Lipomas of the colon are benign tumors that rarely occur. Their size ranges from 2 mm to several cm. They are usually asymptomatic but occasionally they present with clinical manifestations depending on tumor size, localization and complications, which often lead to diagnostic difficulty. A 40-year-old man presented with massive rectal haemorrhage. During colonoscopy a giant polyp of over 50 mm in its bigger diameter, with a thick stalk of 2 cm, located in the transverse colon, was revealed. Endoscopic resection was performed with success. Histologic examination demonstrated a giant lipoma. In this report discussion over endoscopic resection of colonic lipomas mimicking neoplasms is also performed. Lipomas of the colon are benign tumors with extremely low malignant potential that occur rarely. They are the second most common benign colonic tumor after adenomatous polyps . AlthougWe report here a case of a giant colonic lipoma with the endoscopic appearance of neoplasmatic polyp, successfully removed in a single piece.The patient reported more than 15 loose bowel movements mixed with blood during last 24 hours. Physical examination was unremarkable. The patient denied any changes in bowel habits frequency with an average of 1-2 bowel movements per day. He reported no previous medical history and took no medications. Results of laboratory investigation including carcinoembryonic antigen (CEA) were within normal limits. Colonoscopy revealed a large polyp (>5 cm diameter) in the transverse colon  was needed to ensnare the lesion near its base Figure . SubsequThis was the seventh case of giant colonic lipoma in our department and the second in this series initially presenting with massive hemorrhage. In the Mayo Clinic series, 46% of large-bowel lipomas were discovered incidentally in specimens removed for other diseases, 11% were resected because of a neoplasm suspected of being a carcinoma, and 6% were symptomatic . The lesPathologically, lipomas are well-differentiated tumors arising from deposits of adipose connective tissue in the bowel. Sarcomatous changes, in colonic lipomas have not been reported, but intermittent torsion and relative ischemia can give rise to a pseudomalignant appearance. The greatest clinical significance of lipomas lies in their potential to be confused with adenomatous polyps or other aggressive pathology . DespiteThe decision whether to remove lipomas and which method is the best option, either endoscopically or surgically remains controversial. Because the majority of lipomas are submucosal, endoscopic removal entails high risk of morbidity due to perforation compared with adenomatous polyps, since its high water content requires a tremendous amount of heat to cut through the lipoma. Endoscopic polypectomy of large lipomas is difficult, although possible in selected cases  as it both is reported in the literature ,3,6,8-1 and obseIn conclusion, it should be noted that colon lipomas, although rare, should be considered in the differential diagnosis of large bowel tumors. Endoscopic approach remains a safe and effective option for giant lipoma resection, provided each case is selected carefully and procedures are performed by skilled endoscopists in centres with experience.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The author(s) declare that they have no competing interests.GL is a senior gastroenterologist in our department, performed the polypectomy, and was the major contributor in writing the manuscript. DT collected the patient's data and assisted in the procedure. PX performed the histological examination of the polyp, and was a major contributor in writing the manuscript. AN was responsible for all the artwork seen in the manuscript. IP contributed in writing the manuscript. IK is the head of the department and revised the manuscript.


An 11-year-old girl presented to our department to have a second opinion regarding a lesion involving her left mandible. She had previously undergone several radiographic exams including panoramic, helical, and cone-beam computed tomography. Radiographic examinations revealed a well-defined radiolucent region, which contained an irregular radiopaque mass of 3 cm in diameter, localized to the left angle of the mandible. Our presumptive diagnosis was complex odontoma. Excisional biopsy was performed, and microscopic features showed strands and islands of odontogenic epithelium showing peripheral palisading and loosely arranged central cells, identical to stellate reticulum, embedded in a myxoid cell-rich stroma resembling the dental papilla. Dentin and enamel were also presented. The diagnosis was ameloblastic fibro-odontoma, which is a rare mixed odontogenic tumor, derived from epithelial and ectomesenchymal elements that form the dental tissues. Ameloblastic fibro-odontoma (AFO) is a benign, slow growing, expansile epithelial odontogenic tumor with odontogenic mesenchyme. It may inhibit tooth eruption or displace involved teeth although teeth in the affected area are vital –3. RadioMicroscopically, the lesion is composed of strands, cords, and islands of odontogenic epithelium embedded in a cell-rich primitive ectomesenchyme, resembling the dental papilla. Many authors reported that AFO is not aggressive and can be treated adequately through a surgical curettage to the lesion without removal of the adjacent teeth , 5, 7, 8An 11-year-old girl presented to our department on referral from another dentist to have a second opinion about a lesion involving the left mandible. She had radiographic examinations, including panoramic, helical, and cone-beam computed tomography. These examinations were accompanied by a presumptive radiographic differential diagnosis of “odontoameloblastoma”: complex odontoma and AFO. The medical, social and family histories were unremarkable, as were the results of a review of systems and a physical examination. The clinical examination did not display any sign of pain or swelling in the left mandible. There was no history of local trauma or infection. Oral inspection revealed good oral hygiene. The initial panoramic radiography revealed a well-defined radiolucent region, which contained an irregular radiopaque mass 3 cm in diameter. This lesion occupied a zone from the lower left second molar area to the left ramus. The mandibular left second molar was not present .Helical and cone-beam computed tomography showed an expansile well-circumscribed lesion containing at the interior a calcified mass compatible with odontogenic tissue Figures and 3. Considering the clinical and radiographic examinations, our presumptive diagnosis was complex odontoma.The patient underwent enucleation of the lesion and careful curettage of the surgical cavity under general anesthesia. The surgical specimen was fixed in neutral buffered 10% formalin and subjected to pathological analysis. Light microscopic examination of sections stained with hematoxylin and eosin revealed strands and islands of odontogenic epithelium showing peripheral palisading and loosely arranged central cells, identical to stellate reticulum, embedded in a myxoid cell-rich stroma resembling the dental papilla . Dentin In the present case, the patient presented to our department with previous examinations, including panoramic, helical and cone-beam computed tomography. While these radiographic examintions were given a presumptive diagnosis of odontoameloblastoma by the examinaing radiologists, we believed that the findings were more common in this region. Odontoameloblastoma, also known as ameloblastic odontoma, has a more aggressive behavior, similar to an ameloblastoma rather than an odontoma . The histogenesis of this lesion is controversial. AFO is a benign tumor that exhibits the same benign biologic behavior as that of ameloblastic fibroma, showing inductive changes that lead to the formation of both dentin and enamel . This isAccording to the revised World Health Organization (WHO) classification , ameloblAFO is relatively rare, with the prevalence among oral biopsies being about 1%  and its Many authors reported that AFO can be treated adequately through a surgical curettage without removal of the adjacent teeth , 5, 7, 8


Schwannoma, also referred to as neurilemmoma, is a benign tumor of peripheral nerve arising from Schwann cells that form the neural sheath. Schwannoma of ophthalmic interest is rare although it has been reported in relation with the orbit, and less frequently with the uveal tract and conjunctiva. Isolated eyelid schwannoma is extremely uncommon. Up until now, only eight cases have been reported in literature. Herein, we report two cases of eyelid schwannoma. Schwannoma is a benign tumor arising from Schwann cells of peripheral nerves. The tumor is normally solitary, smooth-surfaced, slow-growing and generally asymptomatic. It may develop at any age and there is no gender predilection. Head and neck are one of the most frequent localizations. Ocular tA 47-year-old man was referred to us with a one-year history of a slowly enlarging, painless nodule on his right lower eyelid. Ocular examination was normal except for the presence of a firm, non-tender nodule measuring 7 × 7 × 6mm in the medial third of the right lower eyelid. There were no clinical findings indicative of neurofibromatosis. The patient was operated under local anesthesia; tumor was completely removed via a horizontal eyelid crease incision. The postoperative course was uneventful and the patient was asymptomatic five years later.Pathological studies showed an encapsulated spheric mass (6mm in diameter) on macroscopic examination. Microscopic examination revealed a biphasic pattern, with highly cellular areas of non-pigmented spindle cells, containing elongated nuclei arranged in fascicles, which were separated by homogenous acellular material (Antoni A pattern). In other areas, the cells were more oval, and had rounded nuclei, clear cytoplasm and less basement membrane, and were loosely entwined within a clear myxoid matrix (Antoni B pattern) . No histA 20-year-old woman was referred to us for a chalazion of her left lower eyelid, which had been operated three times in four years. No tissues had been submitted for histopathological examination in earlier surgical procedures. Ocular examination was normal except for the presence of a firm, non-tender painless nodule measuring 13 × 8 × 8mm located in the middle of the left lower eyelid, independent of the lid margin . There wOn macroscopic examination the nodule was well-circumscribed, non-encapsulated, measuring 10mm in diameter with yellow appearance on cut sections. Microscopic examination revealed fascicules and bundles of non-pigmented spindle cells with elongated bland nuclei, and abundant extracellular collagen with adipocytes and squamous cells displaying an Antoni A pattern. There was no mitotic activity. Immunochemistry for S-100 protein was strongly positive, but tumor cells did not react with HMB45 . Final dSchwannoma (or neurilemmoma) is made up of proliferating Schwann cells of peripheral nerve sheaths. It is a neoplasm which occurs wherever schwann cells are present, that is in any myelinated peripheral nerve. In most cases, while schwannoma is sporadically manifested as a single benign neoplasm, the presence of multiple schwannomas in one patient is usually indicative of neurofibromatosis 2 (NF-2) . Howeveret al.[Our two patients had isolated eyelid schwannoma with no family history or clinical findings of NF-1 or NF-2. Schwannoma is well known to develop in the orbit, constituting 1% of the orbital tumors in a series by Rootman et al. Isolatedet al. Its occuet al.. In our et al. Age ranget al.. Clinicaet al. (our Caset al. To avoidPathologically, schwannomas classically show a mixture of two patterns, the Antoni Type A densely cellular pattern and the Antoni Type B edematous and disorganized pattern.The most important feature for diagnosis remains the strong reactivity to S100 protein in immunochemistry. NegativiThe rare occurrence of eyelid schwannoma should be kept in mind in the differential diagnosis of any solid palpebral lesion, especially in case of recurrent chalazion.When they are isolated, they are mostly benign. In neurofibromatosis they might rarely undergo malignant transformation.


Gossypiboma, an infrequent surgical complication, is a mass lesion due to a retained surgical sponge surrounded by foreign-body reaction. A 27-year-old lady presented with palpable abdominal mass five years after cesarean section. Retained foreign body was diagnosed radiologically and confirmed with operation. Retained foreign body should be in the differential diagnosis of any postoperative patient who presents with pain, infection, or palpable mass. Two usual responses lead to the detection of a retained sponge. The first type is an exudative inflammatory reaction with the formation of an abscess and usually leads to early detection and surgical removal. The second type is aseptic with a fibrotic reaction to the cotton material and development of a mass [A surgical sponge is the most common type of retained foreign body (RFB). The condition is sometimes called gossypiboma, derived from the Latin f a mass .In the abdomen the sponge can be surrounded by omentum and intestines, which attempt to encapsulate it. The exerted pressure and irritation on the bowel loops can lead to necrosis of the intestinal wall and the sponge erodes partially or entirely into the lumen of the bowel. This process can lead to obstruction or fistula. Patients develop symptoms of abdominal pain, nausea, vomiting, anorexia, and weight loss resulting from obstruction or a malabsorption type syndrome caused by the multiple intestinal fistulas or intraluminal bacterial overgrowth ,2.A 27-year-old lady presented with discomfort in periumbilical area since one month ago. The only positive point in her previous history was a cesarean section five years back. Vital signs were normal. On abdominal examination, a round mobile mass was palpable. All routine lab data were normal. Abdominal X-ray was in favor of retained sponge figure . CT scanThe possibility of a RFB should be in the differential diagnosis of any postoperative patient who presents with pain, infection, or palpable mass. The first diagnostic modality to rule out a RFB should be a CT scan and often it will be the only test needed. The CT findings of a sponge usually describe a rounded mass with a dense central part and an enhancing wall. Other features of retained sponges or towels include a whorl-like appearance with trapped air bubbles and cystic masses with infolded densities. MRI features can be confusing because the radiopaque marker is not magnetic or paramagnetic so is not visible .Clinicians usually think that the diagnosis of a RFB on an intraoperative radiograph is easy and obvious, but often this is not the case. Intraoperative radiographs can be of poor quality, especially in obese patients. Correctly identifying a sponge on a radiograph can be difficult. The surgical markers may become twisted or folded and present an unusual image . For insThe usual treatment of a RFB is removal. Reopening the previous operative site is one possibility, but endoscopic or laparoscopic approaches may be attempted .One possible complication during surgical removal of RFB is perforation of adherent bowels, which may be missed. We had another case with retained two surgical towels during emergency cesarean section. Her surgeon removed the towels through a small incision. However, she was admitted in our service three days later with clinical picture of generalized peritonitis. Explorative laparotomy revealed a missed small bowel perforation.In some instances the attempt to remove the retained foreign body may cause more harm than the item itself, although in these circumstances the foreign body is usually a needle or small part of a surgical item. In these cases, removal is not recommended. Rarely is this an appropriate course of action for a retained sponge, which should always be removed .New England Journal of Medicine published an article about risk factors of RFBs. Of the 8 risk factors the authors identified , volume of blood loss, female sex, and surgical counts) only 3 were found to be statistically significant by multivariate logistic regression. The 3 significant risk factors were emergency surgery, unplanned change in the operation, and BMI. The counting of sponges and instruments was not a significant predictor in the multivariate model. Although all 3 factors were significant, the 9-fold increase in risk associated with emergency surgery was impressive. In addition, in 88% of the cases where there was a RFB and counts were performed, the counts were falsely called correct. The authors recommended "radiographic screening" at the end of high risk cases as a possible adjunct to improve detection of RFB [Recently, n of RFB . Surgeonn of RFB .New technologies are being developed that will hopefully decrease the incidence of RFB. An electronic article surveillance system has been examined which uses a tagged surgical sponge that can be identified electronically . Bar codRFB should be considered in the differential diagnosis of any postoperative patient who presents with pain, infection, or palpable mass. Identifying a sponge on an intraoperative radiograph is difficult. The best diagnostic modality to rule out a RFB should be a CT scan. One possible complication during surgical removal of RFB is missed perforation of adherent bowels.RFB: Retained foreign body; BMI: Body mass index.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The author declares that they have no competing interests.AA managed the patient and prepared the manuscript.


Liver hemangiomas are the most common benign liver tumors, usually small in size and requiring no treatment. Giant hemangiomas complicated with consumptive coagulopathy (Kasabach-Merritt syndrome) or causing severe incapacitating symptoms, however, are generally considered an absolute indication for surgical resection. Here, we present the case of a giant hemangioma, which was, to the best of our knowledge, one of the largest ever reported.A 38-year-old Asian man was referred to our hospital with complaints of severe abdominal distension and pancytopenia. Examinations at the first visit revealed a right liver hemangioma occupying the abdominal cavity, protruding into the right diaphragm up to the right thoracic cavity and extending down to the pelvic cavity, with a maximum diameter of 43 cm, complicated with "asymptomatic" Kasabach-Merritt syndrome. Based on the tumor size and the anatomic relationship between the tumor and hepatic vena cava, primary resection seemed difficult and dangerous, leading us to first perform transcatheter arterial embolization to reduce the tumor volume and to ensure the safety of future resection. The tumor volume was significantly decreased by two successive transcatheter arterial embolizations, and a conventional right trisectorectomy was then performed without difficulty to resect the tumor.To date, there have been several reports of aggressive surgical treatments, including extra-corporeal hepatic resection and liver transplantation, for huge hemangiomas like the present case, but because of its benign nature, every effort should be made to avoid life-threatening surgical stress for patients. Our experience demonstrates that a pre-operative arterial embolization may effectively enable the resection of large hemangiomas. Liver hemangiomas are the most common benign liver tumors, with a prevalence of 5 to 20%. Most hemangiomas are small and require no treatment or only follow-up. However, giant hemangiomas, having a diameter of more than 4 cm or 5 cm, may give rise to mechanical complaints or coagulopathy requiring intervention . The indHere, we report a case of a single huge hemangioma, completely resected by a right trisectorectomy following two successive TAEs, by which the volume of the hemangioma was significantly reduced.A 38-year-old Asian man was referred to our hospital with complaints of severe abdominal distension and pancytopenia. His past or family medical history was unremarkable. Although his abdominal bloating was impairing his daily life, he had not visited a healthcare facility or undergone treatment for abdominal distension.6/μL, a hemoglobin level of 10.3 g/dL, and a platelet count of 9.2 × 104/μL. The results of liver function tests were normal, including a total bilirubin level of 1.3 mg/dL, an albumin level of 4.3 mg/dL, and an indocyanine green retention rate at 15 min of 8.7%. "Asymptomatic" Kasabach-Merritt syndrome was apparent, however, based on a high international normalized ratio of prothrombin time of 1.46, a decreased fibrinogen level of 82 mg/dL, elevated fibrin degradation products (FDP) of 80 μg/mL, and D-dimer levels of 32 μg/mL.A routine blood count revealed pancytopenia, with a white blood cell count of 2600/μL, a red blood cell count of 3.42 × 10Multi-detector computed tomography (MDCT) on admission revealed a huge hemangioma located on the right liver, and replacing the parenchyma of the right liver and the left para-median sector. The hemangioma occupied almost the entire abdominal cavity, protruding into the right diaphragm up to the right thoracic cavity and extending down to the pelvic cavity, with a maximum diameter of 43 cm Figure . AngiogrBased on the liver function tests, remnant liver volume, and anatomic considerations, urgent primary tumor resection seemed possible, but because of the benign nature of the disease and our patient's stable condition, we decided to perform TAE to reduce the tumor volume before performing the resection to ensure the safety of the future radical resection of the tumor.First, TAE was performed for the right hepatic artery with coils and gelfoam. Thereafter, the tumor volume, anatomical positions, and recanalizations were calculated and investigated by dynamic MDCT once a month, not to misjudge the operation timing. Three months after the first TAE, the tumor volume had decreased to 10,290 mL. Angiography performed at that time revealed a collateral feeding artery from the right subphrenic artery, which was then embolized by TAE. Two months after the second TAE, the tumor volume had further decreased to 8260 mL based on MDCT volumetry Figure . No compThe remarkable volume reduction in the right upper portion of the tumor allowed for a safe approach to the hepatic veins and vena cava, and a radical resection of the tumor was performed by an anatomical right trisectorectomy. The operation was performed through a J-shaped skin incision using a ninth inter-costal thoracoabdominal approach Figure , and aftPathologic investigation of the specimen revealed a cavernous hemangioma, 30 × 25 × 15 cm, weighing 8100 g, comprising a spongy zone and a fibrotic scar zone with massive necrosis. Our patient's post-operative course was uneventful and he was discharged from the hospital 16 days after surgery. At 24 months following surgery, he enjoys an improved quality of life with normal liver function.Changes in the laboratory data and tumor volume are summarized in Figure Of the various treatment options for giant hemangiomas, surgical treatment, including resection and enucleation, provides the only consistently effective outcome with satisfactory results ,5. AlthoA recent major argument in the treatment of liver hemangiomas is the selection criteria for surgery; i.e. observation or operation, and enucleation or resection. Considering the benign and non-progressive nature of the disease, it is currently accepted that a giant hemangioma is not necessarily an indication for surgery just because of its size, and continued observation in asymptomatic patients or patients with minimal abdominal symptoms seems to be justified ,3. BecauHemostasis is important for resection of a giant hemangioma . The larIn the present case, we considered that an urgent resection at the first admission would be dangerous because it seemed impossible to approach the confluence of hepatic veins and inferior vena cava behind the tumor without extra-corporeal circulation. Liver transplantation was also considered an option, but based on the benign nature of this disease and the patient's stable condition, we considered these aggressive options a last resort and decided to first perform TAE to reduce the tumor volume. The timing for the radical surgery after TAE is another problem in the management of huge liver hemangioma. Considering the vascular recanalization and various complications after TAE which might result in the loss of opportunity or the difficulty of the radical resection, some authors recommend urgent operation after TAE ,12. FortWe report a case of a huge hemangioma, one of the largest tumors ever reported, that was successfully resected following effective TAE. The results in our case indicate the importance of pre-operative management to reduce tumor size.FDP: fibrin degradation products; MDCT: multi-detector computed tomography; TAE: transcatheter arterial embolization.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.AN was responsible for the management of this case. AN and SY were major contributors in writing the manuscript. All authors read and approved the final manuscript.


Giant lipomas are benign soft tissue tumours. They are found relatively rarely on the posterior part of the neck. Bleeding pressure ulcer in this giant tumour is a rare presentation. Surgical interventions in these tumours are very challenging because, sometimes, extension to the spinal cord and malignant change may occur, especially in old age. Knowledge of the anatomy and meticulous surgical techniques are needed for such giant lipomas. Giant lipomas are defined by Sanchez A 68-year-old male presented to our surgical outpatient department with a huge lump at the back of his neck for the last 11 years. There was an ulcer at the top of the lump for the last 9 months. Episodic bleeding was also reported from the ulcer for the last 6 months.On local examination of the neck, there was a 22 cm × 12 cm-sized swelling at the posterior side of the neck . It was Fine needle aspiration revealed mature lipocytes indicative of lipomatous lesion. Contrast Enhanced Computed Tomography Scan of the neck revealed a giant subcutaneous lipoma at the posterior triangle without septations. There was no communication with the spinal cord.th postoperative day.After intubation with general anaesthesia, the patient was positioned in a prone position. An elliptical transverse incision of 12 cm was given at around the base of the lump. The superior and inferior skin flaps were raised . SeparatHistopathological analysis of the resected mass revealed mature, proliferative lipocytes with no cellular atypia, and it was diagnosed as benign giant lipoma.Lipomas are the most common mesenchymal tumours. Only appComplete excision is the treatment of the choice for giant lipoma. Liposuction for such tumours has also been reported. Because giant lipomas usually have a well-defined pseudocapsule, dissection around these benign neoplasms is performed rather easily.In conclusion, giant lipoma in the neck region is rare and complications like bleeding decubiitus ulcer and malignant transformation are important complications. Before surgery, CT or MRI neck is an essential imaging study. Early surgical intervention is a must for these giant lipomas.


It plays a key role in detecting sites of occult disease and is widely utilized in the medical and surgical planning of such patients. In the current report, we describe an innovative multimodality approach of perioperative 18F-FDG PET/CT imaging, intraoperative 18F-FDG handheld gamma probe detection, and intraoperative ultrasound for tumor localization and verification of resection of all sites of hypermetabolic tumor foci in a case of occult recurrent metastatic melanoma.The use of diagnostic 18F-FDG PET/CT scan utilizing an intravenous injection of 14.8 mCi 18F-FDG. Then, on the day of surgery, the patient received an intravenous injection of 12.8 mCi 18F-FDG. A multimodality approach of intraoperative handheld gamma probe detection, intraoperative ultrasound tumor localization, specimen PET/CT imaging, and postoperative PET/CT imaging was utilized for accomplishing and verifying the excision of all three sites of occult recurrent metastatic melanoma within the left thigh region.This report discusses a case of occult recurrent metastatic melanoma, isolated to three separate sites within the subcutaneous tissues of the left thigh region, which was not clinically apparent but was found on diagnostic restaging whole body 18F-FDG PET/CT imaging, intraoperative 18F-FDG handheld gamma probe detection, and intraoperative ultrasound is promising combined technology for aiding in tumor localization and verification of excision and may ultimately impact positively upon long-term outcome of selected patients.This innovative multimodality approach of perioperative  In the year 2007, within the United State alone, it is estimated that approximately 60,000 cases of melanoma will be diagnosed and approximately 8,100 people will die of this disease . Despite18F-fluorodeoxyglucose (18F-FDG) positron emission tomography/computed tomography (PET/CT) imaging has become a well-established method for staging, restaging, and monitoring response to therapy of melanoma patients, and is widely accepted as a standard of care [Diagnostic  of care ,4,5. In  of care ,4-6.18F-FDG PET/CT imaging for melanoma patients is generally that of diagnostic image acquisition at the time of the original evaluation in those individuals considered at an elevated risk for regional and/or distant disease. In those instances when regional and/or distant disease can potentially be surgically approached, this current practice of diagnostic image acquisition at the time of the original patient evaluation provides only a static roadmap for guiding the surgical approach, but does not provide the surgeon with real-time intraoperative information on tumor location and verification of tumor resection. Recently, the application of intraoperative gamma probe detection in melanoma patients after preoperative injection of an intravenous dose of 18F-FDG has been reported by several groups of investigators [18F-FDG PET/CT imaging and intraoperative gamma probe detection for a case of recurrent melanoma has been recently reported by Carrera et al [The current application of tigators -11. Furt18F-FDG PET/CT imaging, intraoperative 18F-FDG handheld gamma probe detection, and intraoperative ultrasound for tumor localization and verification of resection of all sites of hypermetabolic activity in a case of occult recurrent metastatic melanoma.In the current Technical Innovations report, we describe an innovative multimodality approach of perioperative The case presented is that of a 50 year-old Caucasian female with isolated recurrence of metastatic melanoma to the subcutaneous tissues of her left thigh. Ten years prior to her current presentation, she underwent a wide excision and skin grafting of her left distal thigh region and a superficial left groin lymph node dissection for a 2.3 mm malignant cutaneous melanoma with 22 negative lymph nodes. She received no adjuvant therapy and subsequently continued routine follow-up by her surgeon.18F-FDG PET scan was performed, utilizing an intravenous injection of 13.9 mCi 18F-FDG, that revealed a solitary hypermetabolic focus within the anteriomedial left mid thigh region (peak SUV of 40.1) which was not palpable on clinical examination. No other hypermetabolic foci were identified elsewhere in her body. As a result, the patient subsequently (23 months prior to her current presentation) underwent isolated left lower extremity hyperthermic limb perfusion with melphalan and a concomitant left deep groin lymph node dissection.Twenty-five months prior to her current presentation, she developed two skin nodules located approximately 4 cm distal to the previous skin graft on her distal thigh region. A wide excision of her left distal thigh region was subsequently performed. A diagnostic whole body 18F-FDG PET/CT scan was performed, utilizing an intravenous injection of 16.3 mCi 18F-FDG, and redemonstrated a solitary hypermetabolic focus within the subcutaneous tissues of the anteriomedial left mid thigh region, with a peak SUV of 7.6. No other hypermetabolic foci were identified elsewhere in her body. Subsequently (15 months prior to her current presentation), CT guided wire localization and wide excision of the nonpalpable subcutaneous focus of disease in her anteriomedial left mid thigh region was performed.A six-month follow-up (17 months prior to her current presentation) diagnostic whole body 18F-FDG PET/CT scan  were located in the subcutaneous tissues of the anteriomedial left mid thigh region and one hypermetabolic focus (peak SUV of 3.4) was located in the subcutaneous tissues of the anterior lower one-third of the left thigh region. No other hypermetabolic foci were identified elsewhere in her body. On clinical exam, no visible or palpable abnormalities were noted in the left thigh region or elsewhere.The patient continued routine follow-up by her surgeon. One month prior to her current presentation, the patient underwent a routine follow-up diagnostic restaging whole body 18F-FDG was injected intravenously into a peripheral vein at approximately 80 minutes prior to the start time of the surgical procedure, as per our 18F-FDG and PET/CT protocols previously described [18F-FDG uptake within the left thigh region. Initially, one predominant site of 18F-FDG activity was transcutaneously identified with the gamma probe within the anteriomedial left mid thigh region. Surgical excision of the subcutaneous tissue (measuring 16.0 × 7.0 × 3.0 cm in size) of this area was undertaken. Post-excision evaluation of the excision bed within this region was performed with the gamma probe and revealed residual increased 18F-FDG activity above background. Therefore, gamma probe directed excision of additional deeper subcutaneous tissue (measuring 3.5 × 2.0 × 1.0 cm in size) of this area was undertaken. Further post-excision reevaluation of the excision bed within this region was again performed with the gamma probe and revealed no 18F-FDG activity above background.On the day of surgery, a dose of 12.8 mCi escribed . The pat18F-FDG PET/CT scan. With the gamma probe, difficulty was encountered transcutaneously distinguishing a finite site of 18F-FDG activity that was distinct from that of the underlying background muscular and vascular blood pool 18F-FDG activity. Therefore, intraoperative ultrasound was performed using a Hitachi HI VISION™ 6500 ultrasound system  with a variable frequency linear transducer EUP-L54M (range 10.0 to 13.0 MHz) . An 8 mm hypoechoic ultrasound lesion  of this area was undertaken. Post-excision evaluation of the excision bed within this region was performed with the gamma probe and revealed no 18F-FDG activity above background. Ex vivo gamma probe evaluation revealed increased 18F-FDG activity within the excised subcutaneous tissue. Likewise, ex vivo ultrasound evaluation revealed the corresponding hypoechoic ultrasound lesion within the excised subcutaneous tissue.Attention was then directed to the anterior lower one-third of the left thigh region where the third hypermetabolic focus was located in the subcutaneous tissues based on the previous diagnostic whole body n Figure  was iden18F-FDG injection. Specimen PET/CT imaging revealed the presence of three hypermetabolic foci, corresponding to the three sites of excised subcutaneous tissue that represented the three hypermetabolic areas in the subcutaneous tissues of the left thigh region that were originally visualized on the preoperative diagnostic whole body 18F-FDG PET/CT scan. The specimens were then transported back to the operating room in order to be sent to and processed by surgical pathology for standard pathologic evaluation.All three resected specimens were then transported to the nuclear medicine department and imaged with the clinical PET/CT scanner Figure  at a tim18F-FDG injection), she was subsequently taken to the nuclear medicine department and re-imaged with PET/CT scan without administration of an additional dose of 18F-FDG. The postoperative PET/CT scan demonstrated no residual sites of hypermetabolic activity, verifying excision of the all three previously visible sites of hypermetabolic activity within the left thigh region  declare that they have no competing interests.SPP organized, wrote, and revised the manuscript. NCH was the nuclear medicine physician who prepared the images for this report. EWM was the supervising senior surgeon for the entire project. MJW was the surgeon on this case. NCH, EWM, and MJW assisted in the writing and editing of this manuscript. All of the authors have approved the final version of this manuscript.


Alveolar soft tissue sarcoma of the tongue is a very rare and aggressive tumor which occurs in infancy with a relatively controversial histogenesis. It may mimic benign vascular neoplasms and may metastasize to the lungs, brain or bones.We present the case of a three-year-old Caucasian boy who was admitted to our department with a history of dysphagia and two episodes of oral bleeding which had lasted for a period of six months. A thorough histological examination together with imaging techniques form the basis of a reliable diagnosis.Alveolar soft tissue sarcoma of the tongue is a rare tumor which occurs in infancy and which is often misdiagnosed pre-operatively. It should therefore be included in the differential diagnosis of oral pediatric lesions. Alveolar soft part sarcoma of the tongue (ASPS) is a rare and aggressive malignancy which comprises 0.4% to 1.0% of all soft tissue sarcomas. It accounts for up to 5% of all pediatric soft tissue sarcomas, apart from rhabdomyosarcomas [A 3-year-old Caucasian boy originating from Northern Greece was admitted to our department in Hippokratio General Hospital of Thessaloniki with a history of dysphagia and repeated episodes of oral bleeding which had lasted for a period of six months. A physical examination revealed a reddish-blue, soft, painful and immobilized mass in the mid-dorsum area of the tongue extending to its base. There was no cervical lymph node enlargement. Computed tomography (CT) and magnetic resonance imaging (MRI) demonstrated a 3.3×1.9×2.0 cm mass with new vessel generation. It was obstructing the entrance to his pharynx and was initially thought to be a hemangioma , producing an ASPL-TFE3 transcript fusion which is detected on tumoral cell walls, is specific to ASPS . PatientThe typical histological characteristics of ASPS include a variable-size that is irregularly circumscribed, and division by thin fibrous connective tissue bands with delicate vessels delineating small pockets of variably-sized polygonal cells. The cells are of a uniform size and shape and contain granular, eosinophilic cytoplasm surrounding a vesicular nucleus that hosts a prominent nucleolus. The pseudoalveolar pattern characteristic of this tumor is seen when the alveolar cells undergo degeneration, lose their cohesiveness and become necrotic.ASPS has a close clinical and imaging resemblance to common benign vascular tumors such as hemangiomas, which may lead to misdiagnosis and inadequate or delayed treatment. Vascular malformations may present at birth, whereas ASPS does not, but if it did it grows rapidly and occurs in an older age group. In addition to hemangiomas and vascular malformations, other less likely differential diagnoses include a hyperplastic lingual thyroid and dermoid cysts. These neoplasms are easily differentiated from ASPS in CT and MRI findings. ASPS can also be differentiated from other histologically similar malignant neoplasms in its cytological uniformity, lack of nuclear atypia, and paucity of mitotic figures. Most ASPSs also contain PAS-positive, diastase-resistant cytoplasmic inclusions that are thought to consist of actin. The differential diagnosis of ASPS includes granular cell tumor, paraganglioma, metastatic renal cell carcinoma, malignant melanoma, alveolar rhabdomyosarcoma and ectopic lingual thyroid .et al. [The treatment of choice for ASPS of the tongue is generally surgical excision with sufficient margins. Surgery with chemotherapy or radiotherapy is useful in some patients. Treatment has been difficult to evaluate because of the small number of cases seen, but it seems that although the ectomy of the primary tumor is often successful, treatment of metastatic tumors is problematic . For suret al.  recommenAnother approach to treatment is the use of IFN alpha-2b as a monotherapy. There is some evidence that high doses of IFN may induce an impressive tumor response in ASPS with pulmonary metastases that have no response to chemotherapy. However, this needs further research . The proASPS of the tongue is a rare tumor which occurs in infants and children, usually in the head and neck region. The orbital and lingual sites are predominantly affected. Treatment is primarily surgical, with limited roles for adjuvant chemotherapy, radiotherapy and perhaps IFN. CT and MRI can be used to make the correct diagnosis of ASPS and to help the surgeon to perform a wide surgical resection to reduce the risk of local recurrence. Additional study of the histological types of ASPS that occur in children, specifically in the head and neck region, is suggested to determine whether differences in patient outcome and treatment exist between the subtypes.Written informed consent was obtained from the patient's next-of-kin 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.GN analyzed and interpreted the patient data regarding the disease. PC performed the data analysis and was a major contributor in writing the manuscript. IP, KK and GK performed the operations. All authors read and approved the final manuscript.


A 16 year old female with systemic lupus erythematosus presents with acute appendicitis. Final pathologic analysis of the appendix describes a lupus-associated vasculitis. Acute abdomen in patients with systemic lupus erythematosus (SLE) can pose a diagnostic and therapeutic challenge. Most of these patients are on chronic steroid treatment which may mask symptoms and lead to delay in diagnosis. Delay in diagnosis and treatment can increase mortality. Lupus-associated vasculitis is reported to be the cause of acute abdomen in up to 60% of SLE patients . We repode novo acute appendicitis.R.S. is a 16 year old female with a past medical history significant for systemic lupus erythematosus who presented to the ER with a one day history of right lower quadrant pain. The pain was described as constant and sharp, becoming steadily worse over the course of a day. She reported a decrease in appetite since the onset of symptoms. Past medical history was significant for SLE with biopsy- proven lupus glomerulonephritis, hypertension and hypothyroidism for which she was taking Prednisone 15 mg each day, Vasotec and Synthroid, respectively. On exam the patient was afebrile with stable vital signs. She did not appear to be in any distress. Her abdomen was obese, and non-distended. She was moderately tender to palpation in the right lower quadrant but did not exhibit any signs of rebound or guarding. Her laboratory values were significant for a WBC count of 10.2 with 84% neutrophils and a urinalysis that was positive for 5–10 WBC/hpf and moderate amount of bacteria. A CT scan of the abdomen and pelvis was obtained which revealed a slightly prominent, hyperemic, non-air or fluid filled appendix with trace pelvic ascites. The patient was then brought to the OR and underwent an open appendectomy. Intra-operatively, the appendix was inflamed with a walled off perforation into the mesentery. The appendix was removed and sent to pathology. The appendix measured 10 cm in length and 1.0 cm in greatest diameter. Upon gross examination and serial sectioning, the serosal aspect of the appendix was focally edematous and congested. The wall of the appendix was grossly unremarkable, and its lumen was empty. The entire appendix was submitted for histological evaluation and showed small vessel vasculitis and large vessel vasculitis. Small vessel vasculitis was characterized by transmural infiltration of lymphocytes Fig . FocallyPatients with SLE presenting with an acute abdomen pose a unique diagnostic dilemma for surgeons, as the differential diagnosis is diverse and often difficult to make. Usually these patients are on chronic steroid treatment making physical exam and laboratory values unreliable therefore leading to a delay in diagnosis and treatment. Surgical intervention is fraught with many potential complications especially in patients with severe disease activity who require chronic steroid and immunosuppressive therapies. Analysis of lupus patients with an acute surgical abdomen failed to identify any clinical, laboratory or radiological features that could reliably aid in early diagnosis and management . In addiStudies have found that vasculitis is the cause of acute abdomen in 35–60% of cases of SLE ,4-6. CauThe typical proposed pathogenesis of appendicitis is obstruction causing increased intraluminal pressure and subsequent collapse of the draining vessels. The ischemic injury that follows favors bacterial proliferation and additional inflammatory exudate and edema leading to the typical clinical manifestations of appendicitis. It is theorized that the lupus-associated vasculitis can follow a similar pathogenetic pathway by causing ischemia, bacterial proliferation and inflammation .Regardless of pathology, delay in diagnosis can be dangerous and a high index of suspicion must always be maintained when dealing with such patients. Early laparotomy shows an increase in survival rate . Emergen


Colonic Intussusception although common in children, is a rare cause of acute intestinal obstruction in adults. The etiology, clinical presentation and management of this condition is different in adults as compared to children. Pre-operative diagnosis is usually difficult due to the non specific and intermittent nature of the symptoms. CT scan can be a helpful adjunct in establishing the diagnosis. We present a case report of adult ileocolic intussusception with classical radiological signs and operative findings. A brief literature review is also presented with emphasis on the controversy of reduction of the intussusception before resection. A 74 year old male presented with a four month history of colicky right upper quadrant pain, altered bowel habits and weight loss. He did not have any rectal bleeding. On examination there were signs of anaemia and a palpable mass in the right upper quadrant. Base line blood tests including FBC were normal. An abdominal USS showed a 5 × 7 cm ill defined bowel related mass in the right upper quadrant. The liver was normal.A subsequent barium enema showed an intra-luminal filling defect measuring 8 × 10 cm in the proximal transverse colon with no flow of barium proximally Figure .A provisional diagnosis of colonic carcinoma was made although the tumour markers including CEA and CA 19.9 were normal. The patient underwent a laparotomy where an ileocolic intussusception was found at the level of hepatic flexure Figure . A rightIntussusception is an uncommon cause of intestinal obstruction and more than 95% of cases occur in the paediatric age group . IntussuThe clinical presentation is very non-specific which makes this a difficult condition to diagnose. Abdominal pain, nausea, diarrhoea and bleeding per rectum are the common symptoms. Rarely this can present with acute intestinal obstruction. The classical triad of abdominal pain, sausage shaped palpable mass and passage of red current jelly stools seen in children is rarely observed in adults ,7. The uBenign lesions account for almost 25% cases of intussusception in adults. The commonest benign lesion is a lipoma in the colon. These are solitary submucosal lesions with 75% occurring in the right colon. Small lipomas are asymptomatic. Other benign lesions include adenomatous polyps and Peutz-Jeghers polyps. However in more than two thirds of cases there is a malignant tumour in the colon or small bowel resulting in intussusception -17.Operative intervention is required in all cases of adult intussusception and unlike children conservative treatment does not work ,6. This In most cases of adult colonic intussusception, primary resection without reduction should be performed particularly in those more than 60 years of age due to a higher risk of malignancy. In cases of small bowel intussusception reduction before resection should be carried out only if there is a pre-operative diagnosis of benign etiology, the bowel is viable or it entails resecting massive lengths of small bowel with the risk of short gut syndrome ,20.Intussusception is a rare cause of acute abdomen in adults. A high index of suspicion and appropriate investigations  can result in prompt diagnosis. Unlike children 75% of cases are due to a malignant tumour in the small bowel or colon. The extent of resection and operative technique depend upon the age of the patient, results of investigations  and the length of the bowel involved.MNK carried out the literature review and drafted the paper. AA designed the paper, literature review and reviewed the manuscript. PS literature review, provided the figures and reviewed the manuscript. All authors have 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.


Adrenocortical carcinoma is a rare malignancy with a poor prognosis and presents with mass effects and less often with signs of hormone excess . Hormonally active adrenocortical carcinomas most commonly secrete cortisol while the co-secretion of multiple steroid hormones is rare.We report the case of a 59 year-old woman with a medical history of hyperaldosteronism due to a right adrenal adenoma. During follow up, she showed symptoms of hypercortisolism and hyperandrogenemia and a rapid growth of the adrenal mass. She underwent right adrenalectomy and the histology revealed the presence of an adrenocortical carcinoma. Six months post-operatively being on treatment with mitotane, she was diagnosed of metastatic disease to the liver. She underwent right hepatectomy and was started on systemic chemotherapy, with no signs of tumour recurrence during the following six months.The hormonal status should be carefully investigated in all cases of suspected adrenocortical carcinoma, as the pattern of hormone secretion may be a clue to the malignancy of an adrenal lesion. In addition, more data are needed to clarify the clinical and prognostic significance of the combined production of all adrenocortical hormones affecting either the survival or the quality of life of adrenocortical carcinoma patients. Adrenocortical carcinoma (ACC) represents a rare malignancy accounting for 0.05-0.2% of all cancers with a poor prognosis depending on the stage of the disease and the completeness of the resection ,2. ApproIn September 2006, a 59-year old Greek Caucasian woman presented to her local hospital due to a hypertensive crisis (blood pressure: 170/100 mmHg). Endocrinological investigation, revealed marked hypokalemia (serum potassium: 2.7 mEq/l), suppressed renin levels, increased aldosterone levels with an aldosterone/renin ratio >30, findings suggestive of primary hyperaldosteronism. However, cortisol, catecholamines or other hormones were not measured, despite the fact that she was a hypertensive patient. Abdominal CT scan revealed the presence of a solid enlargement of the right adrenal (max diam. 2 cm) with radiological characteristics of benign adenoma while attenuation value and wash-out procedure were not performed, making the diagnosis of "benign adenoma" somewhat arbitrary. The patient was started on spironolactone treatment, with gradual improvement of her blood pressure and serum potassium levels and was suggested of surgical excision of the adrenal adenoma, which she denied.One-year later, she was referred to our department with signs of sustained hypertension despite spironolactone treatment, mild hirsutism, irregular vaginal bleeding, increased body weight and central adiposity. Abdominal CT and MRI scan showed a large right adrenal mass (6.1 × 6.5 × 5.5 cm), with morphological characteristics suggestive of carcinoma  Figure . HormonaSix months after surgery, she showed signs of hypertension recurrence accompanied by hypokalemia (serum potassium: 2.9 mEq/l) and hypernatremia (serum sodium: 150 mEq/l). Abdominal CT scan revealed a metastatic lesion in the right hepatic lobe (diam 2.6 cm) Figure . The patWe report a patient with ACC and abnormal secretion of all adrenocortical hormones, namely glucocorticoids, mineralocorticoids and androgens. Although she was initially presented with hyperaldosteronism and radiological characteristics suggesting a benign adenoma, during a rather short-time follow up, she showed hypersecretion of all adrenal hormones, in parallel with an aggressive behavior of the ACC manifested by the rapid growth of the adrenal tumour and hepatic metastasis.In our case, only aldosterone and renin levels were measured during her initial evaluation, despite the fact that other hormones such as cortisol and catecholamines should be measured in a hypertensive patient. However, quantitative and/or qualitative changes in hormone secretion by an adrenal tumour are not uncommon . It has In addition, the temporal development from a 2 cm to a 6 cm tumour being malignant has rarely been reported. Most studies of incidentalomas have a portion of patients found to suffer from ACC, but their tumours are large (at least >4 cm) or grow rapidly (5-15 cm in 3 months) but not one which seem to have a quite modest growth rate (3-fold) in 1 year, as the present case. Our case in support of other cases and studies indicate that the growth rate of an ACC is largely unknown and varies widely between different patients due mostly to the fact that they are treated when they are found and not followed as the present case.In conclusion, the hormonal status should be carefully investigated in all cases of suspected ACC, as the pattern of hormone secretion may be a clue to the malignancy of an adrenal lesion . In addiACC: adrenocortical carcinoma; ACTH: adrenocorticotropic hormone; CT: computed tomography; DHEAs: dehydroepiandrosterone sulfate; MRI: magnetic resonance imaging; o,p'DDD: ortho, para', dichloro-, diphenyl-, dichloro ethane or mitotane.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the journal's Editor-in-Chief.The authors declare that they have no competing interests.MP conceived the study, collected data and drafted the manuscript. VP and GP helped with the patient care and the literature reviewing. AM carried out the operation. TE, SAR and DJ critically revised the paper. All authors read and approved the final version of the manuscript.


Urachal adenocarcinoma is a rare tumor and represents 0.17–0.34% of all bladder tumors. It has an insidious course and variable clinical presentation. We present a case report of a 58 year old white male with an urachal cyst who suffered irritative voiding symptoms and long term mucusuria, since childhood. After surgical removal of the cyst with a partial cystectomy a mucus adenocarcinoma was diagnosed histologically.The patient after a negative for metastatic disease screen underwent a completion radical cystectomy with pelvic lymph node clearance. Clinicians should have a high degree of suspicion for these rare tumors. Adenocarcinoma of the urachus arises from the urachal remnant. It is a rare and devastating disease, representing 0.17–0.34% of all bladder tumors . It is bA 58 year old white male presented in our department with mucusuria, recurrent bacteriuria and lower urinary tract symptoms. He had been treated in the past for recurrent episodes of prostatitis. He also reported mucusuria that had been present since childhood and for that reason thought it was not unusual. He did not report any hematuria. Abdominal examination revealed a round, smooth, firm, non-tender, suprapubic mass. The prostate gland was soft, painless and of normal size. Biochemical and haematological analysis was normal. An abdominal computerised tomography (CT scan) revealed a 7 × 6 cm round supravesical, midline cyst, with extension into the bladder figure . AntegraMost cases of urachal adenocarcinomas occur in the fifth and sixth decade of life. Clinical presentation varies. Hematuria is encountered in more that two thirds of cases. Less common symptoms are mucusuria, lower urinary tract symptoms and a palpable suprapubic mass ,5. MucusIf after a trial of antibiotic therapy symptoms do not resolve, a full urologic workup with cystoscopy and abdominal ultrasound or CT scan is indicated. CT appearance is of a large mixed solid and cystic lesion with the bulk of the tumor outside the bladder. Extravesical spread is common. In relation to the urinary bladder wall urachal carcinomas may be intramucosal, intramuscular or supravesival,  . Tumor cTreatment consists of open radical or partial cystectomy with pelvic lymph node dissection, and excision of the umbilicus and the urachal ligament ,2. LaparMisdiagnosis of urachal carcinoma is still a reality. Accurate diagnosis necessitates a degree of suspicion and appropriate imaging studies. Appropriate local treatment is the key for improved survival.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.EI participated in the operating theatre, was a major contributor in writing the manuscript, and submitted the manuscript and photographs. KS participated in the operating theatre and collected the patient data. MC conducted a literature search, preparation of final manuscript including grammarm and stule. VS participated in the operating theatre and assisted in manuscript writing. XS participated in the operating theatre and collected the references. CI participated in the theatre and revised the manuscript. All authors read and approved the final manuscript.


Primary cardiac paraganglioma is a very rare tumor with less than sixty reported cases in the literature. The clinical presentation is variable, but is most commonly manifested by hypertension and symptoms related to the catecholamine excess.We report a case of a 35 year old man who presented with anginal pain and hypertension. He was found to have a cardiac mass on the computed tomographic scan and echocardiogram. He underwent surgical exploration of the mass which on biopsy was found to be a 'Cardiac Paraganglioma'. Surgical resection of the tumor was successfully done and the patient is doing well five years after the surgery without any evidence of recurrence. His blood pressure, however, failed to normalize and needed single agent antihypertensive therapy.Cardiac paragangliomas have a relatively favorable outcome if diagnosed and resected in time. We briefly review the literature regarding the diagnosis, treatment and prognosis of this rare tumor. Paragangliomas, also known as extra-adrenal pheochromocytomas, are tumors derived from the neural crest cells. Primary cardiac paraganglioma is a very rare tumor with only less than sixty reported cases in the literature . The cliA 35 year old obese African American male, a mechanic by profession and no significant previous medical problems, came to the emergency room with left sided chest pain that started the night before and slowly got worse. By the time he reached the emergency room, the pain had reached 8 out of a maximum of10 in intensity. The pain was precipitated and aggravated by a stressful situation at home. It radiated to his left shoulder and was not relieved by rest. Patient had a five pack-year history of smoking but quit 10 years ago. He would occasionally drink alcohol. On further questioning, the patient revealed that he had been having mild left sided chest pains, sharp in nature and similar to the current episode, for the last few months. The pain would occur either with stress or exertion. He did not seek medical attention before because the pain would be transient and go away on its own after a few minutes. He was not on any medications and did not use any pain relieving medicines either for his prior symptoms.In the emergency room, patient was in mild distress and had a blood pressure of 165/81, something new for him. All the rest of the vital signs were normal and both the cardiac and pulmonary examination did not reveal any abnormality either. The basic laboratory work up was normal except a lipid profile showing elevated cholesterol of 238 mg/dl. The patient had an electrocardiogram that showed some premature supraventricular complexes without any ischemic changes. Cardiac biomarkers including serial troponin-I were also normal. The chest pain subsided in the emergency room with intravenous morphine. The patient was admitted to the hospital in the telemetry unit. He was found to have some runs of non-sustained ventricular tachycardia while in the hospital. He underwent a cardiac exercise stress test and an echocardiogram which revealed that patient had a normal ejection fraction but a dilated left atrium along with apical wall motion abnormality.Patient then underwent a cardiac catheterization. It did not show any obstruction in the coronary arteries, but did reveal a "tumor blush" with neovascularization seen projecting from the left circumflex artery to the "tumor blush". Figure  FollowinAs it was still unclear whether the mass was originating intrinsic or extrinsic to the heart, so to define it better anatomically, a transesophageal echocardiogram (TEE) was performed. The TEE also showed the large mass, likely extrinsic to the heart, compressing the left atrium. Figure  Finally,Although most cardiac paragangliomas are benign, local invasion can occur and metastases have been reported in up to 10% of cases . LocalizTaking in regard the anatomical location of the mass and elevated urinary normetanephrine levels, the patient was referred for surgical exploration. A recent report of a single center surgical experience has shown that the approach to remove the paragangliomas can be either a median sternotomy or posterolateral thoracotomy, depending on the location in the mediastium . Our patThe long-term outlook for patients with excised cardiac paragangliomas is unknown. In the only reported case series, there was one intraoperative death due to blood loss and ten of the 11 patients were alive at follow-up  . SurvivaAfter surgery, symptoms of catecholamine excess are usually eliminated or greatly improved in patients who had a functioning tumor preoperatively, although hypertension may or may not resolve ,9. SurveTEE: Trans-esophageal echocardiography; CT: Computed Tomography; MRI: Magnetic Resonance Imaging; I-123 MIBG: Iodine-123-meta-iodobenzylguanidine scintigraphy.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.TJK and OZ wrote the case report part of the manuscript. LZ wrote the introduction and prepared the images. IK came up with the concept of the manuscript and wrote the discussion portion of the manuscript. All authors read and approved the final manuscript.


Mesenteric panniculitis is a rare pathology of unknown etiology characterized by inflammation and fibrosis in the mesentery. Its protean clinical and radiological manifestations make it a diagnostic challenge. There is no established treatment available for its management. The clinical outcome is inconsistent, with the prognosis ranging from complete resolution without any treatment to rapid progression culminating in death.A 33-year-old Pakistani man presented with vague abdominal pain, an ill-defined epigastric mass and bilateral pedal edema. A detailed review of his history and laboratory investigations did not point to any diagnosis. The patient underwent an exploratory laparotomy based on the finding of mesenteric soft-tissue density on computed tomography. The laparotomy did not prove to be of any diagnostic or therapeutic value. Upon review of the pre-operative computed tomographic scan at our institution, a diagnosis of mesenteric panniculitis was made. An acceptable resolution of abdominal pain and pedal edema was attained after a 4-week trial of immunosuppressive therapy. This is the first reported case of mesenteric panniculitis with pedal edema as part of its presentation.An increased awareness may lead to the development of a less invasive diagnostic approach and optimal treatment for this rarely recognized condition. Mesenteric panniculitis (MP) is a rare inflammatory and fibrosing disorder of unknown etiology involving adipose tissue of the mesentery -5. SinceWe report a case of MP initially presumed to be a liposarcoma. This is the first reported case of MP with pedal edema as part of its presentation. We hope that this report will contribute towards an improved recognition and diagnosis of this rarely diagnosed condition.A 33-year-old male Pakistani sailor presented to us with a 1-year history of generalized, vague abdominal pain. The pain was insidious in onset and aggravated by squatting. The patient also complained of bilateral dependent pedal edema that started 1 month after the onset of abdominal symptoms. Furthermore, he reported occasional nausea but did not divulge any history of vomiting, altered bowel habits, malaise, fever or weight loss.At the onset of these complaints, the patient reported to a military hospital where a palpable epigastric/umbilical mass was discovered. Further evaluation with a computed tomography (CT) scan at the same time demonstrated a fat density mass in the retroperitoneum. A presumptive diagnosis of liposarcoma was made. A laparotomy was performed through a midline incision. Intra-operatively, a vaguely defined thickening was noted in the small bowel mesentery. Complete excision of the mass was limited by its close association with the gut loops. The bowel loops and peritoneal cavity appeared unremarkable. No lymph node enlargement was observed. In the histological evaluation, the excised specimen was described as benign adipose tissue with non-specific inflammatory infiltrates. There was no distortion of the tissue architecture or infiltration of vascular structures. The postoperative recovery was unremarkable.Nine months after laparotomy, the patient presented to our institution due to persistence of the initial symptoms. A detailed history and review of systems did not provide any further clues. The patient reported no change in abdominal symptoms or pedal edema. His weight had remained stable and he had not experienced any constitutional symptoms. During the initial physical examination, we documented a healed midline laparotomy scar, generalized abdominal tenderness of moderate intensity and a firm, ill-defined mass in the epigastric/umbilical region. Bilateral, pitting pedal edema was also noted. The remainder of the physical examination was unremarkable.Laboratory studies, including complete blood count, peripheral blood film, erythrocyte sedimentation rate, serum C-reactive protein, amylase, lipase, liver function tests and autoimmune work-up all had negative results. A repeat CT scan was also contemplated but, unfortunately, was not a financially feasible option for our patient. Upon review of the pre-operative CT scan of the abdomen Figures , 2 and 3The patient was started on prednisone 40 mg/day plus azathioprine 50 mg/day and was re-evaluated after 4 weeks. An acceptable symptomatic improvement was observed. The pedal edema resolved completely whereas the pain reduced from five points to two points on a scale of ten points. Azathioprine was discontinued and a tapering dose of steroids was prescribed. The patient declined follow-up.Mesenteric panniculitis constitutes idiopathic inflammation and fibrosis of the mesentery -5. MostlDiagnosing MP presents a challenge. The patient may present with vague abdominal pain, an abdominal mass, altered bowel habits, intestinal obstruction or ascites. MP has been an incidental finding in as many as half of the cases reported -6. AbdomDuring the diagnostic work up of MP, extensive biochemical investigations, including immunohistochemical staining, are usually negative. Their value is usually limited to the exclusion of other differentials. Nonetheless, a raised erythrocyte sedimentation rate (ESR) may be seen in a minor proportion of patients .A CT scan may point towards the diagnosis in about half the cases ,5,6,9. MHistological evaluation is considered imperative for establishing the definitive diagnosis. It may illustrate fat necrosis, chronic inflammation, fibrosis, a combination of any two of these or all of the three ,5,6. HowCurrently, there is no established regimen for the management of MP . The treThe natural course of MP is as inconsistent as its presentation. It may regress, may follow a benign stable course or may progress to death. This variability has been observed in both treated and untreated cases ,9. PredoMP poses a diagnostic challenge owing to its protean presentation, findings and clinical course. An improved understanding may help in increased recognition of this rare, possibly under-recognized, condition. Such efforts may also lead to the development of a less invasive diagnostic approach and improved therapy ,13. CareMP: mesenteric panniculitis; CT: computed tomography; ESR: erythrocyte sedimentation rate.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.AMZ, MAR and TC were involved in the direct clinical care of the patient and therapeutic planning, AMZ and GK were involved in the interpretation of diagnostic studies especially the CT scan. All authors contributed equally to the manuscript. All authors have seen and approved the final manuscript and stand responsible for its contents.


Primary mesothelioma of the pericardium comprises less than 1% of all mesothelioma cases. Its typical presentation is insidious, with nonspecific signs and symptoms, and usually results in heart failure due to cardiac tamponade, either by a serous effusion or by direct tumorous constriction of the heart. With the exception of several case reports, the outcome is uniformly fatal, and patients typically die within six months of diagnosis.A 45-year-old African American female presented to the emergency department with several days of dizziness, difficulty walking, and low blood pressure. The patient suddenly suffered cardiac arrest, and her death was pronounced. The medical examiner assumed jurisdiction of the case due to the sudden death nature of the case without known medical history. At autopsy, a one-liter hemopericardium was present, and the pericardial sac was thick, granular and adhesed to the heart, suspicious for pericarditis. Microscopic examination of the pericardial tissue instead led to a diagnosis of primary pericardial mesothelioma.Our case demonstrates a pericardial mesothelioma initially masquerading grossly as pericarditis. Microscopic examination of any grossly abnormal pericardial tissue therefore may be warranted so that a neoplastic disease process does not go undetected. Additionally, of the approximately 200 such tumors reported in the medical literature, a case demonstrating marked hemopericardium and resulting in sudden death has not been described until now. Fortunately, mesotheliomas are very uncommon tumors, with an incidence of approximately one per million. Such lesions arising from the pericardium constitute only 0.7% of all cases and therefore represent an extremely rare entity . This neA 45-year-old African American female presented to the emergency department (ED) with a new onset of dizziness, difficulty walking, and low blood pressure. Her past medical history was significant for hypercholesterolemia, hypertension, and a recent urinary tract infection. She smoked tobacco but used neither alcohol nor illegal drugs. There was no history of asbestos exposure or recent viral illness. Physical examination was unremarkable. Lab work was performed and showed normal results, so the patient was released. The following day, she returned to the ED once again, and reported that her symptoms had escalated significantly. Soon thereafter the patient developed multiple organ failure with subsequent cardiac arrest, passing into pulseless electrical activity, and her death was pronounced. The initial significant autopsy finding was the discovery of approximately one liter of blood within the pericardial cavity. The pericardial sac was focally adhesed to the surface of the heart. The pericardial surface of the heart was markedly granular and thickened. An area on the pericardium of the anterior left ventricle was focally disrupted without frank tearing or laceration and blood was observed to ooze through this defect. Gross photographs were not taken due to the initial impression of classic pericarditis. The remainder of the postmortem examination was negative for grossly suspicious malignancy, including pleural, pulmonary and lymphoreticular involvement. No abdominal or pelvic pathology was identified. In particular, there was no omental or peritoneal caking, and no ovarian, renal, or hepatic masses were identified. Microscopic examination of tissue sections taken from the epicardium revealed dense lymphocytic infiltrates admixed with numerous neoplastic, polygonal cells with abundant cytoplasm and enlarged, irregular nuclei containing conspicuous nucleoli. Some of these tumor cell clusters formed gland-like formations and papillary structures Figure . AbundanSeveral studies demonstrate the infrequent nature of primary pericardial mesotheliomas. A ten-year focused autopsy review of malignant tumors by Mukai, et al., recovered a single case out of a total of 2,649 cases while a larger 500,000 case study provided an incidence of less than 0.0022% . Of the Perhaps the reason for the lesion's low detection rate derives from its typical clinical presentation, a group of nonspecific signs and symptoms of insidious onset related to declining cardiac function - dyspnea, chest pain, fever, and weight loss - and manifesting as restrictive pericarditis, pericardial effusion, and cardiac tamponade . Local vThe usually progressive disease process appears to be the only positive characteristic regarding this tumor, in order that the patient's condition be diagnosed and afforded at least some measure of treatment. Unfortunately, the overall survival rate is less than six months, and treatment is focused on palliation, including such procedures as partial pericardectomy for relief of cardiac tamponade . VariousOver the last decade, the SV40 virus has stepped into the forefront of suspected agents in the pathogenesis of mesothelioma . The virThe typical gross pathologic features of pericardial mesothelioma are that of a tan, variably nodular to solid thickening of the pericardium, sometimes extending into the aorta and pulmonary trunk. Epicardial invasion has been reported, but deep invasion of the myocardium is not common . HistoloThe usual immunohistochemical positivity of pleural mesotheliomas for cytokeratin (CK 5/6), calretinin, and HBME-1 with negative staining for carcinoembryonic antigen (CEA), B72.3, and Leu-M1, holds true for the pericardial variety. Kayser, et al., discovered that a combination of probes with antibodies against carrier-immobilized ganglioside GM1, hyaluronic acid, calretinin, and HBME-1 was highly sensitive and specific for distinguishing mesothelioma from metastatic carcinomas, as the markers for ganglioside GM1 and hyaluronic acid showed the highest sensitivity and specificity for mesothelioma. In contrast, CEA and N-acetyl-D-glucosamine-bearing neoglycoprotein displayed high sensitivity and specificity for metastatic carcinomas .In this case, the original diagnosis based upon the gross examination was that of a diffuse, marked pericarditis with associated profound hemopericardium. However, upon initial microscopic examination, while lymphocytic pericarditis was in fact present, the more significant neoplastic entity was additionally identified. Pericarditis, especially the fibrinous variety, can grossly mimic pericardial mesothelioma, as it may exhibit thickening and a fine granularity of the pericardium. A host of tumors may also enter the differential diagnosis. While primary pericardial neoplasms including angiosarcoma, teratoma, and fibroma can occur, they are rare also. More commonly encountered are metastatic tumors originating from lung, breast, skin (melanoma), as well as lymphomas and leukemias .When confronted with the finding of hemopericardium during an autopsy, the forensic pathologist may have a number of different etiologies to entertain. Altun, et al., encountered 40 deaths related to hemopericardium and concluded that the overwhelming majority of these deaths were trauma-related . StabbinSeveral relatively common causes of hemopericardium encountered during autopsy include type A aortic dissections and ruptured myocardial infarctions. A number of other different etiologies, albeit rare, have been reported including vascular lesions , anticoagulation therapy, acute bacterial pericarditis, Takayasu aortitis, and rheumatoid arthritis -16.This challenging autopsy case initially presented as a diffuse, fibrinous pericarditis causing considerable hemopericardium and resulting in sudden death. Histologic and immunohistochemical analyses were essential in elucidating, and confirming, the presence of a primary pericardial mesothelioma. This case, among others encountered in our review of the literature, questions the accuracy of a diagnosis of pericarditis based solely on gross inspection. Without a microscopic examination of grossly abnormal pericardial tissue, a more significant disease process could go undetected. This includes a rare, but possible, neoplastic process.Consent was obtained for publication of this case report and accompanying images. A copy of the consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.DL constructed the majority of the manuscript. DC provided the histological interpretations and contributed to manuscript proofreading. AG provided the case history and autopsy findings and contributed to manuscript proofreading.


Mantle cell lymphoma is a rare non-Hodgkin's lymphoma. It is a subtype of B-cell lymphoma with frequent involvement of the bone marrow and the gastrointestinal tract. Isolated parotid gland involvement seldom occurs. Here we report an unusual case of isolated infiltration of the parotid gland by mantle cell lymphoma. The aim of our study is to correlate magnetic resonance imaging findings with the histological features of the disease. To the best of our knowledge, no similar radiological findings of mantle cell lymphoma have been published before.A 72-year-old Caucasian woman presented with a painful left parotid enlargement. She was diagnosed with mantle cell lymphoma involving the left submandibular gland seven years prior to presentation. Her whole body CT scan showed the absence of pathologically enlarged lymph nodes. However, a magnetic resonance imaging showed enlargement of her left parotid gland and an abnormal parenchyma with mixed-type solid and cystic lesions. A biopsy of her left parotid gland and subsequent histological examination confirmed a mantle cell lymphoma (common variant) relapse.Although rare, the involvement of parotid gland with mantle cell lymphoma must be considered in the differential diagnosis of parotid tumors. It is usually characterized by an aggressive clinical course with a median overall survival of two to five years . The invd cases) . Here weA 72-year-old Caucasian woman was referred to the Department of Haematology, General Hospital G. Papanikolaou, with a painful left parotid enlargement but without any other physical findings. Seven years prior to this presentation, she was diagnosed with MCL in her left submandibular gland, which was treated with surgical removal, radiotherapy, and a chemotherapy regimen of cyclophosphamide, hydroxydaunorubicin (Adriamycin), Oncovin (vincristine) and prednisone/prednisolone (CHOP). Her white blood cell (WBC) count of 5100/μL was represented by a normal differential of 69% neutrophils, 25% lymphocytes , 5% monocytes and 1% eosinophils. Her hemoglobin, hematocrit and platelet levels were normal at 12.7 g/dL, 38.4% and 219/μL, respectively. Her bone marrow smears and biopsy were both normal without evidence of any infiltration by lymphoma cells.An ultrasound examination of our patient revealed multiple cystic lesions in the parenchyma of her left parotid gland. Her MRI showed an enlargement of her left parotid gland and the total replacement of her normal parenchyma with mixed-type solid and cystic lesions Figure . The solA biopsy of her left parotid gland and subsequent histological examination showed the presence of MCL (common variant). The MCL was composed of monomorphous small to medium-sized lymphoid cells which most closely resembled centrocytes with a vaguely nodular growth pattern. The prominent neoplastic nodules were adjacent to the cystic area Figure .Immunostaining results for CD20, CD79a, CD43, CD5, sIgM(λ+) and cyclin D1 were positive Figures  and 3D aOur patient was treated with partial parotidectomy and chemotherapy. Her post-operative course was uncomplicated. Eighteen months after surgery she was asymptomatic and disease-free.Mantle cell lymphoma is a relatively rare and aggressive form of NHL. In the past, it has been referred to with various names including intermediately differentiated lymphocytic lymphoma, centrocytic lymphoma, and mantle zone lymphoma . MCL hasThe immunohistological features of MCL reveal a characteristic phenotype. The cells express relatively intense surface immunoglobulin M (IgM) and/or immunoglobulin D (IgD) and are usually positive for CD5, FMC7 and CD43, but negative for CD10 and BCL6. CD23 is negative or weakly positive. Almost all cases of MCL are moderately to strongly positive for cyclin D1. Cyclin D1 expression can be detected in a subset of cases of chronic lymphocytic leukemia (CLL) and/or small cell lymphoma (SLL) and hairy cell leukemia. Usually, cyclin D1 is weakly positive in these neoplasms. Cyclin D1 can also be expressed strongly in approximately one-quarter of plasma cell myeloma cases.in situ hybridization (FISH) probes [Conventional cytogenetic analysis demonstrates a translocation between the immunoglobulin heavy chain and cyclin D1  genes, tq13;q32) in 70% to 75% of cases. However, almost all cases demonstrate the rearrangement of these genes using fluorescence  in 70% tet al., in a clinicopathological study of 80 cases, extranodal involvement at presentation occurred in 76% of cases and the most common sites were the bone marrow (63%), peripheral blood (34%), gastrointestinal tract (10%) and Waldeyer's ring (10%). In 25% of cases the extranodal location was the disease's primary presentation, while the most common sites were the Waldeyer's ring (6%), intestine (5%), orbit (3%) and salivary gland (3%) [Clinically, MCL occurs in middle-aged to older individuals with a median age of about 60 years, and predominantly in men . Most paal tract 0% and WaMRI is the method of choice when treating patients with palpable parotid gland masses to assess the exact extent of the tumors, the invasion of neighboring structures, perineural spread, and lymph nodes staging . Most paIn this case, our patient's MRI showed an enlargement of her left parotid gland and a total replacement of her normal parenchyma with mixed-type solid and cystic lesions. The solid components were mildly enhanced using contrast medium. This appearance reflected the histological pattern of the lesion as MCL was composed of neoplastic nodules which were adjacent to the cystic area. To the best of our knowledge, there are no other published radiological findings on MCL of the parotid gland.The differential diagnosis includes benign and malignant parotid tumors, especially Warthin tumors and adenoid cystic carcinomas, which may also have a solid cystic appearance. These tumors rarely occupy the total gland parenchyma. In particular, Warthin tumors are bilateral in up to 10% of cases reported. They present as well-circumscribed, partly cystic and partly solid lesions on MRI and are often located in the tail of the parotid gland. Adenoid cystic carcinoma usually presents as an infiltrating mass with a high propensity for perineural invasion. On MRI adenoid cystic carcinoma has an irregular contour, poorly defined margins, and a strong enhancement after the administration of contrast medium .Special caution is required in the follow-up examination of patients with primary Sjögren's syndrome, as the risk of developing a lymphoma is increased. In this case the typical inhomogeneous nodular MRI picture seen in Sjögren's syndrome will change into a homogeneous pattern that can involve the parenchyma partially or even entirely .Isolated parotid gland involvement by MCL is very rare but should be considered nonetheless in the differential diagnosis when replacement of a patient's normal parotid parenchyma with mixed-type solid and cystic lesions is involved.CHOP: cyclophosphamide, hydroxydaunorubicin (Adriamycin), Oncovin (vincristine) and prednisone/prednisolone; CLL: chronic lymphocytic leukemia; CT: computed tomography; Ig: immunoglobulin; MCL: mantle cell lymphoma; NHL: non-Hodgkin's lymphoma; SLL: small cell lymphoma; WBC: white blood cell.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.MP performed the chart review and prepared the manuscript. AA evaluated and treated our patient, and also helped prepare the manuscript. FI was the pathologist who examined the specimens from our patient. TK and PP participated in manuscript preparation. All authors read and approved the final manuscript.


Fibrosarcomas (FS) of bone are a rare malignancy accounting for less than 5% of all primary malignant bone neoplasms. Diagnosis and treatment approaches of this entity are complex and require a skilled and experienced multidisciplinary team.Authors report their experience with a case of FS of humerus showing a pathologic complete response to neo-adjuvant chemotherapy based on adriamycin, cisplatin and ifosfamid. This approach allowed limb-sparing surgery with an excellent functional and psychological result. Fibrosarcomas (FS) of bone represent 5% of all primary bone sarcomas ,2. It ocSurgery is the standard treatment for fibrosarcoma of bone . AmputatThe role of systemic chemotherapy is not established. Few published reports of chemotherapy in FS of bone have been reported. Chemotherapy regimens used are similar to those used for osteosarcoma. Given to the rarity and heterogeneity of published reports it is not possible to draw conclusions about the role of neo-adjuvant chemotherapy in improving patients outcome and survival.We present here a rare case of locally advanced FS of bone experiencing complete pathologic response to adriamycin-cisplatin-ifosfamid neo-adjuvant chemotherapy, allowing limb sparing surgery, to illustrate the antitumor activity of this regimen in this rare tumor.A 28-year-old woman complained of pain and tumefaction in the upper portion of her right arm. She did not have fever, or trauma. Physical examination, showed a raised mass in the proximal portion of the right arm; with no clinical signs of neurovascular damage. There was no local erythema or skin lesions, and no palpable lymphadenopathy. There were pain and limitation of abduction, internal and external rotation of the right limb.The remainder of the physical examination was normal. Radiographs revealed a diaphyseal pathologic fracture involving the right humerus with a periosteal reaction. Magnetic resonance imaging (MRI) of the right arm Figure  showed aA biopsy was performed. Microscopic examination showed fascicles of spindle cells with areas of collagen fibers, with an elevated mitotic index Figure . The micA technetium-99 m labeled methylene diphosphonate radionucl ide (Tc 99 m/HMDP) bone scan revealed an area of increased uptake in the right proximal humerus, without other foci of abnormal isotope uptake that corresponded in location to the abnormalities visualized on MRI. Computed tomographic (CT) scanning of the thorax performed revealed no abnormalities. Laboratory test results were normal, left ventricular fraction ejection (FEV) was normal and equal to 69%.Surgical resection is the standard treatment of FS of bone. Early studies support the use of amputation . At the In our case limb salvage surgery was not possible at the time of first presentation, and consequently neo-adjuvant chemotherapy was considered in order to ovoid amputation, and to achieve a wide surgical excision.The role of chemotherapy in FS of bone is unknown. Up to now, no large chemotherapy studies of FS of bone are published; and only few case reports are reported. There is no recommendation regarding the optimal drug regimens, and the protocols used are formulated at the discretion of the medical oncologist, and were most commonly based on Adriamycin and cisplatin.API regimen is an active combination in the treatment of osteosarcoma (French Sarcoma Group FGS)  with 37-2 and cisplatin 100 mg/m2 on day 1 and ifosfamid 1.8 g/m2/d during 5 days with Uromi thexan (Mesna®) 1800 mg/m2/d during 5 days) as neo-adjuvant chemotherapy. G-CSF (filgastrim) was administrated from day 7 to day 14 of each cycle.Based on this data, we use the API combination  and 1 episode of inter-costal Zona after the second course of chemotherapy, successfully managed with Valaciclovir.Our case showed excellent clinical and radiological partial response Figure  after 3 A conservative surgery was performed after 3 cycles; The patient received limb-salvage procedures with wide local resection of the tumor, reconstruction with humeral centromedullary nailing, and replacement of the excised segment of bone by cemented spacer.Interestingly, histological study of the specimen showed pathologic complete response of the tumor Figure , suggestThree post-operative courses of API were programmed; however, only 2 cycles of chemotherapy were administrated, and the treatment was discontinued because of serious adverse event (medullar aplasia that was successfully managed).Although it is not possible to make a legitimate conclusion with a single presentation, this rare case of locally advanced FS of bone highlights the role of adriamycin-cisplatin-ifosfamid neo-adjuvant chemotherapy to achieve limb-sparing surgery and to avoid amputation. The promising results of API regimen in this case suggest the role of chemotherapy in the management of FS of bone.More studies are needed to confirm the efficacy and safety of this regimen in FS of bone and to determine the optimal treatment plan that will improve the outcome of these patients.The authors declare that they have no competing interests.All authors have made significant contributions by making diagnosis, treatment and intellectual input in the case and writing the manuscript. All authors read and approved the final manuscript.Written informed consents were obtained from the patient for publication of this case report. A copy of the written consent is available for review by the journal's Editor-in-Chief.