Superficial leiomyosarcoma is an exceedingly uncommon malignant tumor which could be located either to cutaneous or subcutaneous tissues. Increased mass size and depth, advanced tumor staging and inadequate surgical excision are the main prognostic factors for poor result.We report a rare case of a 71-year-old man with an extensive exophytic lesion (12 × 10 cm) in the anterior-medial side of the proximal right tibia. The lesion was painless and consistently neglected by the patient until a skin trauma caused ulceration of the affected area. Magnetic Resonance Imaging revealed a soft-tissue mass which was well defined from the surrounding bone and muscles. As initial biopsy in another hospital hadn't clarified the true nature of the lesion, new samples were taken and the diagnosis of leiomyosarcoma was established. Laboratory examination showed no distant metastasis and wide excision of the neoplasm was decided. After tumor resection, the remaining soft tissue and skin defect was covered with a gastrocnemius myocutaneous flap. The postoperative period was uneventful and wound healing was followed by local radiotherapy and systemic chemotherapy. At 3 years follow up, no recurrence or metastasis was identified and the patient was able to walk and stand without impairment of his ambulatory status.Proper surgical management of soft tissue leiomyosarcoma continues to remain the cornerstone of treatment efficacy and the most important prognostic factor for patients' survival. Reconstruction of the remaining soft tissue defect should be always performed at the same operative time when removal of giant size tumors leaves an uncovered cavity with an inadequate sleeve of muscular and skin tissues. Superficial Leiomyosarcoma (LMS) is a quite uncommon tumor which is mainly developed in the proximal portion of the lower limbs -3. The lSubcutaneous soft tissue Leiomyosarcomas (LMSs) are apparently larger and vascular in their origin, grow faster and they may produce similar changes in the skin. Their incidence is even rarer accounting for 2.3 – 5.3% of all malignant soft tissue tumors ,5 and 4 We report a rare case of a giant subcutaneous LMS of the proximal tibia. Wide excision of the tumour in combination with local RTP and systemic CTP led to good functional outcome and no recurrence until the latest follow up (3 yrs postoperatively).A 71-year-old man, without medical history, referred to Tumor Outpatient Clinic of the Hospital due to an extensive exophytic mass of the anterior-medial side of the right proximal tibia. The lesion was appeared ulcerated and bloody with irregular areas of necrosis and haemorrhage and its size was 12 × 10 cm with collar of around 10 cm. Figure . The preen block wide excision of the mass was performed. As the tumor was very close to the bone, a portion of the anterior-medial cortex of the proximal metaphysis of the tibia was also removed to facilitate at least 2 cm resection margins circumferentially  clinical and laboratory examination  didn't reveal any signs of tumor recurrence or metastasis. The patient could stand and walk without any restriction and he was able to perform his usual daily activities.The distinction between cutaneous and subcutaneous LMS is of primary importance for prognostic reasons ,8,10. AlIn the current patient, the size of the tumor 12 × 10 cm) was higher than the previously reported cases. Specifically, the mean size reported in literature for superficial LMSs is about 5 cm, with a wide range from 0.5 to 19 cm  × 10 cm ,8,19. SoWide excision of the lesion is of outmost importance as it is strongly correlated with disease-free survival ,10,12-15Many authors have been supported the application of postoperative RTP and CTP in high grade LMSs ,10,12-15Even though delayed diagnosis and giant size of soft tissue leioymyosarcomas may adversely affect the final result, successful recovery could be anticipated. Special focus should be given to careful and meticulous wide excision of the mass as the role of chemotherapy and radiotherapy is still blur and unknown.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 journalThe authors declare that they have no competing interests.GM, the chief surgeon in charge of the case prepared, participated in the design of the paper and in the drafting of the manuscript. MA, FM, BR participated equally in the design of the paper and in the drafting of the manuscript. RR participated in the study of macroscopic and microscopic features of the lesion. NM participated in the study of the radiological data. BEC revised critically the final version of the manuscript. All authors read and approved the final manuscript.


The purpose of our study was to determine the prevalence and significance of psammoma bodies (PBs) in the cervicovaginal smears of the screening population of Trento district , with the description of the cytological presentation of an asymptomatic bilateral ovarian psammocarcinoma.st, 2007. Clinical information was obtained from retrospective review of women's medical records. The source of PBs was identified with adequate diagnostic procedures.From 1993 to 2006, women with PBs detected on consecutively screened cervical smears were identified from the computerized pathology database of Rovereto Hospital. The follow-up period was set from the time of cytological diagnosis to May 31PBs were found in six of the 201,231 Papanicolaou screening smears (0.0029%). Benign conditions  were found in four patients. In two cases, PBs were associated with malignant cells; a bilateral ovarian malignancy was diagnosed in both cases, a serous adenocarcinoma and a psammocarcinoma.PBs in the cervicovaginal smears are a rare finding, associated more often with benign conditions than with malignancies. Moreover, to our knowledge, our case of primary ovarian psammocarcinoma is the first report in which the presence of malignant cells and PBs in the cervicovaginal and endometrial smears represents the first manifestation of disease. The presence of psammoma bodies (PBs) in cervicovaginal smears (CVS) is a rare finding, the prevalence ranging from 0.00047% to 0.057% in large series studies -6. PBs ost, 2007. Adequate diagnostic procedures  were performed in order to identify the source of PBs.We identified from the computerized pathology database of Rovereto Hospital all cases describing the presence of PBs in CVS, with a retrospective review of 201,231 screening cytological exams from 1993 to 2006. The cervical cancer screening program was performed according to Walton report  and cytoIn the retrospective review of 201,231 screening program cytology reports we identified 6 Papanicolaou smears with PBs (0.0029%).Four of them belonged to premenopausal women and PBs were found in absence of atypical cells Fig . EndometTwo of these patients  had an IUD. Because of a possible coexistence of a gynaecologic malignancy the surgeons performed a laparoscopy with removal of the IUD. The abdominal and pelvic organs, including the ovaries, revealed no abnormalities.The third patient  showed multiple, small cysts in the right ovary. A laparoscopic resection was performed and histological examination revealed benign inclusion ovarian cysts with intraparietal PBs.In the fourth patient , a pelvic ultrasonography showed a cystic structure, 1.5 cm in diameter, in the right ovary. For not specified circumstances, an abdominal hysterectomy with bilateral salpingo-oophorectomy and appendicectomy was performed. Histopathologic examination revealed a serous cystoadenofibroma with PBs of the right ovary Fig . The lefPapanicolau smears were repeated three to six months after the initial identification of PBs in the four patients, and none of them showed persistent PBs in the smears, even after a follow up period of 3–8 years.The last two patients with PBs on the smears had a bilateral ovarian malignancy with a diffuse involvement of the abdominal cavity. Rare blue-stained, laminated, calcified bodies, identified as PBs were found in association with diagnostic malignant cells, which were variable in size and shape and had large nuclei with prominent nucleoli Fig . Both enIn one patient  a preoperative vaginal ultrasonography revealed a dishomogeneous uterine corpus, 7,3 × 5,4 cm in size, with multiple nodular lesions referable to leiomyomas located in the anterior wall of the myometrium. Intraoperatory findings showed a right ovarian mass, white and firm, 4 cm in diameter and another nodular lesion on the surface of the left ovary, grey-white, well circumscribed and 2 cm in diameter. The invasive thickness of the ovarian stroma was 1,2 cm on the cut surface. The omentum and peritoneal surface were involved by multiple tumor nodules (2 cm in maximum diameter). Intraoperative histological examination was consistent with the diagnosis of bilateral moderately differentiated ovarian serous adenocarcinoma. The final pathologic report confirmed the diagnosis made on frozen sections. Additonal feature was the presence of multiple psammomatous calcifications Fig . NeoplasIn the other patient  exploratory laparotomy showed a diffuse white-grey nodularity on the surface of both ovaries. Two omental and three peritoneal nodules (2 cm in maximum diameter) were found. Intraoperative histological examination was consistent with the diagnosis of bilateral ovarian serous carcinoma with extensive psammoma bodies formation, showing the typical proliferative pattern of PC, according to the diagnostic criteria defined by Gilks , and infThe prevalence of PBs in our series of consecutively screened Papanicolau smears is 6 per 201,231 (0.0029%), which confirms that this is a very unusual finding, in agreement with other large series of CVS previously reported in the literature and summarized in Table The observation of PBs on CVS should not be ignored and should alert the clinician in order to evaluate the possibility of a coexistent carcinoma, even though a review of the literature reveals that in more than 50% of patients PBs are associated with benign conditions -6. All bWhen PBs in the CVS are associated with malignant or borderline tumors, the ovary is the most common primary site (51%), serous cystoadenocarcinoma being the most common neoplasm, followed by the uterine corpus (30%) . In our In the first description by Gilks et al. , eight cPrimary peritoneal PC is a very unusual variant of extraovarian neoplasms that histologically resemble surface-epithelial-stromal tumours of ovarian origin.In these cases, an adequate sampling of the omentum, the adnexa, and the uterine serosa, as well as of any dominant mass, is required to establish the primary origin of the malignancy.In our case, a stromal invasion with tumor size more than 5 × 5 mm in diameter and the extensive involvement of the ovary surface have been considered the conclusive criteria of primary bilateral ovarian PC.Clinical behaviour of PCs closely resembles that of borderline serous tumours with a protracted course and a favourable prognosis. Nevertheless, tumors with an aggressive course have been described in the literature ,31. In tOur patient chose to receive adjuvant chemotherapy and is alive and free from disease after 10.5 years of follow-up. The excellent prognosis is in agreement with previous reports.Our study performed on a large screening series confirms that the presence of PBs in CVS is a rare finding, more often associated with benign conditions. Anyway, this finding should never be underestimated and a careful examination of the morphological features of the cell groups is crucial to establish the source of PBs and disclose an underlying malignancy. Additional clinical investigations are always recommendable.Moreover, we have described the first report of ovarian PC in which the presence of malignant cells and PBs in the cervicovaginal and endometrial smears represents the first manifestation of the disease.The authors declare that they have no competing interests.TP, AMP, IP and LM participated equally in the design of the paper and in the collection of the clinico-pathological data. FDN and SL participated in the revaluation of the cytological and histological specimens and in the drafting of the manuscript. SL revised critically the final version of the manuscript. All authors read and approved the final manuscript.


Isolated splenic peliosis is an extremely rare occurrence, and this disease often manifests itself with spontaneous haemoperitoneum.We report a case where an otherwise healthy patient was found to have splenomegaly on clinical examination. On computerised tomography, a diagnosis of splenic malignancy was made, and the patient underwent a splenectomy. Histological examination gave the diagnosis of splenic peliosis, which had not been considered prior to the operation. In retrospect, splenectomy was the most prudent course of action, as the risk of spontaneous haemorrhage and fatality was eliminated. This case emphasises the need to retain an index of suspicion for this condition, even in otherwise healthy patients, and is a reminder of the usefulness of total splenectomy in the current era of minimally invasive diagnostic techniques. Peliosis is a rare condition of unknown aetiology, characterized by the presence of multiple blood-filled cysts within the parenchyma of solid organs . It tendWe describe a case of asymptomatic idiopathic splenic peliosis, in which a patient presented with an incidental splenic mass on computerised tomography. This lesion had radiological features indistinguishable from a primary splenic malignancy, and the patient subsequently underwent a splenectomy. Only on histology was the lesion then confirmed to be peliosis.A 78 year old Chinese gentleman presented to respiratory clinic with a 1 month history of haemoptysis. he had undergone a pneumonectomy fifty years previously in China for pulmonary tuberculosis, and was otherwise fit and well. The only abnormal finding on physical examination was splenomegaly. Bronchoscopy and cultures for mycobacteria were unremarkable. Subsequent computerised tomography of the chest and abdomen revealed a large 13 cm × 10 cm × 13 cm hypodense, inhomogenous, non-enhancing splenic lesion, with no further abnormality seen in the abdomen or chest, apart from signs consistent with previous pulmonary tuberculosis and pneumonectomy (Figure After a multidisciplinary team discussion, a diagnosis of primary splenic tumor was made, and further management in the form of a splenectomy was advised.At operation, the spleen was found to be almost entirely replaced with a large multinodular, haemorrhagic mass. There were no other abnormal findings. A standard splenectomy was performed, and the patient was discharged uneventfully with appropriate prophylaxis.Histopathological assessment revealed the removed spleen comprised a parafollicular zone replaced with blood-filled cystic spaces, and a central fibrinoid encapsulated area, with extensive dystrophic calcification consistent with secondary thrombosis. No amyloid, TB or fungi were found within the sample. A diagnosis of splenic peliosis was made.On one year follow-up, the patient was healthy and well. The cause of his original haemoptysis, which ceased prior to the surgery, was never elucidated.The term peliosis arises from a Greek term meaning "blackish-bluish with sugillation", and was first used by Wagner in 1861 to describe the gross appearance of lesions found on the cut surface of a liver affected by the disease . PeliosiIsolated splenic peliosis is extremely rare , occurriOur case is unusual, as the patient described was otherwise fit and well, with no evidence of chronic disease. Neither was he taking any medications associated aetiologically with peliosis. Consequently, the finding of an isolated splenic mass posed a diagnostic and therapeutic challenge. Incidental splenic masses are sometimes encountered in patients undergoing imaging for other diseases , and priIn our case, histology revealed almost complete replacement of the spleen by peliotic lesions, and we believe a policy of biopsy-guided diagnosis and conservative management would have exposed the patient to the unacceptable risk of suffering from spontaneous haemorrhage. Our patient therefore underwent a splenectomy, which we believe in retrospect, was the best course of action. Splenectomy offers the advantage of a definitive histological diagnosis and the complete elimination of risk of spontaneous haemorrhage. It does however, result in the lifelong requirement of antibiotic prophylaxis.There are no published guidelines on managing patients with suspected splenic peliosis, and as this condition is so rare, it is also frequently overlooked as a potential differential diagnosis in cases of idiopathic splenomegaly. We feel clinicians should retain a high index of suspicion for the presence of this disease, and be aware of the potentially fatal consequences of missed diagnosis. In cases where peliosis enters the differential diagnosis of a splenic mass, total splenectomy should be considered as the most prudent course of action.


We report herein a case of a synchronous presentation of an adenocarcinoma of esophagago-gastric junction type II and an ampullary tumor that was treated by combined Whipple's pancreaticoduodenectomy, total gastrectomy and esophagectomy. The magnitude of this operation was safely achieved with meticulous surgical techniques and perioperative care without any major short or long term complications. Patient returned to a good quality of life at six-month follow up with no further gastrointestinal symptoms or evidence of disease recurrence. Adenocarcinoma of the esophago-gastric junction (AEG) is classified as per Siewert et al. into AEG type I, arising in the distal esophagus usually on a background of Barrett's esophagus, AEG type II which arises at the anatomic cardia, and AEG Type III tumors are sub-cardial gastric carcinomas which invade the junction and lower esophagus from below [There has been an increase in the West in recent years of adenocarcinoma of the esophagus and esophagago-gastric junction. Standard work-up in high volume centers includes computed tomography (CT) scan, endoscopic ultrasound scan (EUS) and 8F-2-fluoom below . Multimoom below . The besom below .Ampullary cancers, unlike pancreatic carcinomas, may present at an early stage. The goal of surgical management, as in all tumors, remains removal of all gross and microscopic disease from the pancreas and draining lymph node basins . The 5-yIn this high volume esophageal (50-80 resections/year) and middle volume pancreatic center (8-15 Whipples/year) a routine work up of a patient with AEG type II junctional tumor uncovered a synchronous ampullary tumor. The case and management considerations are discussed herein.A 62 year old man was referred to our service in 2008 with a history of epigastric pain and melaena of one week duration. An upper gastro-intestinal endoscopy revealed an AEG type II junctional tumour Figure . Biopsie18F-FDG-PET imaging revealed no abnormal uptake. Endoscopic Retrograde Cholangiopancreatography (ERCP) revealed a tumor with a large distorted ampulla  at the duodenal ampulla and major pancreatic duct (MPD) Figure . CT imagThe patient had good overall performance status. He had a background history of insulin dependent diabetes mellitus with secondary complications including retinopathy and peripheral neuropathy. His HbA1c on admission was 8.0 g/dL. He also had a history of ischemic heart disease with subsequent angioplasty in 2006. He had hypertension, diverticular disease and colonic polyps. A pre-operative exercise stress test and ECHO revealed good functional capacity with an ejection fraction of 60-65%. His FEV1 was 2.37 litres and his FVC was 4.30 litres. Pre-assessment by the anaesthetist, cardiologist and endocrinologist certified patient fit for major operation.The decision was made to proceed to synchronous resections with a planned Whipple's, total gastrectomy and distal esophagectomy, mediastinal dissection and Roux-en-Y esophago-jejunal reconstruction in that order respectively via the upper midline laparotomy incision. There was no technical difficulties in reconstructing the four anastomoses namely hepatico-jejunostomy, pancreatico-jejunostomy, esophago-jejunostomy and jejuno-jejunostomy Figure . The opeHistology of the junctional tumor revealed a 6 mm tumor, pT1 N0 Mx, with 0 out of 16 nodes involved and negative margins. The ampullary tumor was invasive adenocarcinoma, pT1 N0 Mx, and 0 out of 7 nodes were involved. No adjuvant therapy is being considered. Patient returned to a good quality of life at six-month follow up with no further gastrointestinal symptoms or evidence of disease recurrence.Synchronous presentation of esophago-gastric and peri-ampullary adenocarcinoma is extremely rare. In a study by Lee et al., 3.4% of gastric cancers were associated with synchronous tumors, most commonly colorectal cancer . AnotherFor both tumors independently, surgical resection is the only curative approach, and the risk of mortality for each procedure should be less than 5 per cent in high-volume centers. Neoadjuvant chemotherapy or chemoradiotherapy for AEG type II junctional adenocarcinoma was discussed but not pursued since this appeared localized and node-negative with a high probability of an R0 resection . The rolThe surgical treatment options considered in our patient were combined Whipple's pancreaticoduodenectomy, total gastrectomy, distal esophagectomy and Roux-en-Y esophago-jejunal reconstruction along with hepatico-jejunostomy, pancreatico-jejunostomy and jejuno-jejunostomy. We were prepared to do a thoracotomy to get adequate clearance if required, but splitting the diaphragm and a mediastinal dissection and anastomosis were achievable via the abdominal incision. Increasing evidence supports the rationale to avoid transthoracic surgery for AEG type II tumors which are predominantly of gastric origin and rarely involve high mediastinal or sub-carinal nodes ,3.For the ampulla, the EUS and ERCP were more in keeping with an invasive rather than an in situ cancer, and a local resection was not considered. A pylorus -preserving approach with gastric preservation combined with a proximal gastrectomy and esophagectomy would have been technically possible, but functional results from total gastrectomy with Roux-en-Y esophago-jejunal reconstruction may be better due to the risk of reflux esophagitis. There appears to be only one previous report in the literature of a similar procedure for synchronous tumors. Mafune et al. described a case of subtotal esophagectomy for SCC, pancreaticoduodenectomy with lymph node dissection and total residual gastrectomy in a patient who had a previous gastrectomy .Synchronous resection of AEG type II junctional tumour and ampullary tumour appears attractive although it takes longer to perform, the benefits are having one general anaesthesia for four anastomotic reconstructions and thus avoiding a second major operation. Whilst a staged procedure may take shorter to perform, the risks are two general anesthesia with potential adhesions complicating second stage procedure and the potential delay in resection of second rapidly progressing tumour. Suzuki et al. reported simultaneous resection of synchronous esophageal and extraesophageal carcinomas can be safely performed with no significant difference in morbidity and mortality compared to solitary esophageal cancer resection as long as complete clearance of both tumours is achieved for favourable long-term results .This is the first report of combined Whipple's and esophago-gastrectomy for these synchronous tumors. The magnitude of this operation was safely achieved with meticulous surgical techniques and perioperative care without any major complications.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.NJ and FO drafted the manuscript. KTL prepared the figures, reviewed and amended and finalized the manuscript. JVR critically reviewed, amended and finalized the manuscript. All authors read and approved the final manuscript.


Lipoma is the most frequent benign tumor of the soft tissue. This lesion is often asymptomatic except in cases of enormous masses compressing nervous-vascular structures. Although the diagnosis is mostly clinical, imaging tools are useful to confirm the adipose nature of the lesion and to define its anatomic border. Sometimes, lipomas may be the result of a previous trauma, such as in this patient.A 45-year-old man presented at our institution with a giant hard firm mass in the upper external quadrant of the right buttock disclosed after a weight loss diet. Subsequent magnetic resonance imaging showed a giant adipose mass developed beneath the large gluteal muscle and among the fibers of the medium and small gluteal muscles. When questioned on his medical history, the patient reported a blunt trauma of the lower back 14 years earlier. He underwent surgery and histological examination confirmed a giant lipoma.Lipomas might result from a previous trauma. It is hypothesized that the trigger mechanism is activated by cytokine and growth factors released after the trauma. We herein present an exceptional case of a giant post-traumatic lipoma which caused a painful compression on the right sciatic nerve. Lipoma is the most frequent benign tumor of the soft tissue. Although lipomas have been described in almost all of the organs occurring in the central nervous system, gastrointestinal tract, muscles and joints, most of the time they arise in subcutaneous tissue and can occur in every part of the body. This lesion is often asymptomatic except in cases of enormous masses compressing nervous-vascular structures. Although the diagnosis is mostly clinical, imaging tools are useful to confirm the adipose nature of the lesion and to define its anatomic border.Sometimes, lipomas may be the result of a previous trauma, such as in the present case. It is hypothesized that the trigger mechanism for lipoma development is activated by cytokine and growth factors released after the trauma . We presA 45-year-old man presented at our institution with a giant hard firm mass in the upper external quadrant of the right buttock disclosed after a weight loss diet. His past medical history was positive for a previous blunt trauma in that area, as a result of a motor vehicle accident 14 years earlier. Since that time, he had felt a mass arising very slowly until the present time. The patient complained of pain and a functional limitation of the right leg. For this reason, he was scheduled for removal of the mass. Magnetic resonance imaging (MRI) was performed to assess the anatomy of the mass and its relationship with the surrounding structures Figures . The masth to 7th decades. Although lipomas have been described in almost all of the organs occurring in the central nervous system, gastrointestinal tract, muscles and joints, most of the time, they arise in subcutaneous tissue in every part of the body. Histologically, there are sub-types of lipomas that can be recognized by the presence of bone formations (osteolipoma), an important myxoid change (myxolipoma), rich fibrous tissue, or cartilage (chondrolipoma).Lipoma represents the most frequent tumor of the soft tissues with a predilection for female, obese patients, in the 5Even though there are different types of lipoma, their gross appearance does not change. However, bony areas or gray glistening formations can be found in osteolipoma and chondrolipoma variants.The typical clinical aspect is soft, floating, lobulated and movable in the case of subcutaneous lipomas, with an average size of 3 cm. Lipomas arising from deeper compartments, such as subfascial, intramuscular or intermuscular lipomas, appear as capsulated, well circumscribed masses with smooth borders and often adherent to the muscle. They represent less frequent lipomas that can reach unusual size exceeding 10 cm, such as in this patient.The majority of lipomas retain a stable volume or slowly increase in size, remaining painless and asymptomatic masses that rarely invade the surrounding structures. Less commonly, they reach giant volume and weight, especially in the case of post-traumatic lipomas, giving symptoms caused by nerve and vascular compression.The reason for the uncontrolled growth is still unclear. In the last few years, various theories have been put forward such as endocrine, dysmetabolic, and genetic origin. Also, acute blunt trauma has been considered one of the possible pathogenic causes ,3. A recet al. reported 15 lipomas developed after blunt trauma [et al. supported the active role of post-traumatic released cytokine and growth factors on precursor adipose cells [The role of blunt trauma in lipoma development is primarily supported by the rearrangement of the lipomatous tissue after its transfascial herniation caused by a deep blunt injury . It has t trauma . It is ht trauma . Signorise cells . These fLipoma represents the most frequent tumor of the soft tissues. Several authors have reported cases of post-traumatic lipoma. The mechanism that causes these lesions is not yet known. It seems to be related to the release of post-traumatic cytokine and growth factors that stimulate precursor adipose cells. Knowledge of this process would be helpful to inhibit post-traumatic lesions such as scars and lipomas, however, further studies are necessary to achieve this goal.The authors declare that they have no competing interests.GN designed the study, drafted and revised the manuscript. MD, SV, GB, PA, FDA, and GR carried out the data and image acquisition and participated in the writing process. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Soft tissue sarcomas in the head and neck region are rare and often present a difficult differential diagnosis. The aim of our presentation is to point out the complexity of the diagnosis, treatment and follow up.An eighty-seven year old female patient was referred to our unit with a fast growing brownish lump on the face. Four months beforehand, a benign fibrous histiocytoma (BFH) had been removed from the same location by excision biopsy with wide tumour-free resection margins. Excision biopsy of the recurrent lesion revealed a malignant fibrous histiocytoma (MFH). Radical tumour resection was completed by extended parotidectomy and neck dissection; the skin defect was covered by a regional bi-lobed flap. No adjuvant radio- or chemotherapy was administered. Full functional and cosmetic recovery was achieved; follow-up has been uneventful more than two years postoperatively.Malignant transformation of BFH is extremely rare and if so, extended radical surgery may give a fair chance for a favourable outcome even in patients with advanced age. Soft tissue tumours in the head and neck region sometimes display borderline pathological features regarding benign or malignant behaviour. Despite similar histological patterns the clinical outcome is often different and difficult to predict.Malignant fibrous histiocytoma [MFH] is a primitive, often pleomorphic sarcoma consisting of partly fibroblast-like, partly histiocyte-like cells. It has been classified as a distinct histopathological entity since the nineteen-sixties . NowadayChanging of the histological picture during progression of the disease and transformation of borderline-benign lesions to malignant phenotype has been described. Reported incidence of malignant transformation lies around 1% .Benign fibrous histiocytoma [BFH] is the most common benign neoplasm in practically all localizations affecting the epidermis. The proliferative, atypical or borderline type is a cell-dense variant mostly growing faster and greater in size, with characteristically frequent local recurrences, reported incidence lying at 3–5%. Both BFH and its borderline variant are known not to give either haematogenous or loco-regional metastases to lymph-nodes .The aim of our presentation is to point out the complexity of the diagnosis, treatment and follow up of patients with soft tissue tumours of borderline character.An eighty-seven year old woman was referred to our department because of a fast-growing, dark-brownish, indurated skin nodule sized 3 cm in diameter, which infiltrated the skin and the deeper soft tissue layers of the face Fig . Four moExcision biopsy of the current lesion revealed an infiltrative, well-vascularised mesenchymal neoplasm containing pleomorphic tumour cells, increased mitotic count with some atypical mitoses, and myxoid stroma rich in collagen fibres. Immunohistochemistry was strongly positive for vimentin, and negative for cytokeratin, smooth-muscle actin, desmin, S-100 and melanoma-specific antigen. The MIB1 positive proliferative fraction was 50% Fig. . The hisClinical staging investigations of the current malignancy  showed superficial infiltration of the cutis and subcutis. Neither infiltration of the Masseter nor metastases in the regional lymph nodes or elsewhere were found. Radical excision of the tumour was completed by parotidectomy and modified neck dissection, which resulted in a cosmetic defect of the right face that was immediately corrected by a local bi-lobed flap Fig. . No postBenign fibrous histiocytomas (BFH) are frequently found in sun-exposed skin of the extremities and of the head and neck. They are among the most common soft tissue lesions of the skin . Their bIn contrast, malignant fibrous histiocytoma (MFH) is a sarcoma with both fibroblastic and histiocytic features. It has been classified as a distinct histopathological entity since the nineteen-sixties . NowadayThe primary treatment of MFH consists of radical excision with wide safety margins and dissection of the loco-regional lymph nodes . Post- oIn conclusion, predicting biological behaviour on the base of cellular features in cutaneous fibrohistiocytic tumours is sometimes very difficult. It is well illustrated by the present case, which showed malignant transformation in recurrence despite the bland histopathological appearance of the primary lesion. It is therefore mandatory that patients with such lesions are closely monitored postoperatively, so as to be able to act promptly in case of local recurrences. Patients' management requires close collaboration between dermatologists, maxillo-facial surgeons, radiologists and pathologists, which may result in sufficient clinical outcome.LS and RS analysed the case, reviewed all patient data and drafted the manuscript. All authors were involved in planning the treatment. The excision biopsy, the tumour removal and the neck dissection was carried out by SK, JP whereas BKL carried out the plastic reconstruction of the face. AB and ABü did the histological and immunhistological analysis and contributed to the final conclusions of the case report. UJ suggested the idea for the case report and reviewed and contributed to the writing of final version. JP, AB, ABü and BKL contributed substantially to discussions and in writing of the paper. All authors reviewed the paper for content, and contributed to the writing of all iterations of the paper, including the final version of the manuscript. All authors approved the final report.


Omental infarction is a rare and often misdiagnosed clinical event with unspecific symptoms. It affects predominantly young and middle aged women.This is a case report of a 26-year-old Caucasian woman with spontaneous omental infarction two weeks after normal vaginal delivery.Omental infarction is a differential diagnosis in the postpartum acute abdomen. As some cases of omental infarction, which are caused by torsion, can be adequately diagnosed via computed tomography, a conservative treatment strategy for patients without complications should be considered in order to avoid any unnecessary surgical intervention. Omental infarction is a rare clinical event that affects predominantly young and middle aged women . It is uThis report highlights the case of a spontaneous omental infarction in a young woman in the postpartum period.A 26-year-old Caucasian woman presented with a five day history of increasing epigastric pain and nausea two weeks after the vaginal delivery of a healthy child of normal weight and size.Physical examination revealed a normal peristalsis and supraumbilical tenderness. A small umbilical hernia (< 1 cm diameter), with no signs of incarceration, was described by the initial examining physician. Pulse and blood pressure were normal . She was apyrexial but adynamic, with pale and clammy skin. In summary, the general status of the patient was impaired on admission (American Society of Anesthesiologists score 2-3).Blood tests revealed an elevated white blood cell count (14.7/nL) and serum C-reactive protein (120 mg/dL). A coagulation study  revealed no abnormalities.Abdominal ultrasound showed no specific pathological findings and, for further clarification, a contrast-enhanced abdominal computed tomography (CT) was performed. The morphologic findings of the CT were interpreted as an incarcerated umbilical hernia by the radiologist. However, due to the clinical presentation of an acute abdomen and the elevated inflammatory blood parameters, the patient was asked to consent to an exploratory laparotomy. A small laparotomy (5 cm long midline incision around the umbilicus) was performed. Contrary to the CT findings, and in accordance to the clinical examination, no umbilical hernia could be detected intraoperatively. Surprisingly, a hemorrhagic greater omentum measuring 11 × 7.5 × 2.5 cm was discovered and resected. A small amount of sanguinous ascites was also found. On further exploration we found no adhesions or other underlying causes for the infarction, such as an external or internal hernia or a vascular pedicle.In a retrospective repeat analysis of the CT scan, a hypoperfused mass of fatty appearance in the anterior portion of the midabdomen and small amounts of free fluid surrounding the liver were observed Figure .Histopathological findings of the resected omental specimen confirmed fresh hemorrhagic infiltrations of the tissue, partial thrombosis of the small vessels and, in some parts, necrotic fatty tissues with an acute inflammatory cellular infiltrate Figure . FurtherThe patient was discharged after an uneventful recovery three days after surgery.Omental infarction was first described in the late 19th century and, since then, only a few hundred cases have been published in the English literature . This isThe etiology of omental infarction without torsion remains uncertain but several mechanisms have been proposed, such as an anomaly of venous vessels . Other pAs most patients show symptoms of an acute abdomen, CT of the abdomen and pelvis should be the diagnostic imaging of choice . If omenDiagnosis of an omental infarction has traditionally been made intraoperatively during an exploratory laparotomy or laparoscopy and the treatment has been partial or total omentectomy. Recent reports highlight cases of patients with CT diagnosed omental torsions who have been successfully treated conservatively without any other complications  -15. WhenOmental infarctions are often not initially considered in the differential diagnosis of a post partum acute abdomen. When omental infarction is caused by torsion, a correct preoperative diagnosis by contrast-enhanced CT scanning can avoid surgery. Recently published case series have reported successful conservative management.The authors declare that they have no competing interests.MT, RG and JTK managed the patient and reviewed the literature. MT and RG were the main authors of the manuscript. AHM analyzed the histopathological specimen. FG, JTK and JRI made modifications to the manuscript. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Angiolipomas are frequently observed benign tumours. They have a typical vascular component and are often located in subcutaneous tissues, and more rarely, in the gastrointestinal tract.We report an uncommon case of an angiolipoma found in the lower portion of the small bowel of an 80-year-old man who was undergoing evaluation for chronic anaemia. A standardised diagnostic pathway was followed. Endoscopic and radiological findings were negative. The diagnosis was finally established with the aid of capsule endoscopy. The case we report is the first in the literature of an angiolipoma without specific painful symptoms. In fact, the patient did not complain of abdominal pain or alvus changes, and abdominal examination did not suggest an expansive process. The endoscopic study performed with the capsule identified the lesion as the cause of the ingravescent anaemia. Intra-operative histological examination of the lesion made it possible to avoid a major surgical procedure and assured a short postoperative course for the patient.This report focuses on the importance of correct pre- and/or intra-operative histological diagnosis in order to offer the best therapeutic choice. An angiolipoma was suspected in this case, even though they are rarely located in the ileum. Angiolipomas are benign tumours consisting of typical proliferative vascular tissue. They are generally located in subcutaneous tissues. The areas most involved include the upper and lower limbs and the trunk, while gastrointestinal localisation is extremely rare ,2.In our opinion, when angiolipoma is suspected, it is crucial to determine the diagnosis pre-operatively and, whenever possible, to clarify the diagnosis intra-operatively by histological examination, in order to perform the optimal procedure.From a clinical point of view, this case involved an asymptomatic patient and because of this, the case report is novel.An 80-year-old man who underwent triple aortocoronary bypass surgery was affected by an aneurysm of the abdominal aorta, bilateral obstructive arteriopathy of the lower limbs, colonic diverticulosis, and chronic obstructive pulmonary disease. He came to our attention in June 2006 for evaluation of a normocytic hypochromic anaemia . He did not complain of melena or haematemesis, although a faecal occult blood test was positive. The patient did not complain of abdominal pain or alvus changes, and abdominal examination did not suggest an expansive process. During hospitalisation, routine haematology and biochemical tests were repeated and anaemia was confirmed. Tumour markers  were negative, except for gastrin (155 pg/ml).To identify the cause of the anaemia, oesophago-gastro-duodenoscopy was performed, which did not reveal any pathological finding, Anorectal-colonoscopy that confirmed diverticulosis only, with no signs of inflammation or past or present haemorrhage.A lesion located in the small bowel was suspected to be the cause of the anaemia, after an episode of enterorrhagia led to a fall in the patient's haemoglobin . Since a double balloon enteroscopy was not possible, a capsule endoscopy was performed. This investigation revealed the presence of a polypoid lesion located in the terminal ileal loop, occupying two-thirds of the lumen and covered by strongly congested and hypervascularised mucosa Figure .Abdominal pre- and post-contrast enhancement computed tomography scans showed only slight thickening and an anomalous impregnation of the mucous profile of the terminal ileal loop, with no loco-regional lymphadenopathy.The patient underwent a second colonoscopy with a retrograde ileoscopy to obtain a tissue diagnosis of the lesion. This examination confirmed the presence of ileal sessile polypoid lesions with a hypervascularised basement, covered by a strongly congested mucosa Figure . As it w2) was removed. Frozen section histological examination was conducted . MA and PDS edited the report and compiled the reference list. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Cavernous hemangioma is the most common benign noninfiltrative neoplasm of the orbit. Most cavernous hemangiomas are intraconal and lateral in location. We present a case of a cavernous hemangioma with an unusual extraconal and superomedial location. Cavernous hemangioma is the most common benign noninfiltrative neoplasm of the orbit. Most cavernous hemangiomas are intraconal and lateral in location. They result from the new formation of vessels, proliferation of tissue components of the vessel wall, and hyperplasia of cellular elements ordinarily concerned with the genesis of vascular tissue. A case oAn 18-year-old woman presented with gradually progressive proptosis of the left eye with swelling of the superomedial quadrant of the left orbit. The swelling did not change in size with the Valsalva maneuver, coughing, straining, or change in head position. The cornea and sclera were normal.USG revealed a well-encapsulated, compressible, echogenic mass lesion (approximately 2.8 × 1.7 cm in size) situated superomedially in the extraconal space of the left orbit. On Doppler, the mass showed multiple vascular channels  suggestive of a vascular lesion . Small lThe tumor was excised using an anterior orbitotomy approach. Histopathology confirmed the diagnosis of a cavernous hemangioma.Hemangiomas (benign vascular neoplasms) are classified as capillary and cavernous. A capillary hemangioma usually presents in the first year of life and often increases in size for 6–10 months before slowly involuting. Cavernous hemangiomas are the most common benign noninfiltrative neoplasms of the orbit and have a slowly progressive mass effect. They are3A slowly progressive proptosis is the typical presenting symptom. Extraocular muscle impairment and impaired visual function are seen with large lesions and with lesions located at the orbital apex. Clinically, these tumors are soft and do not change in size with the Valsalva maneuver or with coughing, straining, or change in the head position.4Most cavernous hemangiomas are typically intraconal and lateral in location. Extraconal and medial locations are uncommon.5 USG, CT5The tumors are round to oval in shape with well-defined borders and a specific ‘honeycomb’ pattern of alternating weak and strong echoes corresponding to their structure, with flow on color Doppler.5 They maCT scan shows discrete lesions with varying degrees of enhancement. On MRI, the lesions are homogenous, isointense to muscle on T1W images, and hyperintense to muscle on T2W images, as was seen in the present case. Variable homogenous or inhomogeneous contrast enhancement is the rule.5 These lHistopathology reveals a fine capsule that surrounds a tumor consisting of large endothelium-lined channels with abundant, loosely distributed smooth muscle in the vascular wall and stroma.6Treatment of the tumor is surgical excision. Complete excision is generally accomplished as the tumor is well encapsulated with relatively few feeding vessels.8


Congenital infiltrating lipomatosis of the face is a rare condition characterized by diffuse fatty infiltration of the facial soft tissues. There may be muscle involvement along with associated bony hyperplasia. It is a type of lipomatous tumor that is congenital in origin; it is rare and seen usually in childhood. We recently saw an 11-year-old girl with this condition. She presented with a swelling of the right side of the face that had been present since birth; there were typical findings on plain radiographs, CT, and MRI. The patient underwent cosmetic surgery. Histopathological examination showed mature adipocytes without any capsule. Congenital infiltrating lipomatosis of the face is a rare entity characterized by collections of nonencapsulated, mature lipocytes that infiltrate local tissues, leading to craniofacial deformities. These ch2An 11-year old girl presented with a swelling of the right side of the face since birth that had been progressively increasing in size . There wFrontal skull radiographs  showed iSubsequent MRI on a 1.5-T scanner showed a nonenhancing, ill-defined, infiltrative, inhomogeneous, hyperintense lesion (isointense to fat) on T1W images in the subcutaneous, muscular, and intermuscular planes of the face and upper neck on the right side Figures  and B; tThe patient underwent cosmetic surgery. Histopathological examination showed mature adipocytes without any capsule. Diffuse infiltration of the intervening muscles was also identified. There was no cell atypia to suggest malignancy. These findings confirmed the radiological diagnosis.et al.[Simple encapsulated lipomaLipoma variants like angiomyolipomaHamartomatous lesionsInfiltrating or diffuse lipomatosisBenign tumor of brown fat – hibernomaCongenital infiltrating lipomatosis was first described by Slavin et al. in 1983.et al. and is cet al. have fivInfiltrating lipomas usually occur after the third decade of life and invoPathologically, these lesions show the following characteristics: they arePlain radiographs show hypertrophy of facial bones and soft tissue swelling. USG may show adipose tissue but cannot delineate the real extent of the lesion. CT scan 4The differential diagnoses include The treatment modalities available are liposuction and excision. This is done for cosmetic reasons. Although these tumors are benign, the rate of recurrence is very high after surgical excision.5In conclusion, congenital infiltrating lipomatosis of the face is a rare benign condition occurring in childhood. It is characterized by diffuse infiltration of fat in the subcutaneous and muscle planes and bony hypertrophy. Clinical examination and imaging, especially MRI, can establish the diagnosis. Surgery is done for cosmetic purpose.


An accessory lobe of the liver is a rare congenital anomaly that can undergo torsion and present as an acute surgical emergency. It is rarely diagnosed preoperatively. We report the preoperative utility of CT scan and MRI in the diagnosis and surgical planning of a case of intermittent accessory hepatic lobe torsion. Accessory hepatic lobe is an extremely rare anomaly. Most casA 24-year-old, white woman, presented to the emergency room with a 1-day history of intense and constant cramping upper quadrant abdominal pain, nausea, and vomiting.She had a prior history of several similar episodes of abdominal pain for which no cause had been found. The past surgical history was significant for treatment of malrotation, anterior abdominal wall repair, and removal of a Meckel's diverticulum and appendix at age 11.At admission, her vital signs and laboratory studies were normal. Her abdomen was soft, nondistended, without rigidity, but she was tender to palpation in the right upper quadrant and epigastric region.A CT scan at an outside facility reported a “right upper quadrant mass.” Since the images were not immediately available, a biphasic contrast enhanced CT scan of the abdomen was performed at our institution. Post-processed, multiplanar reconstructions (MPR) and maximum intensity projection (MIP) thick slab images were also obtained. The CT scan showed a region of liver parenchyma separate from the normal liver and of lower attenuation, attached to the left lobe by a thin stalk of liver tissue that contained a vascular pedicle consisting of hepatic arterial supply from the left main hepatic artery, portal venous supply, hepatic venous drainage directly to the IVC and independent biliary ducts Figure –C. The vDuring the course of her hospital stay, the patient showed significant improvement in her symptoms and was discharged in a stable condition with a diagnosis of intermittent torsion of an accessory lobe. Elective exploratory laparotomy performed 4 months later confirmed the presence of an accessory hepatic lobe . The hisAn accessory hepatic lobe is a rare congenital developmental anomaly that is usually asymptomatic and is seen as an incidental finding at laparotomy.2 There a1The presence of an accessory hepatic lobe occurs from an error in the formation of the endodermal caudal foregut in the third gestational week and segmentation of the hepatic bud. Anterior25An accessory liver is adjacent and attached to the liver by its own mesentery, while an ectopic liver is one that is completely detached from the normal liver parenchyma. While a definite connective tissue stalk attaching the lobe to the rest of the liver was present in our case, a mesenteric covering was not found at surgery. Older literature has described four types of accessory liver: big accessory hepatic lobe (> 30 g), small accessory hepatic lobe (< 30 g), ectopic lobe with no liver connection, and microscopic accessory lobe in the gall bladder wall.Classification can also be based on the biliary drainage and the presence or absence of a common capsule:Type I. The separate accessory lobe duct drains into an intrahepatic bile duct of the normal liver.Type II. The separate accessory lobe duct drains into an extrahepatic bile duct of the normal liver.Type III. The accessory lobe and the normal liver have a common capsule; the bile duct of the accessory lobe drains into an extrahepatic duct.[tic duct.Symptomatic accessory hepatic lobe has been reported in infants as young as 23 days of age as well as in patients presenting late in the third decade of life commonly from torsion.6 The usu3Accessory hepatic lobe may present as an undetermined soft tissue mass on imaging. Also, torsion complicated by congestion or infarction can further confuse the appearance.78 The di72In conclusion, it is important for the radiologist to be aware of this entity as a cause of recurrent right upper quadrant acute abdominal pain. Though rare, routine inspection of vascular and biliary drainage of the liver as part of the check list while evaluating all CT abdomen exams will alert the radiologist to this diagnosis when a mass is present in the right upper quadrant. In the correct setting, a past history of an abdominal wall abnormality is also suggestive of the presence of an accessory hepatic lobe. Preoperative diagnosis of this abnormality and mapping its vascular and biliary anatomy help reduce the overall morbidity associated with abdominal surgery.


We report a rare case of echinococcosis, primarily involving the right kidney and ureter, presenting with gross hydatiduria. We also present the salient diagnostic features of renal hydatid. Kidney involvement in echinococcosis is extremely rare, constituting only 2-3% of all cases. Primary involvement of the kidney without the involvement of the liver and lungs is even more rare. Hydatiduria accompanies only 10-20% of all cases of renal hydatidosis and is usually microscopic. We present a rare case of primary right renal hydatid having multiple daughter cysts in the upper and mid ureter with gross hydatiduria. The diagnosis of primary hydatid cyst of the kidney, in the absence of hydaturia, is usually radiological as most patients have negative immunological tests. We also present the salient radiological features of primary hydatid cyst of kidney.A 23-year-old male presented with history of intermittent passage of small, white, balloon-like, grape-sized structures in the urine for the last three months. Abdominal examination did not reveal any palpable lump. Rest of the systemic examination was normal. Gross examination of a single balloon-like structure in the urine revealed a membranous cyst measuring around 2 cm; histopathology showed an outer laminated layer with an inner germinal layer. The laminated structure was consistent with a hydatid cyst.His routine blood investigations were normal with no eosinophilia and normal renal function tests. X-ray chest P-A view was normal. The USG abdomen revealed multiseptate cyst in the right kidney; liver was normal. The CT scan revealed a cystic lesion in the right kidney and upper ureter . The whoE. granulosus in this case. Echinococcus belongs to the order Cestoda and the family Taenia. It is about 5 mm long. The adult E. granulosus worm resides in the large bowel of foxes and dogs. Man is the intermediate host and gets the disease by ingesting vegetables and water contaminated by the affected dogs. Hydatid disease involves the liver in approximately 75% of cases and the lung in 15%. Secondary involvement due to hematogenous dissemination may be seen in almost any anatomic location. Kidney involvement is extremely rare (2-3%).  (2-3%).1 Gross paPlain films are usually nonspecific and mostly nonrevealing. A thin rim of calcification delineating a cyst is suggestive of an echinococcal cyst. Ultrasonography helps in the diagnosis of hydatid cysts when the daughter cysts and hydatid sand are demonstrated. On changing the patient's posture under real time, there is shifting of hydatid sand, which may give rise to the "falling snowflake pattern". The accuracy of ultrasound evaluation remains operator-dependent. The CT scan has an accuracy of 98% and sensitivity to demonstrate the daughter cysts. The CT scan usually demonstrates an expansile, hypo-attenuating tumor with a well-defined wall and daughter cysts within the parent cyst. The central cystic part of the lesion has an attenuation of 30-35 HU, in contrast to the much lower attenuation of the fluid in the surrounding cysts (5-15 HU), giving the mass a wheel-like or rosette appearance. Magnetic resonance imaging usually reveals a solitary, high-signal-intensity mass consisting of multiple thin-walled lesions and outlined by a thick, hypointense rim. The high signal intensity is due to the characteristic high fluid content of the mass. The small peripheral cysts are usually hypointense relative to the central component. The MRI shows the cysts adequately, but MRI offers no real advantage over CT scan.[In general, surgery is the treatment of choice in renal hydatid cyst. Kidney-sparing surgery  is possible in most cases (75%). Nephrectomy (25% of cases) must be reserved for destroyed kidney. Very few cases of laparoscopic removal of renal hydatid are reported. There is fear of cyst rupture and dissemination during dissection, entrapment and removal of the hydatid cyst during laparoscopy. Utmost care should be taken during the surgery to prevent spillage and resultant disseminated hydatidosis. Pre and postoperative one-month courses of Albendazole should be considered in order to sterilize the cyst, decrease the chance of anaphylaxis and decrease the tension in the cyst wall (thus reducing the risk of spillage during surgery) and to reduce the recurrence rate postoperatively. During kidney-sparing surgery scolicidal solutions such as hypertonic saline should be used before opening the cavities to kill the daughter cysts and therefore prevent further spread or anaphylactic reaction.


Pigmented intraosseous odontogenic lesions are rare with only 47 reported cases in the English literature. Among them, pigmented calcifying cystic odontogenic tumor, formerly known as calcifying odontogenic cyst, is the most common lesion with 20 reported cases.A case of pigmented calcifying cystic odontogenic tumor associated with odontoma occurring at the mandibular canine-premolar region of a young Japanese boy is presented with radiographic, and histological findings. Special staining, electron microscopic study and immunohistochemical staining were also done to characterize the pigmentation.The pigments in the lesion were confirmed to be melanin by Masson-Fontana staining and by transmission electron microscopy. The presence of dendritic melanocytes within the lesion was also demonstrated by S-100 immunostaining.The present case report of pigmented calcifying cystic odontogenic tumor associated with odontoma features a comprehensive study on melanin and melanocytes, including histochemical, immunohistochemical and transmission electron microscopic findings. Pigmented odontogenic lesions are rare, with only 47 cases reported in English literature since 1961 [Table A 15-year-old Japanese boy was referred to the Okayama University Hospital by the orthodontist for management of a mixed radiolucent and radiopaque lesion in the left mandibular canine-premolar region detected during routine radiographic examination. The lesion was asymptomatic and the patient's medical and family histories were non-contributory.On intraoral examination, there was no bony expansion and the overlying mucosa was also normal. Panoramic radiograph showed a well-defined unilocular radiolucent lesion with distinct sclerotic margin containing radiopaque masses Fig.  and CT sMicroscopic examination of the soft tissues revealed dental follicle-like loose connective tissue lined by a non-keratinized stratified epithelium of uneven thickness. The thickest portion of the epithelium showed basal cuboidal ameloblast-like cells and suprabasal stellate-like cells Fig. . Few rouAmong the three calcified masses, two were small organoid denticles composed of enamel matrix, well-developed tubular dentin and pulp-like tissues with intervening loose connective tissues Fig. , inset. The melanin granules were confirmed by Masson-Fontana staining Fig.  and melaA final diagnosis of pigmented CCOT associated with compound odontoma was undertaken due to the presence of well-developed denticles within the small cystic structure lined by typical odontogenic epithelium based on the criteria for the diagnosis of CCOT by the World Health Organization . ReviewsAmong those pigmented CCOTs, 5 were associated with odontomas, 2 compound type and 3 complex or composite odontomas. All cases were intraosseous lesions and 10 out of 20 cases included special staining for melanin but only one with TEM examination. The present case report of pigmented CCOT associated with odontoma (CCOTaO) features a comprehensive study on melanin and melanocytes with special and immunohistochemical staining together with identification of melanosomes by TEM.The exact etiology of melanin pigmentation in intraosseous odontogenic lesions is unknown, leaving room for speculation. As described in previous reports, most of the patients were Blacks and Asians, thereby implicating racial pigmentation to be an important factor ,4. MelanAlthough the 2005 WHO classification includes CCOT as a benign odontogenic neoplasm , CCOT fePigmented compound odontoma was considered as a differential diagnosis because of the presence of ghost cells with subsequent calcification and that the occurrence of odontogenic epithelium is not rare in odontomas -20. CombThis is a case of pigmented CCOTaO with comprehensive studies on melanin pigments that will add to the rare literature of pigmented odontogenic lesions. All the reported cases of pigmented intraosseous odontogenic lesions in the English literature since 1961, especially pigmented CCOT, were reviewed and summarized for academic purpose.Calcifying cystic odontogenic tumor (CCOT), Calcifying cystic odontogenic tumor associated with odontoma (CCOTaO), Calcifying odontgenic cyst (COC)The author(s) declare that they have no competing interests.PPH, HN, CHS carried out the case study, discussed and reviewed the literature and prepared the manuscript. MG, RT and NN participated in the discussion and review process and also in critical revision of the manuscript. SB and KN helped in data collection and reviewing of the literature. All the authors have read and approved the manuscript.


We report the case of a 61-year old man who was treated with neoadjuvant imatinib for a massive gastric GIST with the hope of avoiding a potential multi-visceral resection.Gastrointestinal stromal tumors (GISTs) are the most common sarcoma of the alimentary tract and are believed to derive from the interstitial Cell of Cajal. Imatinib mesylate (Gleevec A 61-year old man presented with a left upper quadrant abdominal mass after experiencing several intermittent episodes of nausea, vague abdominal discomfort, and mild acid reflux. He also reported a nine kilogram weight loss over the prior six to eight months. Physical examination revealed a large mass in his upper abdomen.Abdominal computed tomography (CT) revealed a 21 × 12 cm heterogeneous mass occupying his mid and left upper quadrants  were the most frequent adverse effects in the Demetri study.A new approach is evolving that relies on neoadjuvant imatinib to down-stage the tumor in cases of advanced GISTs. Prior studies have described patients with inoperable or metastatic GISTs who underwent treatment with imatinib and had a dramatic response allowing surgical resection ,9.The median time for an objective response to imatinib is four months, but maximal response is reported to take six months or longer . ResponsWhile most patients respond to imatinib, many eventually develop resistance. Initial (primary) resistance is defined as progression of disease at the time of first follow up after start of therapy . These pThe role of imatinib in the neoadjuvant setting is illustrated in this case. While the GIST found in this patient may not have been inoperable before imatinib treatment, the procedure may have required a multi-visceral resection. Neoadjuvant imatinib was given and after dramatic radiographic response, a simple wedge gastric resection was needed.Surgeons should consider the use of neoadjuvant imatinib therapy in patients with marginally resectable GISTs. A response to imatinib can allow for a less extensive though still therapeutic oncologic resection.The authors declare that they have no competing interests.SA drafted the manuscript. JG reviewed an amended the manuscript. JMH reviewed and amended the manuscript. All authors read and approved the final manuscript.


Extraosseous osteosarcoma (EOO) is a malignant mesenchymal neoplasm that is located in the soft tissues without direct attachment to the skeletal system and that produces osteoid, bone, or chondroid material. EOO is an extremely rare disease, accounting for only 1% of soft tissue sarcomas, and typically presents in either an extremity or the retroperitoneum. This paper presents the case of a 45-year-old Caucasian male with extraosseous osteosarcoma of the esophagus. A 45-year-old Caucasian man first noticed a change in his normal state of health with some hoarseness in his voice. This hoarseness persisted for just over a week with no other associated symptoms. As the hoarseness resolved, the patient began to experience significant dysphagia to both hard and soft foods. Associated symptoms included nausea and regurgitation, but no emesis. He had also developed a new persistent cough, which was productive of clear mucus and sometimes scant hemoptysis. The patient reported fatigue, mild dyspnea on exertion, and a 10–15 pound weight loss over the ensuing 6–8 weeks. The patient was a lifetime nonsmoker and up until 3 years earlier had drank approximately 24 twelve ounce beers per week for several years. The family history was reviewed and was negative for any malignancy or familial syndromes with a predisposition for malignancy. The patient was seen by his primary care physician who ordered a chest CT scan, revealing multiple small polypoid lesions, all measuring less than 5 mm, along the wall of his distal trachea and proximal left main bronchus. There were also multiple enlarged calcified lymph nodes in the superior mediastinum. The work up continued with an esophagogastroduodenoscopy, which showed an ulcerated lesion extending from 20 to 30 cm from the incisors . EndoscoThe patient underwent placement of an esophageal stent, which helped relieve his dysphagia. Medical oncology was consulted to determine the role of systemic therapy. The patient was seen, evaluated, and scheduled to receive 4 cycles of doxorubicin and cisplatin. Given the advanced disease present in the patient, chemotherapy consisting of doxorubicin and cisplatin was decided to be the best course of treatment. The patient went to the OR for placement of a port with repeat esophagoscopy. The tumor had increased considerably from the previous endoscopy and the patient was not able to be extubated due to significant local invasion of the tumor involving his entire trachea. He was eventually extubated and the patient and his family decided on placement in hospice care.EOO is a rare condition, comprising approximately 1% of all soft tissue sarcomas . In contMost EOOs are high grade. Microscopically EOOs consist of neoplastic osteoid and bone, and frequently neoplastic cartilage. EOOs will stain positively for vimentin and there are now monoclonal antibodies to osteonectin and osteocalcin to help recognize skeletal and extraskeletal osteosarcomas. The above case involved areas of eosinophilic stromal hyaline-like material, interpreted by the pathologist as osteoid. The samples taken were also positive for osteonectin. The differential diagnosis of bone- and cartilage-forming soft tissue lesions can be long and sometimes difficult to narrow. Malignant lesions that can produce bone or cartilage include synovial sarcoma, epithelioid sarcoma, malignant fibrous histiosarcoma, liposarcoma, malignant melanoma, as well as others. Most of these neoplasms will have osteoid or bone in only a small portion of the tumor in a well-differentiated pattern. Oftentimes this is the only way to distinguish some of the above diagnoses from EOO . Patients presenting with localized EOO have traditionally been treated with surgical resection and chemotherapy. Radiation has been used in the past with little success, and is now used sparingly for palliation . PatientThe overall prognosis for patients with EOO remains poor. Only about 1/3 of patients will survive for a long term. Many patients will have local recurrence and most will die of metastatic disease in 2-3 years . Those p


Multiple Osteochondromas (MO) is a disease of benign bony growths with a low incidence of malignant transformation. Secondary chondrosarcoma in children is rare even in children with MO. Making a diagnosis of malignancy in low-grade cartilage tumors is challenging and requires consideration of clinical, radiographic, and histopathological factors. We report two cases of skeletally immature patients with MO who presented with rapidly enlarging and radiographically aggressive lesions consistent with malignant transformation. Both underwent allograft reconstruction of the involved site with no signs of recurrence or metastatic disease at a minimum of four-year follow-up. Multiple Osteochondromas (MO) is an autosomal dominant disease of benign osseous tumors occurring primarily in the metaphyseal regions of the appendicular long bones and the flat bones of the axial skeleton. The risk of malignant progression has been estimated to range from 1 to 25% of patients with MO , though Approximately 80% of patients with MO have an identifiable mutation in one of two genes, EXT1 and EXT2, on chromosomes 8 and 11, respectively –21. ThesDiagnosing sarcomatous progression of an osteochondroma is challenging, as the histology may reveal only subtle changes of malignancy , 27. AddWe report two pediatric patients with MO with sarcomatous progression to illustrate the challenges in differentiating benign osteochondroma from chondrosarcoma.An otherwise healthy 11-year-old male with a family history of MO and multiple osteochondromas presented with a nine-month history of mild pain and a worsening limp, with two months of a palpably enlarging pelvic osteochondroma. There was no prior history of malignancy in the patient or his affected family members. Imaging studies revealed a 10 × 12 × 12 cm sessile osteochondroma on the left posterior ilium of the pelvis. Much of the lesion consisted of a 5-6 cm thick cartilaginous cap, metabolically active as seen by uptake of gadolinium on T2 images in a nodular  or ring Partial resection of the ilium was performed with an intercalary pelvic allograft reconstruction. Microscopic examination revealed a well-differentiated chondroid neoplasm composed of hyaline cartilage with diffuse myxoid degeneration and areas of tumor necrosis, consistent with a low-grade chondrosarcoma arising from an osteochondroma. The areas considered to have progressed cytologically deviated from areas of the lesion that had features of an osteochondroma . Invasiomax of 1.6 in the inferior portion of the lesion.A healthy 13-year-old male with a family history of MO and multiple osteochondromas first noted in infancy presented to an outside institution with a six-month history of a painless, enlarging osteochondroma of the left distal femur. The preoperative radiographic appearance of this mass demonstrated a 10 × 10 × 7 cm lesion with a sclerotic margin at the distal femoral diaphysis displaying obvious growth, with an apparent 50% increase in volume over the preceding six months, including invasion of the cortex of the stalk of a nearby osteochondroma . Due to A distal femoral resection and intercalary allograft reconstruction were performed. At the time of resection, a 10 cm posterior distal femoral lesion with a large cartilaginous cap (3.5 cm in thickness) was excised. Histopathologic examination revealed a well-differentiated chondroid neoplasm composed of abundant hypercellular nodules of hyaline cartilage consistent with a low-grade chondrosarcoma arising from an osteochondroma , similarThese two cases of chondrosarcomatous progression of an osteochondroma in a skeletally immature patient reflect an unusual and rarely reported complication of MO. Clinically, the symptoms of malignant progression in adults are typically subtle with insidious onset followed by a period of rapid growth of the tumor mass. Although pain, swelling, and enlargement of the lesion may be hallmarks of malignant progression in adults, none are specific symptoms in the growing child , 10, 34.Histologic features of sarcomatous progression of cartilage neoplasms have traditionally included atypical, plump, or double-nucleate chondrocytes, and hypercellularity of tissues . As low-max of 2.0 has been reported as the cut-off above which chondrosarcomatous progression of an osteochondroma has likely occurred, though lesions with an SUVmax as low as 1.3 have been found in Grade I chondrosarcomas [The radiographic features of malignant transformation include loss of distinct and regular margins at the periphery of the lesion, cortical erosion of the osseous stalk or base of the lesion, and irregular or punctate calcifications in an enlarged cartilage cap , 33, 39.sarcomas , 41. Obtsarcomas .The slow growth and low metastatic potential of secondary peripheral chondrosarcomas such as these suggest the need for a cautious approach to treatment. Biopsy, which is often difficult to interpret, followed by excision may lead to unnecessary additional surgery for the majority of these lesions that are likely to be benign. For those biopsied lesions that prove to be malignant, simple osteochondroma excision may be intralesional, with a higher risk of recurrence as well as progressive tumor dedifferentiation and metastases over time . CarefulNumerous studies describe a 10%–25% risk of malignant progression in MO. However, these represent select populations of patients who have undergone surgical resection of benign or malignant osteochondromas, likely resulting in an overestimation of the rate of malignancy. Clinically based studies report much lower rates of malignant progression, generally ranging from 0% to 5% , 6, 11. These two pediatric cases of chondrosarcoma in children with MO presented with grossly enlarging osteochondromas. Both patients had lesions with cartilage caps greater than 35 mm with punctate calcifications. While there is no gold standard imaging study to establish the presence of malignancy in adults or children, serial radiographic imaging is warranted for all large osteochondromas, particularly those in high-risk locations . Routine use of CT and MRI scans should be employed for cases of suspected malignancy to identify cortical invasion by tumor and thickness and metabolic activity of cartilage caps. With thorough evaluation and preoperative planning, wide excision and limb-sparing surgery can provide effective treatment, if necessary , 28. The


There are several surgical complications which can occur following simultaneous pancreas-kidney transplantation (SPKT). Although intestinal obstruction is known to be a common complication after any type of abdominal surgery, the occurrence of small bowel volvulus, which is one of the rare causes of intestinal obstruction, following SPKT has not been published before. A 24-year-old woman suffering from type I diabetes mellitus with complications of nephropathy resulting in end stage renal disease (ESRD), neuropathy and retinopathy underwent SPKT. On the postoperative month 5, she was brought to the emergency service due to abdominal distention with mild abdominal pain. After laboratory research and diagnostic radiological tests had been carried out, she underwent exploratory laparotomy to determine the pathology for acute abdominal symptoms. Intra-operative observation revealed the presence of an almost totally ischemic small bowel which had occurred due to clockwise rotation of the mesentery. Initially, simple derotation was performed to avoid intestinal resection because of her risky condition, particularly for short bowel syndrome, and subsequent intestinal response was favorable. Thus, surgical treatment was successfully employed to solve the problem without any resection procedure. The patient's postoperative follow-up was uneventful and she was discharged from hospital on postoperative day 7. According to our clinical viewpoint, this study emphasizes that if there is even just a suspicion of acute abdominal problem in a patient with SPKT, surgical intervention should be promptly performed to avoid any irreversible result and to achieve a positive outcome. Small bowel volvulus (SBV) is rarely encountered in adults. SBV generally occurs in a normal abdominal cavity and its etiology is still not clear. There are two categories of small bowel volvulus: a) primary small-bowel volvulus, in which there are no predisposing anatomical abnormalities, and b) secondary small-bowel volvulus, in which an acquired or a congenital abnormality causes rotation of the bowel . There a3/mm3), high serum lactate dehydrogenase (852 U/L) and high D-dimer (532 mcg/L) levels. A plain upright radiography of the abdomen was found to be non-specific. Abdominal ultrasonography indicating dilatation and edema of the small intestine, and abdominal tomography were then performed. Both revealed minor intestinal dilatation without determining the primary cause. Seventeen hours after admission to the hospital, we performed exploratory laparotomy. During the operation, we observed that almost the entire small intestine was gangrenous, but only 40 cm at the proximal part had adequate blood supply. The distal segment of the small intestine seemed to be ischemic due to clockwise rotation of the mesentery  declare that they have no competing interests.UA helped perform the operation and was involved in drafting the manuscript and in critical content revision.PY was involved in drafting the manuscript and in critical content revision.HT made substantial contributions to the study's concept and designCH made substantial contributions to the study's concept and gave the final approval of the version to be publishedAC  was involved in critical content revision and gave the final approval of the version to be publishedAll authors read and approved the final manuscript


Fetus in fetu is a rare abnormality secondary to the abnormal embryogenesis in a diamniotic, monochorionic pregnancy. It is a rare pathological condition and fewer than 100 cases have been reported in the literature.A 2 month old girl with an abdominal mass since birth, was referred to the Cancer Institute with a suspected diagnosis of a Wilms' tumor. Conventional radiograph of the abdomen revealed a mass containing numerous calcifications. CT scan showed a heterogeneous retroperitoneal mass containing well-defined calcified structures. The decision was made to recommend surgical exploration and the mass was successfully excised. Physical examination of the mass with review of literature confirmed the diagnosis of fetus in fetu.Although it is a rare condition imaging may play an important role in the correct prospective diagnosis of fetus in fetu. Surgical excision is the recommended treatment. There is controversy as to whether fetus in fetu is a distinct entity or represents a highly organized teratoma. A teratoma is defined as a neoplasm with a slight potential for malignancy that is composed of multiple tissues foreign to the part in which they are located [However, many other investigators suggest that fetus in fetu is a pathologic entity that is distinct from teratoma for several reasons ; MalignaA final important feature that has been used to distinguish between fetus in fetu and teratoma is the presence of a vertebral column. Willis  emphasizIn case reports in the medical literature up to 1980 preoperative diagnosis of fetus in fetu was made in only 16.7% of cases because CT scan was not performed. Since that time CT scan has proven very helpful in suggesting the preoperative diagnosis . The difIn our case imaging did not play an important role in our ability to make a preoperative diagnosis.Fetus in fetu is a rare and interesting entity that typically presents in infancy or early childhood. Current imaging modalities may allow us to accurately diagnose the condition before surgery. Complete excision is curative and allows confirmation of the diagnosis.The author(s) declare that they have no competing interests.NMK: Examined the patient, admitted her, ordered the investigations and then referred her to the surgery department and participated in writing the drafts, DWM Carried out the surgery, AAA-E: followed up the patient's condition and survival after surgery, wrote the drafts and the final manuscript and kept track of the patient records and investigations. All authors read and approved the final manuscript.Written informed consent was obtained from the parents of our 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.


In this case we report a 23 -year-old primigravida with 30 weeks presenting with torsion of the ovarian cyst. She presented to the antenatal clinic with acute pain abdomen. She was diagnosed to have torsion of ovarian cyst during pregnancy and a cystecomy was carried out. Her histopathology report showed a benign serous cystadenoma. Her pregnancy was followed up. She delivered a healthy female baby at term. Although the safety of antepartum surgical intervention has been accepted, abdominal surgery nevertheless carries some risks to a pregnant woman and unborn fetus, and so the choice of management necessitates a weighing of risks based on characterization of the adnexal mass and gestational age. Torsion of ovary is the total or partial rotation of the adnexa around its vascular axis or pedicle. Moderate size, free mobility and long pedicle are predisposing factors. The exact etiology is obscure. Most commonly seen are dermoid and serous cystadenomas. Complete torsion causes venous and lymphatic blockade leading to stasis and venous congestion, haemorrhage and necrosis. The cyst becomes tense and may rupture. Patient usually presents with acute severe pain abdomen and pelvic examination may reveal a tender cystic mass separate from the uterus. The risk of ovarian torsion rises by 5 fold during pregnancy. Incidence is 5 per 10,000 pregnancies.23 year old primigravida presented to the antenatal clinic with 7 months amenorrhoea and pain abdomen and 3 episodes of vomitings, since one day. Her menstrual cycles were regular. She described the pain as sharp non-radiating type of pain in the right iliac fossa with sudden onset, with no relieving factors. She gave no history of vaginal bleeding or discharge. There was no history of diarrhoea, constipation, fever, urinary complaints or any recent illness. She conceived spontaneously. She had regular antenatal checkups. Her first & second trimesters were uneventful. No significant past medical and surgical history noted.On examination, patient was conscious, coherent with pulse 82/min, blood pressure 130/80 mm of hg, temperature normal, cardiovascular and respiratory systems normal. Abdominal examination revealed fundal height corresponding to 30 weeks gestation. Uterus was irritable. There was a single fetus in longitudinal lie with breech presentation. Fetal heart rate was good & regular. There was severe tenderness in right iliac fossa. On vaginal examination, cervix was posterior, 50% effaced, 2 cm dilated, and breech presentation.All her blood and urine investigations were within normal limits. Ultrasonography revealed a 10 × 5 cm single anechoic cystic lesion in right iliac fossa with single thin septation and no solid components. It also showed a single intrauterine live fetus in longitudinal lie with breech presentation of 30 wks gestational age. Estimated fetal weight-1629 gms, Amniotic fluid adequate, placental position posterior upper segment with grade 2 maturity. No evidence of free fluid in the abdomen.MRI showed extended breech presentation of the fetus and a large hyper intense mass lesion on the right side of the abdomen, outside the uterus measuring 10.4 × 5.0 cms suggestive of right ovarian cyst. Figure With the provisional diagnosis of twisted ovarian cyst, emergency laparotomy was done under regional anaesthesia. A 10 × 5 cm right ovarian cyst was found to be twisted around its pedicle by 3 rotations. After untwisting, cystectomy was done by carefully enucleating the cyst and separating it from the capsule. The cyst was sent for histopathological examination. (Figure th post operative day. Her histopathology report showed benign serous cystadenoma of the ovary. She was followed up, her pregnancy continued unremarkably and she delivered an alive female baby of birth weight 2.75 kg at term gestation by caesarean section.Patient recovered with an uneventful postoperative period and was discharged on 9The commonest type of ovarian tumours encountered in pregnancy are cystic teratoma, paraovarian cyst, serous cystadenoma, corpus luteal cysts, luteomas etc. Serous Differential diagnosis includes: uterine leiomyomas, non preganant horn of bicornuate uterus, appendiceal abscess, diverticular abscess, pelvic kidney, retroperitoneal tumours, ectopic pregnancy and retroverted gravid uterus.Complications of the cysts associated with pregnancy are torsion of the cyst, rupture, infection, malignancy, impaction of cyst in pelvis causing retention of urine, obstructed labour and malpresentations of the fetus. Some stCysts less than 6 centimetres in diameter and appearing benign on ultrasound are generally treated conservatively as they may undergo spontaneous resolution. Corpus luteal cysts regress by 12 to 16 weeks. Cysts more than 10 centimetres in size are usually resected due to increased risk of malignancy, rupture or torsion. Management of cysts between 5 to 10 centimetres is controversial. If the cysts contain septae, nodules, papillary excrescences or solid components then resection is recommended. Those with simple cystic appearance may be managed expectantly with serial ultrasound surveillance. However they may require emergency exploratory laparotomy for rupture, torsion or infarction in as many as 50% cases . With thIf the ovarian cyst is diagnosed in the first trimester, it is better to wait till 16 wks when the implantation of pregnancy is more secure and also the cyst may disappear spontaneously. Persisting tumours are treated by cystectomy or ovariotomy as indicated. Ovarian tumour or cyst can be easily removed till 28 wks of gestation thereafter it is not readily accessible and may precipitate preterm labour. Ovarian cyst which ruptures, or undergoes torsion or if it shows evidence of malignancy, requires immediate surgery, irrespective of the period of gestation .A simple cystectomy can be performed in the absence of overt malignancy. Previously untwisting of the pedicle was avoided to prevent emboli and toxic substances related to hypoxia, from entering peripheral circulation. but recently, re-establishing ovarian circulation by untwisting, has shown to result in viable ovarian tissue with no systemic complications.Ovarian torsion is relatively uncommon in the second trimester of pregnancy. Diagnosis can usually be made on the basis of the characteristic clinical presentation in conjunction with ultrasound evidence of a unilaterally enlarged adnexal mass. Treatment options are limited to surgery, either by laparoscopy or laparotomy, but the former becomes more difficult after second trimester.Written informed consent was obtained from the patient for publication of this case report. 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.The case was managed and operated by RG, VK, and VVS. The authors have read and approved the final manuscript.


Although gastrointestinal haemorrhage from aortoduodenal fistulae secondary to previous aortic grafts are well known, a primary fistula from an aortic aneurysm is a rare consideration resulting in inappropriate management and poor outcomes.We report a previously fit 65-year-old Sri Lankan man who presented with severe anaemia , recent onset low backache. There was no history of analgesic abuse, peptic ulceration, alcohol excess, weight loss or malena. The abdomen was soft and there was no visceromegaly. A routine ultrasound detected an abdominal aortic aneurysm without signs of a leak. Two days later, while undergoing routine diagnostic tests for anaemia and backache, he had a massive haematemesis. Standard resuscitation was commenced with hope that common sources, either peptic ulcers or varicies would eventually stop bleeding enabling endoscopy and definitive treatment. However, persistent hypotension coupled with the clinical suspicion of an aortoduodenal fistula led to immediate surgical exploration rather than continued aggressive resuscitation. An aortoduodenal fistula was confirmed and both the duodenum and the aorta were successfully repaired by direct suture and synthetic graft replacement respectively. This man remains well nine months later.Gastrointestinal bleeding in the presence of an ‘asymptomatic’ abdominal aortic aneurysms should be assumed to be from a primary aortoduodenal fistula unless another source can be identified with certainty without delay. Abdominal aortic aneurysms (AAA) and haematemesis are common clinical problems which are expected to increase as the population ages. An AAA is usually detected either incidentally on routine palpation and ultrasound examinations or when complicated with pain or extraperitoneal rupture. Haematemesis is commonly from gastro-oesophageal varicies or ulceration. Nevertheless, rupture of an AAA in to the duodenum, an aortoduodenal fistula (ADF) is a rare complication of an AAA  and haem® tube graft, and the duodenum was closed in two layers. An omentopexy was performed between the aorta-graft anastomosis and duodenum. The patient made an uneventful recovery. Although intraoperative bacterial cultures were negative antimicrobial prophylaxis with co-trimoxazole was prescribed for two weeks. Nine months later, he remains well with a haemoglobin level of 12.5 gm/dl.We report a 65-year-old Sri Lankan man who was hospitalised with recent onset low backache and severe pallor . He had been previously well and there was no history of ingestion of aspirin or non-steroidal anti-inflammatory drugs, alcohol abuse, chronic abdominal pain, weight loss, change in bowel habits or malena. He had given up tobacco smoking five years back. He was not on anticoagulants. His abdomen was soft and there was no visceromegaly. Investigations were being arranged to determine a cause for his anaemia and backache and a routine abdominal ultrasound detected a 6 × 7 cm abdominal aortic aneurysm with no signs of a leak. On the second day after hospitalisation he had a massive haematemesis and became hypotensive (systolic blood pressure 70 mm Hg). The emergency care team commenced resuscitation with blood and considered common causes such as gastro-oesophageal varicies and ulcers. However, the poor initial response to fluids and clinical suspicion of an ADF led us to hold back continued aggressive resuscitation until the source was controlled at surgery. At laparotomy gastro-oesophageal varicies and chronic ulceration were ruled out. The third part of the duodenum was densely adherent to the right lower lateral wall of the aneurysm and there was no evidence of overt sepsis .. The anThe need to consider an ADF as the cause of upper gastrointestinal bleeding in a patient with a previous aortic anastomosis is well established . InteresThe pattern of bleeding from an ADF is of interest. A “herald” haemorrhage, which may be occult or mild, is followed hours, days, or weeks later by catastrophic haemorrhage that is characteristic of an ADF. This initial slow bleed is the result of a small fistula occluded by thrombus. Since 70% survive at least 6 hours  and up tSurgical repair without delay is the only chance for survival . SurgeryEven though primary ADF is a rare cause of enteric bleeding, what makes it important is the high mortality from lack of awareness and non diagnosis. The key to its successful management is a high degree of clinical suspicion when there is gastrointestinal bleeding in combination with an AAA. We recommend that in such instances bleeding must be assumed to be from an ADF unless another source can be identified with certainty. The search for other causes is advisable only in normotensive patients while hypotensive patients should be referred to vascular surgeons to be operated on without further delay.


Nodular fasciitis is a rapidly growing mass, with high cellularity and mitotic activity, that can be both clinically and histologically misdiagnosed as a soft tissue sarcoma. Nodular fasciitis of the hand is an extremely rare condition. We report a 17-year-old male hand-ball player with nodular fasciitis in the dominant hand. The patient presented with a rapidly growing mass in his right hand and no history of major trauma. On physical examination, a painful mass measuring 2 cm in diameter was observed in the first web space. Magnetic resonance imaging (MRI) demonstrated a subcutaneous mass with isointensity on T1-weighted images and inhomogeneous high intensity on T2-weighted images. The lesion was inhomogeneously enhanced after intravenous administration of gadolinium. Moreover, thallium-201 scintigraphy showed high uptake at the early phase and no wash-out at the delayed phase. We performed an excisional biopsy. The mass was present subcutaneously and adhered to the interosseous muscle fascia. Although a pathological examination by frozen section during surgery showed a low-grade spindle cell sarcoma, the final histological diagnosis was nodular fasciitis. There was no evidence of local recurrence at the recent follow-up 2 years after the operation. We speculate that repeated small injuries as a result of sports activities played an important causative role in the nodular fasciitis. Nodular fasciitis is a rapidly growing benign, self-limiting, reactive lesion, which due to clinical findings and pathologic appearance can be mistaken for a soft tissue sarcoma. This lesion is commonly found in the forearm and is extremely rare in the hand. The precise cause of nodular fasciitis is unknown, but despite a benign clinical behavior injury or infection is possible.We present a case of nodular fasciitis in the hand of a hand-ball player. In our patient, thallium-201 scintigraphy showed high uptake at the early phase and no wash-out at the delayed phase in a so-called malignant pattern. These scintigraphy findings have not been previously reported in nodular fasciitis. To our knowledge, this is the first report of nodular fasciitis occurring in the hand of an athlete.A 17-year-old male high school hand-ball player noticed an approximately 1-cm mass in the first web space of his dominant right hand 1 month before presentation at our hospital. He sought medical attention because the mass became rapidly and painfully enlarged, doubling during the 4-week interval from first observation. He belonged to a high school hand-ball club and played every day. He had not suffered any infection or major trauma, and there was no relevant family history. On physical examination at our hospital, a painful mass measuring 2 cm in diameter was observed in the first web space. The mass was mobile, elastic-hard, and tender to palpation with a smooth surface and clear margin. No local heat and redness on the mass were present. Neurovascular examinations including Tinel's sign were normal. His hematological profile was unremarkable. Radiographs of the right hand showed an increased soft tissue shadow in the first web space . On compEleven days after the first visit, the mass was even more enlarged measuring 25 mm in diameter. We performed an excision biopsy . A well Nodular fasciitis is a benign reactive lesion first reported by Konwaler et al. in 1955 as a subcutaneous pseudosarcomatous fibromatosis ,3. It isThe macroscopic appearance of nodular fasciitis is solitary round to oval nodules, well circumscribed, and usually measuring less than 2 cm in diameter; only 8% are larger than 4 cm . NodularThe features of nodular fasciitis on MR imaging are non-specific. The lesions appear as well circumscribed, round to oval masses . LiteratHistologically, the lesion is composed of plump but regular spindle-shaped fibroblasts or myofibroblasts lacking nuclear hyperchromasia and pleomorphism. The lesion may be highly cellular. Although mitotic figures are fairly common, atypical mitoses are almost never seen. Five important histologic features of nodular fasciitis aid in diagnosis, including spindle-shaped fibroblasts, clefts separating the fibroblasts, extravasated erythrocytes, interstitial mucoid material, and a loosely textured ‘feathery’ pattern of the mucopolysaccharide ground substance . ImmunosThe current report documents a rare case of nodular fasciitis in the hand. Particularly, nodular fasciitis in the hand of a sports player has never been reported. Repeated minor injury to the hand during catching or throwing the ball may have caused the nodular fasciitis. Nodular fasciitis should be added to the differential diagnosis of ball game players who present with a soft-tissue mass in the hand.