharynx and only one reported case, of nasopharyngeal chondrolipoma . In the In this case, it was possible to excise the chondrolipoma via an endonasal endoscopic approach. A 70-year-old male was referred for ENT assessment with a twelve month history of increasing nasal congestion and globus sensation. There was no history of weight loss, epistaxis or loss of smell. He denied any otological symptoms. Nasoendoscopy demonstrated a mass in the postnasal space. It was not possible to pass the scope beyond the mass due to its size. Oropharyngeal examination revealed a large smooth mass extending beyond the soft palate. It was clearly visible with the soft palate retracted . A magnetic resonance scan of the head and neck confirmed the presence of a mass in the postnasal space arising from the right eustachian tube cushion. The scan signal intensities were consistent with a lipoma. The patient proceeded to examination under anaesthetic of the nasopharynx. Following nasal decongestion nasoendoscopy demonstrated the mass attached to the post nasal space by a narrow pedicle. Using a transnasal endoscopic approach the pedicle was dissected and divided, allowing the mass to be mobilized and removed via the mouth. It was submitted for histological examination. There was minimal bleeding and nasal packing was not required.The mass had a lobulated cream appearance with a smooth surface measuring 5 × 4 × 2 cm. The cut surface was cream coloured, gelatinous with a yellow outer rim . Microscopically, the mass was covered by a ciliated columnar epithelium showing focal squamous metaplasia. The stroma was composed of fibrous tissue exhibiting myxoid changes and mixed with mature fatty tissue and thin and thick walled blood vessels, some containing thrombi. Islands of cartilaginous tissue were present There were scattered mast cells and perivascular cuffing by lymphocytes and plasma cells. The appearances were concluded to be consistent with a chondrolipoma .There are a variety of clinical manifestations associated with a nasopharyngeal mass. In this case, the patient presented with nasal obstruction and a globus sensation. Other symptoms may include: anosmia, halitosis, rhinorrhoea, post nasal drip, nasal obstruction, epistaxis, snoring, obstructive sleep apnoea, dysphagia, cervical lymph node enlargement, palatal mass, aural fullness, otalgia, hearing loss, and voice change . Lipomas are one of the most common mesenchymal tumours and 13 percent of lipomas occur in the head and neck region . The nasLipomas are benign tumours consisting of mature fat cells and are single or lobulated. They may be soft, firm, or cystic and may arise in any location where fat is normally present. The growth of lipomas is due to proliferation of cells similar to fibroblasts. Lipomas may contain a variety of mesenchymal elements. These may include fibrous connective tissue (fibrolipoma), myxomatous (myxolipoma), capillary angiomatous tissue (angiolipoma), and mucoid substances (myxoid lipoma) , 3.There has been only one reported case of nasopharyngeal chondrolipoma . HistoloThe described nasopharyngeal lipomas arise from adipose tissue in the submucosa and are attached by a sessile stalk or pedicle. In the present case, the pedicle was divided under nasoendoscopic guidance.The investigation of a patient with these symptoms would include a full ENT history and examination including nasoendoscopy. A CT scan may provide a definitive diagnosis by calculating the density of the suspected mass. The CT attenuation number is low due to fat being of lower density than water. MRI scans may also accurately diagnose lipomas preoperatively because of lipomas typical signal intensity patterns .Treatment of nasopharyngeal lipomas is surgical with curative intent. Chondrolipomas are large and well demarcated facilitating their excision. We report a case of a nasopharyngeal chondrolipoma treated by nasoendoscopic dissection and removal. Although rare, it should be considered in patients presenting with symptoms of a nasopharyngeal mass. Cartilaginous metaplasia giving rise to chondrolipomas is found in large, long-standing lipomas. The treatment of these tumours is by surgical excision, following which there have been no reported cases of recurrence.


The occurrence of intussusception in adults is rare. The condition is found in 1 in 1300 abdominal operations and 1 in 100 patients operated for intestinal obstruction. The child to adult ratio is 20:1.A 52-year-old Irish Caucasian woman was investigated for a 3-month history of intermittent episodes of colicky midgut pain and associated constipation. Ileocolonoscopy revealed a pedunculated lesion in the terminal ileum prolapsing into the caecum. Computed tomography confirmed a smooth-walled, nonobstructing, low density intramural lesion in the terminal ileum with secondary intussusception. A laparoscopic small bowel resection was performed. Histology revealed a large pedunculated polypoidal mass measuring 4 × 2.5 × 2 cm consistent with a submucosal lipoma. She had complete resolution of her symptoms and remained well at 12-month follow-up.This case highlights an unusual cause of incomplete small bowel obstruction successfully treated through interdisciplinary cooperation. Ileal lipomas are not typically amenable to endoscopic removal and require resection. This can be successfully achieved via a laparoscopic approach with early restoration of premorbid functioning. Neoplasms of the small intestines are rare . GastroiWe report a case of terminal ileal lipoma causing intermittent intussusception in a 52-year-old woman. The lipoma was diagnosed at ileocolonoscopy and successfully removed through laparoscopy. A review of the literature on small bowel intussception and gastrointestinal (GI) lipomas is also presented in this report.A 52-year-old Irish Caucasian woman presented with a three-month history of intermittent central abdominal pain and constipation. She did not describe gastrointestinal bleeding or weight loss. She previously underwent a transabdominal hysterectomy for menorrhagia. Her physical examination was unremarkable. Initial investigations, such as blood tests, abdomen ultrasound and gastroscopy were unremarkable. Ileocolonoscopy revealed a pedunculated terminal ileal lesion prolapsing into her caecum. Computed tomography (CT) of her abdomen and pelvis demonstrated a smooth-walled, low-density, intramural lesion in the terminal ileum. It measured 3.2 × 1.6 cm. The ileum at the proximal end of the lesion was mildly dilated with a centrally placed narrowed channel of contrast, which was consistent with an intussusception possibly secondary to an intramural lipoma. There was no evidence of obstruction (Figure th postoperative day. She remained free of symptoms at three-month follow-up.She underwent an elective laparoscopic small bowel resection and stapled functional end-to-end anastomoses. On macroscopy the lesion appeared as a large pedunculated polypoid mass measuring 4 × 2.5 × 2 cm with focal mucosal ulceration (Figure Lipomas are benign tumors of mesenchymal origin. They are the second most common benign tumors in the small intestine and account for 10% of all benign gastrointestinal tumors and 5% of all gastrointestinal tumors. They are predominantly submucosal and protrude into the lumen . OccasioIntussusception in adults is a rare entity that it is generally caused by definable intraluminal pathology . DiagnosLipomas can be diagnosed through conventional endoscopy, capsule endoscopy, barium studies and, most importantly, CT. Typical endoscopic features are smooth, yellowish surface with pedunculated or sessile base, as seen in this case. Other endoscopic characteristics are the "cushion sign" and "naked fat sign" . CT usuaThe treatment for lipomas depends on the clinical manifestations. Indications for their removal include intestinal obstruction, hemorrhage and malignant potential . There iIn this case, we illustrate the importance of a thorough interdisciplinary evaluation of patients with midgut abdominal pain. It highlights the diagnostic values of CT scanning and completed ileocolonoscopy. Despite preoperative localization, laparoscopy facilitates a thorough evaluation of the intraperitoneal contents and therapeutic resection of the affected segment. This report confirms the recognized benefits of laparoscopic surgery with associated early return to premorbid functioning. In patients with persistent episodes of incomplete intestinal obstruction, atypical causes, such as the etiology we describe here, should be considered.Written informed consent was obtained from our patient for publication of this case report and any accompanying images.The authors declare that they have no competing interests.NOA contributed in collecting the requisite literature and wrote the case report. DOK also collected the requisite literature and reviewed the literature. DCM also contributed in collecting the requisite literature. BR and PCN were involved in the diagnosis of our patient. PCN also performed the surgery. All authors read and approved the final manuscript.


Primary mantle cell lymphoma of the esophagus is quite rare, and we report here a case of a submucosal polypoid tumor of the esophagus that was pathologically similar, but not identical, to mantle cell lymphoma.A 66-year-old man underwent surgery in our hospital for a submucosal tumor of the esophagus. Histopathologically, the submucosal tumor uniformly showed a vague, nodular pattern composed of a regular proliferation of CD3-CD10-CD20+CD79a+bcl2+ small lymphoid cells with islands of abortive CD10+Ki67+ germinal centers, without evidence of marginal zone formation or lymphoepithelial lesions. These features were consistent with mantle cell lymphoma. However, the proliferating cells weakly expressed the D-type cyclins, IgD and IgM. The tumor was diagnosed as a “mantle cell lymphoma-like tumor”. Postoperative examination confirmed no tumour involvement in other organs. The patient was treated with rituximab postoperatively and has been disease free for more than 28 months after surgery.The present case might be regarded as a “hyperplastic change of the mantle zone” without the molecular features of mantle cell lymphoma, a case that adds to the limited clinicopathological data on B-lymphoproliferative diseases of the esophagus. The esophagus is the least common site of involvement of lymphoma in the digestive tract, accounting for less than 1% of lymphoma patients. Primary esophageal lymphomas are usually large B-cell types or low-grade B-cell mucosa-associated lymphoid tissue (MALT) . Here weA 66 year-old Japanese male underwent upper gastrointestinal endoscopy by his practitioner for complaints of dysphagia, which revealed an esophageal submucosal tumor. He was referred to the National Hospital Organization Mito Medical Center in 2006 for treatment. He did not complain of fever, weight loss, or night sweating. On admission, physical examination revealed no remarkable features; the liver, spleen, and superficial nodes were not palpable. His laboratory data showed no abnormalities except a 10-year history of abnormal glucose tolerance treated by his primary physician. No elevated tumor markers or autoimmune antibodies were observed. A barium esophagogram, CT, and upper GI endoscope showed an esophageal submucosal polypoid lesion at the cervical esophagus . Its locin situ hybridization. In brief, DNA was hybridized with Vysis® LSI® IGH/CCND1 Rearrangement probes , as described previously [The tumor macroscopically showed a submucosal polypoid appearance . Resecteeviously . The IgHThe patient experienced a good post-operative course, and was enrolled in the Department of Hematology. Lymphoma metastasis was evaluated by a whole body CT scan, PET-CT, bone marrow examination, Ga-scintigram, and total colonoscopy, but no other sites were involved. The diagnosis was stage IAE, and rituximab was given as adjuvant chemotherapy. This patient has been disease free for more than 28 months after surgery.Primary esophageal lymphoma is rare, with no MCL reported except a case in cardiac-type glandular tissue replacement of the lower esophagus ,2,5-7. TMALT lymphoma can arise in the esophagus. Hosaka and colleagues showed that three cases previously reported as a B-cell type non-Hodgkin's lymphoma might be MALT lymphomas . The difth edition, cyclin-D1-negative MCL should overexpress cyclin D2 or D3, as confirmed by gene expression profiles [Although this case resembled MCL, it lacked the immunostaining and molecular features typical for MCL, including weak expression of cyclins D1, D2, D3, CD5, IgD, or IgM. According to the WHO classification 4profiles . In addiprofiles . The curTaken together, the current case has similar histological features to MCL, but does not fit the MCL diagnosis. The present case may be “hyperplasia of the mantle zone without neoplastic transformation,” but this conclusion would require careful follow-up. We also need further clinicopathological data regarding B-cell lymphomas of the esophagus.


Adrenal myelolipomas are relatively rare, non-functioning benign tumours composed of mature fatty and active hematopoietic elements. They can be asymptomatic, even if their size is massive. Diagnosis is relatively simple using ultrasound, computed tomography and magnetic resonance imaging. Surgical resection through an extraperitoneal approach is advocated in cases of symptomatic or large myelolipomas exceeding 5-cm in diameter. Their low incidence seems to be increasing from 0.2% to 10% during the last decade.We present a case of a giant adrenal myelolipoma in a 68-year-old Caucasian male, who was presented with left lumbar pain. Renal ultrasound, CT and MRI demonstrated a well demarcated mass, with a maximum diameter of 10-cm. The differential diagnosis comprised the adrenal myelolipoma, the retroperitoneal liposarcoma and the renal angiomyolipoma. Thus, the patient was subjected to a left adrenalectomy.Multiple theories have been proposed for the increasing frequency and natural course of the adrenal myelolipoma, with chronic adrenal stimulation and the contemporary stressful lifestyle to be the most appealing. Surgical treatment is advocated through an extraperitoneal approach because of the quicker recovery of the patient and the smaller postoperative complication rate. Adrenal myelolipoma is an uncommon urological entity, which has an increasing incidence. Despite its benign biology, when presented as a giant tumor, the urologist can face diagnostic dilemmas or even an acute clinical setting. We present a case of an asymptomatic giant adrenal myelolipoma, one of the largest reported in the literature and discuss issues concerning the diagnosis, the management and the proposed theories for the increased prevalence of this lesion.A 68-year-old Caucasian male, with history of untreated left nephrolithiasis, was presented to our hospital, complaining of left lumbar pain, typical of a left renal colic. During the kidney ultrasound, a large, well demarcated mass, with a maximum diameter of 10-cm was demonstrated, in the upper pole of the right kidney, mimicking a renal angiomyolipoma. Contrast-enhanced computed tomography (CT) was performed the next day, verifying a heterogeneous mass with predominant fatty components in the right retroperitoneal space, measuring approximately 12-cm in diameter, slightly compressing the right kidney and vena cava . MagnetiSurgery was performed through a right subcostal incision for the extraperitoneal approach of the right adrenal gland. The mass was totally dissected from the upper pole of the right kidney and excised en-block with the right adrenal gland. The macroscopic examination of the mass revealed a giant mass measuring 19 × 12 × 2 cm, surrounded by a thin capsule. Yellow adipose tissue was unevenly scattered on the capsule with varying amounts of red hematopoietic tissue . The patThe patient had an uneventful postoperative course and was discharged on postoperative day 4.Adrenal myelolipomas are also called incidentalomas, since their diagnosis is based on autopsy or imaging techniques performed for reasons unrelated to adrenal diseases. They are relatively rare, non-functioning benign tumors composed of mature fatty and active hematopoietic elements. Their incidence ranges from 0.01% to 0.2% and less than 300 cases were reported until 2000 [Multiple theories have been proposed for the aetiology and natural course of the adrenal myelolipoma -5. The mDiagnosis of adrenal myelolipomas can be unproblematic with the use of ultrasound, contrast-enhanced CT, MRI or positron emission tomography (PET). We believe that fine-needle aspiration bears the risk of rupture, especially in cases of giant lesions and spillage when a malignant neoplasia is encountered. No endocrinological evaluation is needed, since myelolipomas are non-functioning tumours.The management of adrenal myelolipomas should be individualized. A small, less than 5-cm, asymptomatic myelolipoma could be followed-up over a 1-2 year period with imaging controls. On the contrary, a symptomatic lesion or a large >5-cm myelolipoma should be surgically excised, since there are reports of spontaneous rupture and haemorrhage of the mass presented with life-threatening cardiovascular shock . We prefThe adrenal myelolipoma is a rare urological entity, which seems to increase its frequency, probably due to causes affecting primarily the function and physiology of the adrenals. Our case described one of the largest ever reported myelolipomas, which despite its size, can be rather asymptomatic. However, even with the contemporary imaging modalities, precise diagnosis is difficult. Thus, we advocate open surgical management, since a laparoscopic approach might be extremely complicated in cases of such large tumours.


The chance of an intrathyroidal occurrence of a parathyroid gland is about 1–3%. Among the causes of hyperparathyroidism, parathyroid cases occur in less than 1% of patients. Here we present the case of a 63 year old Saudi female suffering from an intrathryroidal parathyroid carcinoma. The suspicion coming from the clinical investigations that the removed tumor tissue may be a parathyroid carcinoma could be confirmed by histology. Additionally non-radioactive in situ hybridization to localize mRNA transcripts for Cyclin D1 and immunohistochemical localization of Cyclin D1 was performed. Although parathyroid adenoma and carcinoma have disparate natural history, it can be difficult to differentiate between the two entities. Clinical presentation, operative findings may raise suspicion, but may not be conclusive especially if there is no evidence of invasion or metastasis, especially if the gland was intrathyroidal. Parathyroid adenomas account for 85% of primary hyperparathyroidism . On the A 63-year-old Saudi female, who was known to have hypertension and hyperlipidaemia, complained of fatigue, bone and muscle pain of several years of duration. She also had a history of recurrent nephrolithiasis. Her symptoms aggravated recently with several attacks of abdominal pain, nausea and vomiting. Neck examination revealed a left lower cervical mass, consistent with thyroid enlargement. Ultrasonography showed an enlarged left lobe with multiple hypoechoid and isoechoid nodules. One of them in the anterior-inferior aspect of the left lobe displayed a rim of calcification. Thyroid function test were within the normal range. Parathyroid imaging scan using 99m Tc-SestaMIBI-99c-Pertechnetate was consistent with scintigraphic features of a parathyroid adenoma Figure  in the rThe patient was admitted and loaded with intravenous saline followed by Furosemide (10 mg) the day before surgery, which was able to bring the serum calcium to 3.59 on the day of surgery. The patient was taken to the operating room where a video-assisted exploration of the left neck was attempted. The surgeon's plan was to remove the diseased parathyroid gland and to take out the enlarged left hemithyroid, followed by frozen section evaluation. Total thyroidectomy is only performed in our clinic, if frozen sections yield evidence of a differentiated thyroid carcinoma greater than one centimeter in diameter. A normal lower left parathyroid gland was identified in the left thymic horn and preserved. The left thyroid lobe appeared enlarged and showed only one distinct nodule near the upper pole, which was sent for frozen section diagnosis. It turned out to be benign thyroid tissue. The surgeon decided then to remove the left thyroid lobe after full neck exploration including the superior mediastinum that revealed four benign central neck lymph nodes.The pathology report described a thyroid lobe with a large tumor mass, measuring 6 × 5 cm and weighing 8.5 g. It appeared poorly circumscribed and of hard consistence. The mass showed a gray-whitish color with occasional foci of necrosis. A firm fibrous capsule surrounded it. The tumor occupied nearly the whole thyroid gland. Histology showed that thick, sharply outlined bands of collagenous tissue extended from the thickened capsule into the interior and completely divided the tumor into irregular lobules. The tumor cells were arranged in diffuse masses, solid sheets or closely packed nests of cells Figure . The tum. The sequences of the antisense oligonucleotides for Cyclin D1 was as follows: 5'gtt cct cgc aga cct cca gca tcc ag3' Acc.Nr: NM_053056 (NCBI). Immunohistochemical staining was performed with monoclonal anti-Cyclin D1 anti-body , at dilution of 1:100, using a standard avidin/biotin complex (ABC) method. Breast cancer cases known to express Cyclin D1 served as positive control for Cyclin D1. For negative control, non-immune serum was substituted for primary antibody.Clarification of the molecular pathogenesis of parathyroid carcinoma can aid in diagnostically difficult cases and may provide important clues for a more effective therapy. Therefore, tumor samples  were studied using in situ hybridization and immunocytochemistry with monoclonal antibodies. Formalin-fixed (3.7%), paraffin-embedded samples were used to localize mRNA transcripts for Cyclin D1. In situ hybridisation was performed as previously described. The sequences of the oligonucleotides were designed using the program Primer3 Using in situ hybridization we found a weak to distinct signal for CD1 in the cytoplasm of glandular cells of the parathyroid adenoma tissue Figure . No staiAt the postoperative patient visit, a week after surgery, the serum calcium has dropped to 2.38 mg/dl (2.10–2.55), the parathormone (PTH) level has returned to normal values, however alkaline phosphatase remained high (628 U/L). CT scan of the brain and lungs revealed no evidence of metastasis. At follow-up three months after surgery, the patient appeared disease free with normal levels of PTH and calcaemia and improvement of the bone disease symptoms. The patient is now, two years from surgery, disease free and her serum calcium and parathormone level remained in the normal range.Parathyroid carcinoma is a rare malignancy with an incidence of less than 1% in all patients surgically treated for primary hypoparathyroidism -12. A hiThe diagnosis of malignancy was made post operatively. The fine needle aspiration performed intra operatively was done to evaluate the left thyroid lobe lesion rather and revealed normal thyroid tissue. None of the preoperative studies raised suspicion of the presence of parathyroid malignancy and the probability to find a parathyroid carcinoma in patients with palpable parathyroid glands, marked elevation of serum calcium and parathormone ,15 is leThe suspicion that the removed tumor tissue may be a parathyroid carcinoma could be confirmed by histology. In our case, invasion of tumor cells was found in the vessels and neighboring muscle tissue, which has been seen also in other studies ,10. CellA change in the expression of CD1 mRNA has been found during the progression from benign to malignant parathyroid tumor cells CD1 oncoprotein is overexpressed frequently in parathyroid carcinoma -7. It waIn summary, parathyroid carcinoma is a rare malignancy. Signs and symptoms of hypocalcaemia, hyperparathyroid bone disease and features of renal involvement along with a palpable neck mass are strong predictors of parathyroid malignancy. It is usually difficult to recognize the tumor preoperatively and it is not conclusively identified intraoperatively. Pathologic histology including in situ hybridization and immunohistochemistry of cyclin D1 helps to characterize the lesion and confirms its nature.The patient has given her consent for the case report to be published.The authors declare that they have no competing interests.LT, and FS drafted the manuscript. WIH and OAS compiled the clinical data. LT and HEK confirmed the clinical diagnosis by histopathology. FS evaluated the immunohistochemical stainings and non radioactive in situ hybridization. All authors read and approved the final manuscript


A middle age lady presented with abdominal pain was diagnosed to have multiple peritoneal and hepatic hydatid cysts on CT scan. Retrospectively she was found to have suffered blunt abdominal trauma. A forty five year old lady presented with chronic dull aching pain with gradually increasing lump in the epigastric and pelvic region since 3 years. A well defined, firm mass of about 20 × 20 centimeters was palpable in epigastrium extending more on right hypochondrium, probably arising from left lobe of liver. A similar mass was palpable in the hypogastrium while multiple small nodular masses palpable in the right iliac fossa and lumbar region. Detailed inquest revealed a 5 year old incidence of blunt abdominal trauma, while working in her farm. The ultrasonography and CT scan  were given with the interval of 1 month. The follow up after 1 month did not show any decrease in the size of the cysts and decision of laparotomy was taken. Peritoneal cavity was crammed with cysts ranging from 1 to 12 centimeters , E. multilocularis, or E. vogeli . This diPeritoneal hydatidosis is almost always secondary to hepatic disease, although some unusual cases of primary peritoneal hydatidosis have been described. The overall frequency of peritoneal disease in cases of abdominal echinococcus is approximately 13%. Peritoneal involvement is usually undetected unless cysts are large enough to cause symptoms. Most of the cases of peritoneal hydatid disease are secondary to previous surgery for liver hydatidosis. In present case blunt abdominal injury was the probable cause of dissemination. Systemic anaphylaxis is usually associated with cyst rupture and can be predicted by positivity of Casoni reaction.USG is the first line of screening for abdominal hydatidosis. USG is particularly useful for detection of cystic membrane, septa, and to look for hydatid sand. CT scan best demonstrates cyst wall calcification and cyst infection . ImmunoeThe treatment of choice for localized hydatid cysts in liver or lungs is principally surgical while the therapy for disseminated peritoneal hydatidosis remains medical . TherapyAnaphylctic shock occurs when there is prior sensitization. The present case might have been sensitized at the time of rupture of hepatic cyst when she sustained blunt abdominal trauma. Utmost care of preventing the contact of hydatid cyst contents with the body tissues during operation can not avoid the anaphylactic shock. The surgeon should be ready for this catastrophe while operating hydatid cyst especially with the suspicion of prior sensitization.A written consent was taken from the patient and her father regarding the publishing of this article.The authors declare that they have no competing interests.All the authors read and approved the final manuscript. NJS did the surgery, RKS and NV assisted the surgery. PNM is the corresponding author of the article who drafted and finalized the manuscript.


Gallbladder perforation is a rare complication of cholecystitis and cholelithiasis. The high morbidity and mortality rates associated with this condition are due to delays in diagnosis and treatment since signs and symptoms of perforation do not differ significantly from those of uncomplicated cholecystitis. We report on a patient who was affected by Child-Pugh A alcoholic liver cirrhosis and who developed an acute gallbladder perforation with spillage of stones into the peritoneal cavity and give a review of the current literature. Asymptomatic cholelithiasis is a frequent condition which affects up to 10% of the adult population in wealthy nations. Acute cholecystitis develops in up to 2% of patients affected by asymptomatic cholelithiasis. Gallbladder perforation occurs in 2 to 11% of acute cholecystitis cases. Due to the high mortality that can be caused by a delay in the correct diagnosis and following adequate surgical treatment, gallbladder perforation represents a special diagnostic and surgical challenge [According to Niemeier 1934), perforations are classified into three categories: type I includes patients with free perforation into the peritoneal cavity, type II describes patients with localized perforation and type III patients with cholecysto-enteric fistulas. Less frequent forms include cholecystobiliary fistula and more complex fistula formations . Cases o4, perforA 49-year-old man with liver cirrhosis and a history of esophagial varices presented to a clinic with upper abdominal pain. He described colicky pain radiating to the back. He denied nausea, vomiting, diarrhea or obstipation. There was no history of gallbladder disease, no prior episode of abdominal discomfort, no medication - especially no NSAIDs - and no history of trauma. A distended abdomen with normal bowel sounds, tenderness in the right upper quadrant and signs of beginning peritoneal irritation were present. The laboratory studies showed a slightly elevated white cell count (12 G/L). All other findings were within the normal limits, including lipase and amylase, bilirubin, liver enzymes and coagulation parameters. Sonography revealed no abnormalities but failed to visualize the gallbladder. Gastroscopy confirmed the presence of type I esophageal varices. No signs of gastritis and no ulcers were reported. Computed tomography of the abdomen revealed several calcified stones in a thick-walled gallbladder and a tumorous mass of the liver. Considering the patient's history of alcoholic liver cirrhosis this was thought to be a hepatocellular carcinoma. The patient was then referred to our surgical department for further evaluation.On admission he had no elevated temperature (35.9°C), was hypotensive (80/40 mmHg) and tachycardic (120-140 beats/minute). He complained of upper abdominal pain persisting for about twenty-four hours. He had been treated with analgesics in the other clinic but with no relief. The physical examination confirmed tenderness of the right upper quadrant with initial signs of peritoneal irritation. At this point the laboratory studies revealed a significantly elevated white cell count (25 G/L) but once again no other abnormalities. The urine analysis showed elevated urobilinogen levels (2.0 mg/L). Sonography was repeated and it revealed a 7 × 6 cm conglomerate tumor of the gallbladder suspected of being an empyema, blood or a gallbladder carcinoma. Ascites was noticed around the liver . The diagnosis of a perforation was obvious since the gallstones were now found outside the gallbladder Fig.  and 3.The patient received parenteral fluids, analgesics and antibiotics. Two hours later he was taken to the operating room for open cholecystectomy. A large quantity of blood and stones Fig.  as well Perforation can develop early in the course of acute cholecystitis (one or two days) or it may even occur several weeks after onset. The most common site of perforation is the fundus, presumably because of its poor blood supply . If theSince there are no classical symptoms and signs of perforation diagnosis is challenging. Right upper quadrant pain, palpable right upper quadrant tenderness or high fever may indicate an acute onset. On the other hand patients may also show weakness, malaise and a palpable right upper quadrant mass, mimicking a malignacy. As most of these features are also present in acute cholecystitis, it is difficult to discriminate clinically between patients with perforated gallbladder and those with uncomplicated acute cholecystitis. A sudden decrease in pain intensity caused by the relief of high intracholecystic pressure might herald the perforation according to Chen et al. . Gore etAlso the sonographic appearances of gallbladder perforation are diverse and nonspecific. They include wall thickening (>3 mm), distension (largest diameter >3.5-4.0 cm), gallstones, coarse intracholecystic echogenic debris and bile duct dilatation. Distention of the gallbladder and edema of its wall may be the earliest detectable signs of imminent perforation. The 'hole sign'  is the most specific finding . An intrAlthough ultrasound remains the preferred initial examination for evaluation of suspected gallbladder perforation, unfortunately it often fails to demonstrate the perforation because of increased intestinal gas and pain. In the current case the blood in and around the gallbladder led to a misinterpretation of the sonographic image. In contrast, CT imaging is the most sensitive tool to diagnose gallbladder perforation ,8. CT scEarly diagnosis and surgical intervention are the key factors to decrease mortality and morbidity in the management of acute cholecystitis with gallbladder perforation. Both have significantly improved over the last few decades. This is partly due to shifting treatment paradigms in recent years with a larger number of cholecystectomies being performed for symptomatic cholelithiasis compared to the past but also the result of better diagnostic possibilities through the use of CT scans.Despite this development, the management of cirrhotic patients with gallbladder perforation - as in this case - remains a greater challenge. Edema of the gallbladder wall, leukopenia caused by hypersplenism and the presence of ascites that predispose to spontaneous bacterial peritonitis make the diagnosis of gallbladder perforation more difficult than in the general population . In addiIn conclusion gallbladder perforation is a rare but very serious condition and should be diagnosed and treated as soon as possible to decrease morbidity and mortality. The most important diagnostic tool is an early CT scan, followed by cholecystectomy on an emergency basis.The authors declare that they have no competing interests.CC  and MS  examined the patient in the ER, SW (radiology attending) diagnosed gallbladder perforation. CC and MS operated on the patient and took the photographs. All authors read and approved the final manuscript.