Tumoral calcinosis is a rare and benign condition characterized by massive subcutaneous soft tissue deposits of calcium phosphate predominantly around large joints.Familial tumoral calcinosis was present in two members of a Han Chinese family, namely, the son and daughter. The 14-year-old son had the first operation on his right sole of the foot at the age of six, and then experienced subsequent surgeries at a lesion in his right sole of the foot and left hip, respectively. The 16-year-old daughter underwent her first operation at the age of six in her left gluteal region, and subsequent surgeries were performed due to recurrence at the same lesion. Pathologic diagnoses of surgical specimens in both of the patients were reported as tumoral calcinosis. The laboratory results showed hyperphosphatemia with normal levels of serum calcium and alkaline phosphatase. Only surgical treatment was performed in both patients with satisfactory prognosis.This is the first report of Chinese familial tumoral calcinosis. The etiopathogenisis and treatment are discussed. Tumoral calcinosis (TC) was first described by Inclan  in 1943 A 14-year-old Han Chinese boy presented with an eight-year history of TC. He first noticed a painful mass on the bottom of his right foot at the age of six, and mass resection was performed. Then an operation was conducted for a mass that developed on the bottom of his left foot at the age of eight. A mass first occurred on his left hip at the age of 12. This mass excised and diagnosed on pathology as a tendon calcification tumor. After that, a recurrent mass on his lateral left hip was observed. On physical examination, the mass was firm, sessile, with a clear edge and normal skin temperature, and measured 4 × 5 cm. Distal circulation, muscle strength, motion, and sensation of the left lower limb were all intact. His serum phosphorus level (2.7 mmol/L) was higher than the upper normal range (0.97 to 1.61 mmol/L). Calcium and alkaline phosphatase levels were normal. Radiography revealed a multilobular, calcified mass around the left hip joint Figure . It showA 16-year-old Han Chinese girl presented with a 10-year history of TC. Several resections had been performed due to recurrence. On physical examination, a large mass was found on her left hip and buttock. It was hard, fixed, nodular, and with a chalky effusion from fistula of involved skin. The motion of left hip articulation was intact. Her serum phosphorus level was higher than the upper normal range. Calcium and alkaline phosphatase were normal. Radiography revealed multilobular calcification near the left hip articulation and within the soft tissue of her buttock. The function of her skeleton and articulation nearby was normal. Pathological examination confirmed the diagnosis of TC. No similar complaint was made by other family members. The parents were first cousins.et al. [TC is a rare disorder of mineral metabolism characterized by tumor-like periarticular deposition of calcium phosphate. There are two major clinical categories of TC based on its pathogenesis: familial tumoral calcinosis (FTC) with two subtypes: hyperphosphatemic FTC (HFTC) and normophosphatemic FTC (NFTC) based on serum phosphate status ; and secet al. . Our twoet al. , coding et al.  encodinget al. , encodinet al. . NFTC iset al. .Resection of the mass is the preferred treatment for TC in a relatively stable stage in which the mass is capsulated, but recurrence is common. Phosphate depletion  and low-phosphate, low-calcium diets, have a varied effect on FTC, but the benefits are limited . In our In summary, our presentation is the first report regarding FTC in Chinese patients. Imaging and pathological examinations are the commonly used diagnostic procedures. Further study will focus on epidemiology in Asia, the mutations in genomics and the variance between Asian and Caucasian patients. Although the pathogenesis of the calcification process in TC is still controversial, surgical removal is the mainstay treatment with a satisfactory prognosis.Written informed consent was obtained from the patients, with their parents' witness and consent, for publication of this manuscript 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.XLC collected the patient data regarding FTC. JWG performed the pathological examination. CZ analyzed and interpreted the data, and was a major contributor in writing the manuscript. KX provided constructive suggestions during manuscript writing. All authors read and approved the final manuscript.


Extraskeletal mesenchymal chondrosarcoma is rare and metastasis to the pancreas is extremely rare, with only four cases reported in the literature. The therapeutic effectiveness of chemotherapy remains uncertain. We report a 39-year-old man with extraskeletal mesenchymal chondrosarcoma of the buttock, who had metastases to the pancreas, bones, and lung. He underwent distal pancreatectomy, resection of the buttock tumor, and chemotherapy. He had a good response to chemotherapy and survived for about 3 years after surgery. Mesenchymal chondrosarcoma is a rare malignant neoplasm characterized by a biomorphic pattern of poorly differentiated small round cells and islands of well-differentiated hyaline cartilage. This tumor has a widespread distribution. The craniofacial bones , ribs, ilium, and vertebrae are the most common sites. One-fifth to one-third of the lesions are extraskeletal, primarily affecting the somatic soft tissues .Metastasis to the pancreas is extremely rare, with only four cases reported in the literature –6. We heA 39-year-old man presented with a 4-month history of severe upper abdominal pain after drinking alcohol. The patient denied any significant past medical or surgical history. Physical examination did not reveal any abdominal mass, and laboratory data including tumor markers  showed no abnormalities. Abdominal computed tomography (CT) showed a well-circumscribed, low-attenuation, inhomogeneously enhancing mass measuring about 5 cm in diameter in the body and tail of the pancreas. There were no signs of invasion of the surrounding tissues or dilatation of the distal pancreatic ducts. The CT findings were not typical for pancreatic cancer . CT alsoHistological examination of both specimens showed a poorly differentiated round cell malignant neoplasm, growing in sheets. The tumor cells had scant, pale eosinophilic cytoplasm . These fEWSR1 gene rearrangement. These histological and genetic findings led to a diagnosis of extraskeletal mesenchymal chondrosarcoma. As the tumor in the buttock was larger and was more typical of extraskeletal mesenchymal chondrosarcoma based on the histological findings, we concluded that the primary site was in the buttock and that the pancreatic tumor was metastatic.Intraoperative examination showed that the intra-abdominal tumor occupied the body and tail of the pancreas and pushed against the lesser curvature of the stomach. No ascites, peritoneal dissemination, or infiltration into the surrounding tissues was observed, and the tumor was resected. The resected tumor mass measured 6 cm × 5 cm. The buttock tumor had poorly defined margins with invasion into the gluteus maximus muscle and was widely resected with the muscle. The resected tumor mass measured 7.2 cm × 5.6 cm. Histological examination of both specimens showed a poorly differentiated round cell malignant neoplasm growing in sheets, with extensive necrosis. The tumor cells had scant, pale eosinophilic cytoplasm Figures . The butThe patient received postoperative adjuvant chemotherapy consisting of three cycles of ifosfamide and doxorubicin, three cycles of cisplatin and doxorubicin, and two cycles of vincristine, doxorubicin, and cyclophosphamide to treat his multiple bone and lung metastases. He had a good response to chemotherapy, and most of the metastases were no longer detectable on positron emission tomography at the end of therapy . HoweverMesenchymal chondrosarcoma was first reported by Lichtenstein and Bernstein in 1959 , and extP = 0.008), suggesting that addition of chemotherapy improves disease-free survival. Several cases that were sensitive to combined radiotherapy and chemotherapy have also been reported [Mesenchymal chondrosarcoma is an aggressive malignancy, and the primary treatment is surgical resection. Huvos et al.  publishereported , 14. Tumreported , as withreported .The prognosis of patients with mesenchymal chondrosarcoma is poor because of the high frequencies of early recurrence and remote metastasis , 15. TheMesenchymal chondrosarcoma with metastasis to the pancreas is extremely rare, with only four cases reported in the literature. In this case, the patient presented with both a pancreatic tumor and a soft tissue tumor in the buttock, and it was difficult to determine the definitive diagnosis by biopsy. Because the patient was young and complained of abdominal pain, we resected both tumors to determine the definitive diagnosis and determine the therapeutic strategy, including chemotherapy. Although the prognosis was expected to be poor because of the widespread metastases to the pancreas, bones, and right lung, chemotherapy was beneficial and the patient survived for 2 years and 10 months after surgery.We report here a rare case of mesenchymal chondrosarcoma with metastasis to the pancreas. Further development of novel agents for treatment is essential for improving the prognosis of this type of tumor.


Giant-cell tumor of the bone (GCTB) is a rare neoplasm that affects young adults. The tumor is generally benign but sometimes can be locally aggressive. There are no standardized approaches to the treatment of GCTB. Recently, the RANKL inhibitor denosumab has shown activity in this tumor type. We present the case of a young female who presented with locally advanced disease and was successfully managed with the neoadjuvant use of denosumab allowing for surgical resection of the tumor that was previously deemed unresectable. Following surgery, the patient is being managed with continued use of denosumab as ‘maintenance,' and she continues to be free of disease. Our case highlights a novel approach for the management of locally advanced and aggressive giant cell tumor of the bone. Giant-cell tumor of bone (GCTB) is a relatively rare neoplasm that affects young adults , 2. It iA 27-year-old woman presented with complaints of back pain in 2007 while living in Japan. Initial imaging revealed a T6 vertebral mass, and biopsy confirmed a diagnosis of GCTB. She underwent resection of the tumor with spinal stabilization. The patient was then lost to followup. In January 2010, she presented again with a 10.3 cm paraspinal mass in the T6 area on surveillance magnetic resonance imaging (MRI). This mass pushed the upper portion of the lungs posteriorly, the carina anteriorly, and the thoracic aorta laterally. Recurrent GCTB was confirmed by fine needle aspiration and biopsy. A resection was planned, and the patient was taken to surgery. Unfortunately, the tumor was unresectable due to extensive vertebral and vascular involvement. The patient was referred to radiation oncology and underwent intensity-modulated radiation therapy (5040 cGy) to the mass from March to April 2010. Follow-up computed tomographic imaging in May 2010 demonstrated an increase in the size of the mass from 9 × 8.9 cm AP to 10.2 × 9.2 cm Figures . The patAlthough GCTB is a benign neoplasm, recurrent GCTB can behave in an aggressive fashion and can be difficult to surgically resect or to treat with standard chemoradiation. Our case not only highlights this difficulty, but it also underscores the importance of multimodality management in the treatment of this tumor. In particular, our experience provides additional evidence that denosumab may be particularly useful in the neoadjuvant setting. At this time, there are no randomized studies that can help direct therapy for recurrent GCTB. The discovery of the role of RANKL signaling in the pathogenesis of GCTBs has ushered in a new treatment era for this neoplasm, and phase II studies have shown clinical benefit from the use of the RANKL inhibitor denosumab. In our case of a recurrent GCTB that was not only unresectable but also nonresponsive to traditional radiotherapy, denosumab treatment markedly shrank the tumor and enabled complete surgical resection. To date, she remains recurrence-free, while continuing to receive denosumab. This remarkable case is encouraging, and hopefully ongoing clinical studies will better define the role of anti-RANKL biologics in the multimodality management of this unusual neoplasm.


E. coli is sensitive to all antibiotics tested. Transrectal ultrasound (TRUS) revealed a cystic mass in the outer prostate. Seminal vesicle cyst and left renal agenesis were confirmed by magnetic resonance imaging (MRI). Maximum flow (Qmax) at uroflow was greater than 15 mL/sec. We therefore decided to manage this disease conservatively with alpha blockers and antibiotics. After 6-month' followup the patient did not report any complain and the uroflow test was similar to a normal urination. From one case report and literature review, the authors suggest a diagnostic and therapeutic strategy for the management of this rare condition. Seminal vesicle cyst is an extremely rare disease. Its association with ipsilateral renal agenesis is even more exceptional. We present herein one case of a 16-year-old male who presented with a four-month history of lower urinary tract symptoms (LUTSs) and micturition pain. The digital rectal examination revealed a small mass arising from the prostate. The urine culture showed that  A seminal vesicle cyst in combination with ipsilateral renal agenesis is rarely encountered with fewer than 100 cases reported in the literature . TreatmeWe present one case of seminal vesicle cyst with ipsilateral renal agenesis, who was successfully treated conservatively with alpha-blockers.E. coli was sensitive to all antibiotics tested and Q max at uroflow was greater than 15 mL/second. As surgery is very demanding and features many complications, we decided to manage this disease conservatively with alpha-blockers and antibiotics. After 6-month followup, the patient did not report any complain and the uroflow test was similar to a normal urination.A 16-year-old Moroccan male presented with a four-month history of LUTS and micturition pain. The digital rectal examination revealed a large, soft, fluctuant mass arising from the prostate. TRUS revealed a cystic mass measuring 5 × 4 × 3.5 cm arising from the prostate and no left kidney was seen. On MRI, a tube-like structure meandering along the rear wall of the bladder was recognized as the lower portion of the left ureter. The mass showed low T1- and high T2-weighted signal intensity compatible with a cyst in the left seminal vesicle area . Most of them are asymptomatic. The clinical presentation is either dysuria or urinary tract infection . The cysApplicable diagnostic methods include digital rectal examination, excretory urography, TRUS, CT, MRI, and cystoscopy.  Digital Sonographic findings can confirm the cystic nature of the pelvic mass and determine the relative size and location. Findings include an anechoic pelvic mass with a thick, irregular wall and occasional wall calcifications, or the mass may contain internal debris reflecting prior hemorrhage or infection .Abdominal computed tomography, magnetic resonance imaging, and seminal vesiculography are useful for the detection of accompanying deformity and differential diagnosis. A cystoscopy helps to confirm a hemitrigone, absence of ureteral orifice and other anomalies in bladder .If the condition is asymptomatic, observation without treatment is the norm, but surgical resection, transurethral resection, cyst puncture, and so forth may be performed in patients with symptoms . RecentlSurgical management would improve subjective symptoms; however it remains uncertain whether it would improve the patient's sperm properties as well . Besides


Airway gastric fistula (AGF) is a rare but catastrophic complication after esophagectomy. Surgical repair with viable tissue interposed between the airway and alimentary tracts remains the definitive treatment. However, it is challenging for surgeons, and only anecdotally described in sporadic case reports due to the complexity of the techniques necessary for successful surgical intervention. Here, we report two cases successfully managed via single-stage surgical re-exploration. On outpatient follow-up, the two Chinese patients were progressing satisfactorily without complaint of any dyspnea or dysphagia. Airway gastric fistula (AGF) is a rare but catastrophic complication after esophagectomy for esophageal cancer with an occurrence of 0.3 % to 1.9 % [th postoperative day. Although immediate esophagogram showed no evidence of anastomotic leakage, an esophagogastroscopy revealed an approximately 3-cm ischemic mucosal area covered with exudate beneath the anastomosis (Figure A 50-year-old Chinese man with a 3-week history of progressive dysphagia was diagnosed with a squamous cell carcinoma of the lower esophagus. He underwent a esophagectomy and 2-field lymphadenectomy through a right thoracotomy. The first postoperative week progressed smoothly. However, he experienced mild coughing with aspiration while given clear liquid diet on the 8s Figure A. A furts Figure B. On theHe was brought back to the operating room for an exploratory thoracotomy. Mediastinal abscess was debrided, and the gastric conduit was taken down. Then, an oval defect, measuring 2 × 6 mm, with necrosis of the membranous wall of the proximal 2 cm of the intermediate bronchus was found Figure C. All neA 48-year-old Chinese male patient was admitted to our hospital for the purpose of repair of a AGF. One year ago, he was diagnosed with lower esophageal cancer and underwent radical esophagectomy via left thoracotomy. Post-operative diagnosis was well differentiated squamous cell carcinoma, T2N1M0, Stage IIB. His initial postoperative course was uneventful. An esophagram showed no evidence of leak, and then he was given a regular diet. One month following the operation, he experienced a cough productive of gastric contents. Further evaluation by bronchoscope revealed a large AGF in the left main bronchus (Figure ® sheet was interposed between the airway and the new gastric tube (Figure His general condition remained good, and he was taken to the operating room for definitive repair. The fistula was approached through a left thoracotomy along the original incision. The gastric conduit was remobilized and resected around the fused fistula beyond 2 cm, to ensure the blood supply of the residual gastric tissue which was to be used as a patch. A fistula measuring 1 × 3 cm was then revealed (Figure Fistula between the airway and the gastric conduit following esophagectomy has been reported both early and late in the postoperative course. Early fistulas is related to postintubation injury, tracheobronchial erosion by gastric staple line, or an unrecognized thermal or blunt injury to the membranous airway. Late fistulas have been associated with chronic contained anastomotic leaks, cancer recurrence, and dilation of an anastomotic stricture. Prompt diagnosis is essential for successful treatment.Definitive treatment of AGF after esophagectomy is challenging and influenced by the location, size and the pathogenesis of the fistula, and also by the presenting symptoms and the accompanying comorbidities of the patient. Over the past years, various management strategies have been described including conservative, endoscopic and surgical treatment ,2. In ge® sheet can be used as a covering material to prevent postoperative perforation after endoscopic submucosal dissection, while here it was used as a interposition between gastric tube and bronchus [Surgical re-intervention, though fraught with high morbidity, would probably be the optimum radical treatment especially for large fistula or fistula with severe symptoms. Debridement of the septic focus, resection of the fistula, reconstruction of the airway and alimentary tracts, and interposition of various material if necessary were useful ,6. In cabronchus .If operative risk was believed to be too great owing to the instable general condition of a patient, an endoscopic stent may be temporarily used to avert ongoing contamination of the airway from gastric contents. When improvement of systemic condition occurs, a surgical re-intervention should then be considered.Management of AGF requires the multidisciplinary knowledge and individual strategy. Although promising effective therapy is not established, it is unquestionable that aggressive surgical repair produces survival benefit for properly selected patients. Early and proper diagnosis with endoscopy, control of pulmonary and mediastinal infection, aggressive consideration of surgical intervention, complete resection of the fistula and restoration of functional airway and alimentary tracts are the keys to achieving a better outcome in acceptable patients.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.AGF: Airway gastric fistula.The authors have no commercial association or sources of support that may pose competing interests.HS wrote the draft of the manuscript and obtained the written consent. WPW and QG participated in the manuscript writing and helped to the final writing of the paper and gave final approval of the manuscript. LQC participated in the manuscript revision. All authors have read and approved the final manuscript.


Leiomyoma is a benign tumour commonly encountered in the genitourinary and gastrointestinal organs in adults. Cutaneous leiomyomas are rare benign tumors arising from the arrector pili muscle of hair follicles. Cutaneous leiomyomas are more likely to occur in adults than in children. We describe a case of a 10-year-old female who presented with multiple, firm, red-brown masses on the back. A punch biopsy was performed. Under high-power examination, spindle cells with an eosinophilic cytoplasm were observed and immunohistochemical studies were performed; the cells stained strongly positive for smooth muscle actin (SMA). The patient was subsequently diagnosed with pilar leiomyoma and referred to a plastic surgeon for surgical treatment. Although cutaneous leiomyoma is a rare disorder, we identified a case of pilar leiomyoma in a young female. A careful clinical assessment led to the correct diagnosis and therapy in the present case. We propose that leiomyoma ought to be considered in the differential diagnosis of any cutaneous or mucosal mass in children. Leiomyoma is a benign tumour commonly encountered in the genitourinary and gastrointestinal organs in adults . CutaneoIn the present study, we describe a case of a 10-year-old female submitted to the Dermatology Department of Recep Tayyip Erdoğan University Medical Faculty, Rize, Turkey, with a two-month history of a lesion in the right scapular and lumbar regions. During the second month, the mass was observed to have increased in size and become painful. On clinical examination, multiple firm red-brown masses were observed on the back of the patient, the largest of which measured ∼10×15 mm and was located in the left scapular region . ThorougThis study was approved by the ethics committee of the University of Rize, Turkey. The patient consented to the publication of this study.The anatomical distribution of cutaneous leiomyomas is extensive. Leiomyomas may present clinically as either solitary or multiple lesions that have a skin-colored or red surface, and are most commonly located in the extremities . SolitarWhile solitary lesions may be easily treated by surgical excision, multiple lesions covering large areas are more difficult to treat . Our patIn conclusion, although cutaneous leiomyoma is a rare disorder we identified a case of pilar leiomyoma in a young female. In the present case, a careful clinical assessment led to the correct diagnosis and therapy. Although cutaneous leiomyomas occur more frequently in adults, we suggest that leiomyoma ought to be considered in the differential diagnosis of any cutaneous or mucosal mass in children.


Mucus-producing tumours of the appendix or mucoceles can, if left untreated, lead to dissemination of its contents into the peritoneal cavity causing substantial morbidity to the patient. Symptoms for complicated mucoceles can mimic those of acute appendicitis and the final diagnosis is most likely made intraoperatively. We here present a case that is, to our knowledge, one of only ten described in the literature and the first to characterize torsion of an appendiceal mucocele with abdominal magnetic resonance imaging.The patient, a 34-year-old Caucasian female presented at the emergency department with acute abdominal pain in the right lower quadrant. Initial diagnostic work-up including ultrasonography and abdominal magnetic resonance imaging showed a large tubular mass at the base of the appendix with indirect signs of torsion. A laparoscopic appendectomy was performed the following day where the finding was confirmed. The patient went on to have an uneventful recovery and was discharged from the hospital on the first postoperative day.Magnetic resonance imaging is a useful tool in identifying unknown lesions of the appendix and should be considered the primary imaging modality in especially younger patients requiring diagnostic imaging. In this case the preoperative imaging findings aided in choosing the correct timing and treatment option for the patient. The frequency of primary appendiceal tumours is reported to be 0.5-2% in removed specimens-4, mucocA 34-year-old Caucasian female presented at the emergency department with an 8-hour history of nausea, vomiting and abdominal pain. The patient had suffered from occasional and transient episodes of abdominal discomfort during the last year. At the day of presentation the patient first experienced diffuse upper abdominal discomfort with later sudden pain in the right lower abdomen. The patient had palpable tenderness in the right lower quadrant, without signs of peritoneal irritation. The patient was afebrile and had a hemoglobin level of 145 g/l and a white blood cell count of 10.7 E9/l. The level of C-reactive protein was normal. Urine analysis was negative as well as analysis for human chorionic gonadotropin. The patient responded poorly to analgesic medications and a gynecological consultation was requested to exclude torsion of the ovary. Vaginal ultrasonography revealed a regular cystic mass between the uterus and the right ovary. This finding was verified with transabdominal ultrasonography, which showed a tubular mass of unknown origin. In order to characterize the finding further an MRI of the lower abdomen was performed. On MRI a fluid-filled appendiceal mass (8x3x3 cm) with relative wall-thickening was observed. The surrounding structures showed inflammatory changes with edema of the appendiceal mesentery. Abrupt tapering of the appendix was seen at the base of the mucocele Figure . The abdet al. showed whorling of the appendiceal mesentery on computed tomography indicative of appendiceal torsion[et al., a cystic appendiceal mass showed a target-like appearance on ultrasonography and was later confirmed to present torsion of an AM[Secondary torsion of the appendix due to a mucocele is rare, with only 10 previous case presentations in the English literature-14. Preo torsion. In anotof an AM. Table In our case, the imaging findings were indicative of an appendiceal mucocele. The mesenteric edema and abrupt tapering of the base of the appendix were considered signs of torsion. MRI findings in AM with torsion have not previously been presented in the literature. Based on our findings, the discrimination between simple and complicated mucoceles by imaging is difficult. The patient’s sudden onset of abdominal symptoms and disease presentation were however inconsistent with classical appendicitis and the MRI findings aided in the pre-operative work-up of the patient. Notably, the appendicular wall had already developed necrosis highlighting the risk of rupture. Prompt laparoscopy provided the definitive macroscopic diagnosis and allowed for safe removal of the diseased appendix. Performing the procedure during office-hours ensured access to histopathologic analysis and confirmation that there was no neoplastic involvement of the caecum. The underlying pathology of the mucocele could however not be established on frozen sections. In cases where resection margins are negative and the muocele intact simple appendectomy is considered sufficient. Frozen It is the opinion of the authors that the threshold for performing imaging studies in patients with suspected appendicitis and atypical disease presentation should be low, in order to identify lesions that preferably should be operated on during daytime. To avoid complications and the need for reoperation in cases like the one presented here, the availability of senior surgeon and pathologist consultations during the operation is recommendable. Abdominal MRI is a useful tool in identifying unknown lesions of the appendix and should be considered the primary imaging modality in especially younger patients requiring diagnostic imaging when ultrasonography is inadequate.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.CS prepared the manuscript, and it was reviewed by MJ and BE. MJ analyzed the imaging results and prepared the MRI images. BE performed the appendectomy assisted by CS. All authors read and approved the final manuscript.CS is a resident completing his basic surgical training at Päijät-Häme Central Hospital (PHCH) and a PhD student at the University of Turku. BE is a senior surgeon at PHCH. MJ is a consulting radiologist at PHCH.


Postsurgical pathological diagnosis revealed “carcinosarcoma”.Primary liver carcinosarcoma is a very rare disease. There have been only a few cases described to date, none of which has been imaged by bone scintigraphy. A 69-year-old man who developed right back pain and weight loss was admitted to our hospital. Tenderness of the right upper abdomen, hepatomegaly, and a giant mass were the main physical examination signs. Abdominal ultrasonography showed a large lobulated heterogeneous echogenic solid mass with multiple cystic areas of varying size and a highly echogenic region that had posterior acoustic shadowing within the mass in the right lobe of the liver. Labeled erythrocyte imaging and Conflict of interest:None declared. Primary liver carcinosarcoma is a very rare disease. They represent less than 1% of primary malignant tumors of the liver ,3,4,5,6.A 69-year-old man who developed right back pain and lost weight (12 kg in the last 2 month) was admitted to our hospital. Tenderness of the right upper abdomen, hepatomegaly, and a giant mass were the main physical examination signs. Serum markers for hepatitis B was positive, but the patient had no evidence of cirrhosis. He had moderately elevated bilirubin levels , direct bilirubin 0.35 mg/dl (0-0.2 mg/dl)) and the other biochemical and hepatic function tests were as follows: erythrocyte sedimentation rate 103 mm/h, AST 65 U/l (0-35 U/l), ALT 34 U/l (0-45 U/l), alkaline phosphatase 382 U/l (30-120). The tumor markers revealed negative results: alpha-fetoprotein 2.7 ng/mL (<9.6 ng/mL); carcinoembryonic antigen 1.5 ng/mL (<5.0 ng/mL); serum carbohydrate antigen 19.9 10.97 U/mL (<39 U/mL).Abdominal ultrasonography showed a large lobulated heterogeneous echogenic solid mass with multiple cystic areas of varying size and a highly echogenic lesion that had posterior acoustic shadowing within the mass in the right lobe of the liver.On magnetic resonance imaging, this mass covered segments 7-8 completely and segment 4 partially and observed to be 152x120x135 mm in diameter. It was heterogeneously hypointense on the T1-weighted image  and hete99mTc-nanocolloid liver-spleen scan  of 99mTc-MDP, the patient was imaged on a dual-head coincidence camera (Siemens) with a low-energy high resolution (LEHR) collimator attached. Standardized anterior and posterior whole-body images were obtained. Images were acquired on a 256x1024 matrix using a scan speed of 12 cm/min. This study  and sarcomatous elements, including malignant mixed tumors. This neoplasm consists of increased cellular pleomorphism, spindle cell stroma with abundant mitoses, osteogenesis, and atypical osteoid formation .On MRI, hepatic carcinosarcoma is hypointense on the T1-weighted image and heterogeneously hyperintense on the T2-weighted image. Calcification and ossification, which are better seen on CT, have low or dark signal intensity on T1- and T2-weighted images ,9.A literature review showed that there are few reported cases of this type of primary hepatic tumor ,10,11,1299mTc MDP bone scans are primarily used to assess skeletal pathology, many other non-osseous lesions may accumulate bone seeking radiopharmaceuticals. These are often identified incidentally, while assessing underlying skeletal abnormalities. Extra osseous uptake of bone-seeking radiopharmaceuticals is seen in a wide variety of pathologic processes involving almost any organ. Although some possible mechanisms were proposed, soft tissue uptake of phosphate compounds such as diphosphonates is not well understood. Cause of extraosseous uptake of bone seeking radiopharmaceuticals may be related to ion exchange between intracellular calcium phosphates, increased calcium content of the tissue and the possibility of high concentration of phosphatase enzyme systems in certain tumors (While n tumors .99mTc diphosphonates is an unusual finding in nuclear medicine practice. There is a need for awareness of the pathophysiologic basis underlying such uptake, as it may be of critical clinical relevance in the evaluation of the patient. Localized areas of increased tracer uptake in the hepatic parenchyma are usually due to primary hepatic carcinoma or liver metastases.Hepatic uptake of In conclusion, the scintigraphic findings of hepatic carcinosarcoma presented in this case showed a large mass with irregular, dense activity accumulation thought to be osteoblastic activities. Especially if localized dense activity accumulation giving a mass image is seen in bone scan, carcinosarcoma should be included in the differential diagnosis of a large hepatic mass.


Hemangiomas of the external auditory canal, involving the posterior bony canal and the adjacent tympanic membrane, although rare, are considered a specific disease entity of the human external auditory canal. Hemangiomas of the tympanic membrane and/or external auditory canal are rare entities; there are 16 previous case reports in the literature. It is a benign vascular tumor. It generally occurs in males in the sixth decade of life. Total surgical excision with or without tympanic membrane grafting appears to be effective in the removal of this benign neoplasm. The authors present a case and a review of the literature discussing diagnostic and surgical approaches. Hemangiomas are benign vascular lesions that are most common in infancy and childhood. The tumor occurs frequently in head and neck. One third of these lesions will present at birth. Besides, 20% of hemangiomas are multiple. Hemangiomas of the tympanic membrane and/or external auditory canal are rare entities, with 16 previous case reports in the literature. It is a benign vascular tumor. It generally occurs in males in the sixth decade of life. It appears as a small vascular lesion interesting the deep posterior bony external auditory canal and/or the posterior superior tympanic membrane. The first two cases of hemangioma of the tympanic membrane and posterior external ear canal were reported by Freedman et al. in 1972 .We present a case and a review of the literature discussing diagnostic and surgical approaches.A 55-year-old man presented with a 3-month history of left otalgia, hypoacusia, and pulsatile omolateral tinnitus.The otoscopic examination revealed a purple exophytic ovoid nonpulsatile lesion of the superior quadrant. It had pedicle in correspondence of malleus's manubrium .A CT scan showed a round smooth mass (3 mm of diameter) external to the tympanic membrane . There wA transcanal surgical resection was performed. The lesion was removed, leaving the fibrous and the mucosal drum layers intact.Pathologic examination revealed a well-defined proliferation of dilated tortuous vascular structures, closely adhering to each other, filled by erythrocytes in the lumen. They varied in size and were delimited by a single layer of flat endothelial cells. Superficially, the lesion was lined by hyperkeratotic squamous epithelium. These features were consistent with a cavernous hemangioma . The posHemangiomas involving the tympanic membrane (TM) and/or the adjacent posterior bony external auditory canal (EAC), although rare, do appear to be a specific disease entity of the human external auditory canal. Till now in the literature, there have been 17 reported cases of this disease . The hemTympanic hemangiomas, 40% of external ear hemangiomas, arise from the lamina propria of the TM and has pedicles in the posterior part of the tympanic membrane , 13, 14.From the histological point of view, in the overwhelming majority of cases, these neoplasms appear as cavernous hemangiomas (13 out of 17 cases). Capillary haemangiomas are rare (3 out of 17 cases) , 11, 13,The hemangioma may be incidentally found, not accompanied by any symptomatology, whether interesting only the TM or extending to the EAC (40% of cases). Sometimes, it is accompanied by rather nonspecific otologic symptoms: hearing loss, fullness, tinnitus, and otorrhea.Differential diagnosis includes glomus jugulare or tympanicum, high jugular bulb, aberrant internal carotid artery, and arteriovenous malformation. In particular, the presence of bone erosion, the skull base relationship, and the middle ear status are essential in the aim of differential diagnosis. For this reason, temporal bone CT scan is considered the first choice to evaluate these entities. MRI is useful to carefully describe vascular lesions but does not provide any data regarding bony structures. For advanced lesions, an angiographic examination is advisable in order to plan an embolization before surgery .The analysis of the cases, described so far, shows how hemangioma may occur either with exclusive involvement of the tympanic membrane, or EAC, or as a more extensive lesion affecting both sites. In the first situation, the surgical excision is relatively easy; we simply have to remove the external layer of the tympanic membrane, and rarely do we have to perform a myringoplasty in order to close a tympanic perforation . In the All authors, except Magliulo, exclude the involvement of middle ear or mastoid. In fact, in no case histological examination after the surgical procedure showed involvement of the middle ear and/or mastoid. Even in the case described by Magliulo, hemangioma interested in full-thickness the tympanic membrane in correspondence of the handle of the hammer but without signs of erosion or pathology of the same . In the


We present a 25-year-old male patient with a diagnosis of multiple enchondromatosis, who developed chondrosarcoma on the proximal humerus of the right upper limb. The patient had the pre-existing lesions of Ollier’s disease discovered during his childhood. The patient underwent wide resection of the sarcoma with a prosthetic replacement of the proximal humerus. So far we have followed up the patient for 8 years with no evidence of local recurrence and/or metastasis. The therapeutic results have been satisfied with a good functional recovery of the treated limb, enabling the patient to return to the pre-disease daily living and occupational activities. The reconstructive procedures represent an effective surgical strategy for limb salvage in the treatment of large segmental defects after resection of humeral tumors, substantially solving the functional and esthetic problems due to such a wide resection, and significantly improving the quality of life for the patient. Ollier disease is a rare non-hereditary skeletal disorder characterized by the presence of multiple enchondromas (enchondromatosis), and these cartilaginous lesions can be limited to one limb, or localized to one half of the body , 2. The A 25-year-old male patient was admitted to our department, presenting progressive pain and numbness with a rapidly growing mass located in the upper part of the right arm for 2 months in 2005. At the time of the hospital admission, he had completely lost his occupational capacity of the arm (100% unable to work). Physical examination revealed a palpable mass on the upper part of his right arm with a size 13 × 12 cm. Radiographic examination demonstrated a large mass over 10 cm in diameter on the proximal humerus, with massive cortical erosion, extension of the tumor into soft tissues and indistinctness of the surface of the tumor . A diagnA limb-salvage strategy with treatment of the large segmental defects following resection of the tumor and a prosthetic replacement of the proximal humerus was designed for the patient. The informed consent was obtained from the patient prior to the operation. The tumor resection prosthesis was applied in the same session following the tumor resection . The tumDuring the 8-year follow-up, clinical and radiologic examinations were done at the periodic controls. There are no signs of local recurrence and/or remote metastasis so far. The therapeutic results have been satisfied with a good functional recovery of the treated limb, enabling the patient to return to the pre-disease daily living and occupational activities. The overall functional outcomes were assessed by the musculoskeletal tumor society (MSTS) scoring system. The patient had the overall score 24 in the last examinations of the follow-up, and the ranges of motion of the shoulder joint are shown in Proximal humerus tumors treated with amputation usually result in a complete loss of the hand/limb function. Limb-salvage procedures have been popularly put into clinical practice in spite of amputations in bone tumor surgery. However, the surgical procedures remain challenges for orthopedic surgeons toward simultaneous replacing the defects of bone/soft tissue and restoring the functions of the involved joints/limbs after tumor resection. Preservation of the functional capacities of the involved upper extremity along with a complete removal of the tumor is considered as the mostly important criteria in the surgical management of the bone tumor of the proximal humerus. Moreover, concerns also should be paid for preservation of length and cosmesis of the upper extremity toward supporting quality of life for the patients. The indications for the prosthetic replacement of the proximal humerus tumors as proposed by Ross et al  have beeIt is worth noting that good function of a treated upper limb with the procedures is largely based on the stability of the shoulder joint, while the stability of the shoulder joint can only be ensured by reliable reconstruction with minimizing the amount of resection of muscle and soft tissue within the complete removal of the tumor during surgery . Various


Increased deposition of cementum is observed in a wide number of both benign and malignant conditions. Many cases are often diagnosed during routine examination as an incidental finding. Diagnosing correctly without confusing it with other similarly appearing lesions, thus avoiding subjecting the patient to unnecessary investigations and stress, is of prime importance. We report one such case, where the patient presented with the routine complaint of a painful tooth, during the investigation of which he was also diagnosed with hypercementosis affecting the mandibular second premolars and molars bilaterally. The literature review reveals that not many cases of hypercementosis are frequently reported. A 60-year-old male patient reported to a private outpatient clinic with the chief complaint of pain in the right lower back tooth region for a duration of two weeks. The patient's past medical history was uneventful and he was apparently healthy. On intraoral examination, root stump of the mandibular second premolar was seen, surrounded by inflamed gingiva, and the adjacent first molar was affected by caries, supraerupted and mobile. Both the teeth were associated with tenderness. An intraoral periapical radiograph of the region was taken . The radThe term cementum is derived from the Latin word cementum or “cement,” meaning the stone particles used to make mortar, ideally describing its role . The cemExcessive deposition of cementum occurs in a wide spectrum of neoplastic and nonneoplastic conditions, such as benign cementoblastoma, cementifying fibroma, periapical cemental dysplasia, florid cementoosseous dysplasia, hypercementosis, and other benign fibroosseous lesions of periodontal ligament origin . HyperceHypercementosis may be localized to a single tooth or few teeth in a quadrant or may be generalized affecting multiple teeth. The local causes suggested include an adaption to functional changes, trauma, inflammation . GeneralExtent and pattern of cementum deposition vary among tooth groups and tooth surfaces . In a stRadiographically, teeth affected by hypercementosis may show either a generalized or localized enlargement. Generalized hypercementosis is commonly either of the dense type or the transparent type . In the Microscopically, hypercementosis appears as thick layers of cementum characterized by symmetric, highly basophilic lines parallel to the dentin surface .Since hypercementosis presents with no clinical signs or symptoms, it is conservatively managed. Surgical excision is usually indicated in suspicious, atypical cases , 5.Hypercementosis is a rarely reported frequent condition. When presented with a case, it is important to differentiate it from other similarly appearing lesions and additionally rule out any underlying systemic causes. It is important to identify the need for invasive management. Additionally, such cases may present an endodontic or orthodontic challenge, which has to be managed accordingly.


Clear cell meningioma is a rare subtype of meningioma seen mainly in pediatric patients. Supratentorial location is an unusual site of occurrence and its natural history and prognosis are not well described in the literature. We present a unique case of a left-sided frontoparietal tumor in a 15-year-old child who was managed successfully with gross total surgical excision and is recurrence-free at 18 months follow-up. A favorable clinical behavior and a longer symptom-free interval can be expected after gross total removal. The patients should be followed after successful surgery and other modalities of therapy should be used for recurrence. Meningiomas occurring in the first two decades of life are uncommon, accounting for only about 1-2% of all brain tumors.1–5 The ocA 15-year-old male child presented with history of worsening right-sided weakness of 2 years duration. He had progressive vision loss in the left eye for the past 1 year. He had multiple episodes of focal-onset generalized tonic clonic seizures with poor drug compliance. Headache with projectile vomitings had started lately. On clinical examination, the patient was blind in the left eye, with a normal right eye. There was mild right-sided seventh nerve weakness and hemiparesis. A magnetic resonance imaging scan of the brain revealed a left-sided large supratentorial tumor extending from the brain surface in the frontal and parietal cortex to the atrium and frontal horn of the lateral ventricle, causing significant compression . There wCCM is a recently described histological subtype of meningioma and is reported to occur more frequently in the pediatric age group.5–8 The m14et al. reported only two pediatric patients as having supratentorial intraparenchymal CCM.[5Supratentorial location of pediatric CCM is rare. In a review of 35 cases of intracranial CCM, Ma mal CCM.5 They tremal CCM.5Radiologically, the lesions show strong and homogenous enhancement following contrast injection. The hist31This child presented with a lesion in the left frontoparietal region. Such a location of the tumor with absence of any subarachnoid or CSF seedling is extremely rare. The radiological study revealed the tumor to be heterogenous in consistency with solid and cystic components and had a nonuniform enhancement after gadolinium injection. At surgery of this child, it was revealed that the cyst had caused an impression on the inner surface of the parietal bone, which may be suggestive of the gradual progression of the tumor. With reference to the literature review, CCM may have benign histological characteristics, but, clinically, it shows a high risk of progression and recurrence. Given the large dimension of the tumor (6-7 cm), local impression on the bone and long duration of symptoms, the rate of growth of the tumor seems to be slow in this case. The MIB-1 index in the patient was 8%, making the course of illness unpredictable. The recurrence rate for these tumors has mainly been reported with reference to spinal CCM and the guidelines for therapy are based there upon. Given the behavior of the tumor in the present case, we can safely recommend a close follow-up in the postoperative period, with radiotherapy reserved for cases with recurrence.


A 13-year-old boy with a history of bicycle handlebar injury to the left side of his abdomen was brought to the Emergency Department of our center. On admission, his vital signs were stable and abdominal examination revealed ecchymosis and tenderness of the injured areas. Mild to moderate free fluid and two small foci of free air in the anterior aspect of the abdomen, in favor of pneumoperitoneum, were detected in abdominopelvic sonography and CT-scan, respectively. In less than 6 hours, the patient developed generalized peritonitis. Therefore, exploratory laparotomy was promptly performed, which revealed appendiceal transection and rupture of the small bowel mesentery. Appendectomy and small bowel mesoplasty were done, with pathological diagnosis of acute appendicitis and periappendicitis. After surgery, the patient had a non-complicated hospital course. This rare case highlights the significance of the early management of appendiceal traumatic injuries in order to prevent further complicated events, especially in patients who are much more exposed to this risk due to their traumatic background. Abdominal trauma is assumed to have an indirect causative role.3 There is still doubt as to the direct pathogenic effect of abdominal trauma in acute appendicitis;4-6 nevertheless, further appendiceal injuries would render the situation far more challenging. This report presents a 13-year-old boy with manifestations of generalized peritonitis within a few hours after a blunt abdominal trauma, in whom exploratory laparotomy revealed an appendiceal transection.Trauma and acute appendicitis are the two most common entities of childhood surgical emergencies.st, 2012, following a collision between his bicycle and a motorcycle, during which the patient had the left side of his abdomen injured by his bicycle handlebar. On admission, the patient only complained of left flank pain. A 13-year-old boy was brought to the Emergency Department of Nemazee Hospital, Shiraz, Iran, at 10:00 p.m., June 13, and platelet count of 313000/mm3. Other blood tests and urinalysis were normal. The findings of plain chest and abdominal graphies were inconclusive, and abdominopelvic sonography showed mild to moderate free fluid in the abdominopelvic cavity. Spiral abdominopelvic CT-scan revealed two small foci of air in the anterior aspect of the abdomen, in favor of pneumoperitoneum. Two areas of faint increased density and fat stranding in the subcutaneous fat of the left side of the abdomen and another one in the anterior right side of the abdomen were detected, all due to post-traumatic changes. A small area of increased opacity, resembling hematoma, was seen inferior to the spleen and lateral to the psoas muscle in left lower abdominal quadrant  yielded a hemoglobin count of 13.5 gr/dl, white blood cell count (WBC) of 14700/mmquadrant . Within 4 It is thought that there is a multifactorial mechanism indirectly contributing to blunt trauma-induced acute appendicitis. As a complication of internal or external blood loss, visceral hypoperfusion occurs. The following reperfusion will lead to visceral edema, resulting in a rise in intra-abdominal pressure (IAP). This condition is deteriorated by extensive fluid resuscitation and diminished size of the abdominal cavity by other traumatic complications such as intraperitoneal bleeding or free air occupying space, retroperitoneal hematoma, acute gastric dilatation, and external compression. An abrupt rise in IAP increases intracecal pressure and a subsequent rapid distension of the appendix, resulting in mucosal abrasion. Both mucosal abrasion and decreased blood flow will end in acute appendicitis and its complications.7 Despite this complex mechanism, the association between acute appendicitis and blunt abdominal trauma is still controversial. Due to their high prevalence, some have considered their coexistence as an incidental event, while others have argued that acute appendicitis may cause the patient to be vulnerable to a traumatic event.4 In our case, visceral hypoperfusion and resultant increased IAP does not seem to have a pathophysiological role due to the absence of a significant volume loss.The appendix is a highly mobile, small structure so that it is rarely affected by direct blunt abdominal trauma.Appendiceal rupture after blunt abdominal trauma is also exceedingly rare. Whether appendiceal rupture occurs as a complication of advanced acute appendicitis or as a consequence of direct blunt abdominal trauma has yet to be fully clarified. In our case, concurrence of appendiceal rupture and acute appendicitis rendered it difficult to determine which one was prior to the other. As we mentioned, the patient had been asymptomatic before the trauma and there was no histopathological evidence of advance acute appendicitis to be responsible for the subsequent appendiceal rupture. Furthermore, consideration of transaction as an antecedent event does not justify the pathologic report of inflammation because of trauma-induced vascular injury and tissue ischemia. 8 In 1977, a 30-year-old man was reported to have developed acute abdominal pain two days after a blunt severe direct trauma to the abdomen. Surgical exploration revealed appendix avulsion from its distal three quarters with fibrinopurulent mucosa and surrounding bruising of the cecal wall. Consequently, appendectomy and caecostomy were performed. Nonetheless, the patient experienced a complicated postoperative course due to the formation of multiple subcutaneous parastomal abscesses and resultant septicemia.2 Reviewing the literature lists a few other such conditions.9-11 However, we found only one case of bicycle handlebar injury presented by acute appendicitis. In the said case, the bicycle handlebar had injured the lower abdomen and symptoms started 2 days after the trauma with the diagnosis of perforated suppurative appendicitis in pathological examination.12 The appendiceal transection in our case is in fact a contrecoup injury due to the opposite primary side of the handlebar harmful contact, which was visible in the left part of the patient’s abdomen. It is also worthy of note that in the majority of the available reports, late presentation of symptoms features prominently. Furthermore, in a patient with trauma, diagnosis of acute appendicitis is difficult and may cause delay in early management.13 It may contribute to more complex pathologic forms of acute appendicitis. In our case, rapid development of the symptoms and signs of generalized peritonitis hinted at chemical peritonitis, which was subsequently confirmed by our observations during exploratory laparotomy. Our early management precluded such further problems as fibrinopurulent peritonitis and its complications.Appendiceal rupture was first reported in 1938 with a two-week history of pneumatic drill use resting on the right iliac fossa.Despite the hitherto uncertain relationship between blunt abdominal trauma and acute appendicitis, consideration of this rare co-existence would result in early management and an uneventful postoperative course.


We report two clinical cases of primary granulocytic sarcoma of the pancreas that were diagnosed on the surgical specimen. Atypical clinical and morphological presentations may have lead to pretherapeutic biopsies of the pancreatic mass in order to indicate primary chemotherapy. Literature review of this rare clinical presentation may help physicians to anticipate diagnostic and therapeutic strategies. Granulocytic sarcoma (GS) is an extramedullary solid tumor mass composed of immature myeloid cells . GS is aThe first patient was a 45-year-old woman, without significant comorbidity, who was referred to our institution for surgery. Epigastric pain with jaundice began one month previously without performance status alteration. Standard blood exams exhibited cholestasis  and hyperamylasemia (1.9 N) with normal values of hemoglobin, white blood cells, platelets, carbohydrate antigen 19-9 (CA19-9) and carcinoembryonic antigen (CEA). Abdominal computed tomodensitometry (CT scan), magnetic resonance imaging (MRI) and endoscopic ultrasonography (EUS) of the pancreas all identified the distension of both the common bile duct (15 mm) and the Wirsung duct (6 mm), above a 28 × 20 mm irregular, hypoechoic and hypodense mass of the pancreatic head, without any lymph node or vascular invasion or distant secondary lesion detected. Based on the symptoms, a suspected diagnosis of pancreatic adenocarcinoma and a resectable mass, it was determined to proceed with primary surgery without obtaining preoperative sample biopsies. Curative whipple pancreaticoduodenectomy with regional lymphadenectomy was performed with no specific peroperative discovery and uneventful postoperative course. Histological examination of the surgical specimen revealed a pancreatic GS based on the presence of cells of myeloid lineage with positive immunostaining for CD43 myeloid-associated antigen Figure , whereasThe second patient was a 19-year-old woman, without significant comorbidity or any alcohol consumption, who presented at our institution for epigastric pains combined with hyperamylasemia (1.7 N) and hyperlipasemia (7.8 N). Hemogram, hepatic enzymes, C-reactive protein, CEA and Ca 19.9 values were normal. The abdominal CT scan showed a 9-mm Wirsung duct dilation Figure  within tGS, also called chloroma, refers to the infrequent green color observed as a result of myeloperoxydase action in neoplastic cells . GS usuaGS can occur in patients of all ages with a focus on male patients  during the last decades of life  ,16. EvenClinical behavior and response to therapy were not influenced by any of the following factors: age, sex, anatomic site, de novo presentation, histotype, phenotype or cytogenetic findings . It remaThese observations show that clinicians should think about pancreatic GS when the pancreatic mass develops during or after AML. However, in the cases reported here in which GS was the first and the only manifestation of AML, diagnosis is challenging. Because surgery is not required and may probably worsen the prognosis due to the delayed administration of induction chemotherapy, all efforts should be made to obtain pretherapeutic biopsies for a pancreatic mass, especially if all of the biological and morphological exam results are not typical and in agreement. The negative value of CA19.9 as well as the young age of our patients may have been warnings that indicate the value of EUS cytological examination for detecting differential diagnoses of pancreatic adenocarcinoma.A positive diagnosis of GS is sometimes challenging and requires expert pathologists. Histological observation reveals myeloblats, promyelocytes and sometimes neutrophils. The definitive diagnosis of GS requires positive immunostaining for at least one of the myeloid-associated antigens  associated with negative immunostaining for the lymphoid lineages (CD3 for T-cells and CD20 for B-cells) . Major dThe risk of metachronous AML occurrence in non-leukemic patients with GS is very high, with a median delay of 5 months; most patients will develop AML within 1 year ,12. TherThe authors described two cases of isolated granulocytic sarcoma of the pancreas. The experience of these cases highlighted the difficulties of correct diagnosis and care. To conclude, pretherapeutic biopsies should be the cornerstone for the diagnosis of a pancreatic mass with atypical clinical presentation.Written informed consent was obtained from the patient for publication of this case report and the accompanying images. For the patient who died, consent was sought from the next of kin of the patient.The authors declare that they have no competing interests.Dr. DA and Dr. CC contributed to data collection. Dr. MM and Pr. CM contributed to writing the manuscript.


Granulocytic sarcoma (GS) is a rare extramedullary tumor composed of immature myeloid cells. It is usually associated with leukemia or other myeloproliferative disorders. It occurs very rarely without overt hematologic diseases. A 19-year-old man presented with left knee mass. Biopsy with pathological analysis showed lymphoma aspect. Immunostains yielded the diagnosis of GS with myeloperoxidase and CD43 positivity. There was no systemic manifestation of leukemia, and bone marrow biopsiy was negative for neoplastic infiltration. Chemotherapy by CHOP was efficient, and the patient remaind alive and healthy 40 months after the end of treatment. The case is discussed in the framework of the existing literature about the diagnosis, treatment, and prognosis of this very rare condition. Granulocytic sarcoma (GS) is a rare localized tumor composed of immature granulocytic precursors. These neoplasms are known as monocytic sarcoma and chloroma which refers to the green coloration of some of these tumors on gross examination . It is uThe most common sites of involvement are the skin, lymph nodes, soft tissues, and bone, but the tumor can occur virtually anywhere –6. AlthoA 19-year-old Berber Moroccan man began experiencing pain in his left knee. His medical history was otherwise unremarkable. Physical examination revealed no abnormality except for an 8 cm, mobile mass of left knee with no involvement of overlying skin. CTscan revealed a large mass of anterior left knee involving bone and soft tissue suspicious of sarcoma . A radioBiopsy showed a green colour of the tumour, and pathological analysis revealed diffuse proliferation of round cells with medium-sized or large nuclei lymphoma aspect. Immunostains showed myeloperoxidase and CD43 positivity and leukocyte common antigen (LCA) and CD 3, 5, 56 negativity indicating a diagnosis of a well-differentiated granulocytic sarcoma. There was no systemic manifestation of leukemia. The blood cell count, thoracoabdominal CTscan, and bone marrow histology were normal. The patient received 6 cycles of chemotherapy by CHOP . The patient responded well to the treatment, and complete remission was achieved. He remained alive and healthy 40 months after the end of treatment. GS is a rare extramedullary tumor that consists of immature granulocytic cells. this tumor was first described in 1811 and originally called chloroma by King in 1853 . The terGS usually presents concomitantly with or after the onset of acute myelogenous leukemia, blastic phase of chronic myelogenous leukemia, or myelodysplastic syndrome . HoweverMacroscopically, GS is usually green in appearance. The green color, which is due to the presence of myeloperoxidase (MPO) in the leukemia cells, is not present in all tumors of this type . AccordiThe rarity of cases of nonleukemic GS has limited the availability of prospective studies on the effectiveness of various treatment strategies. Although no standard treatment protocol has been established, several previous reports suggested that local treatments such as surgical excision and/or radiation therapy alone would not be sufficient for the treatment of nonleukemic GS , 2, 14 aThe long-term prognosis of nonleukemic GS remains poor , and theGS is rare, and clinical diagnosis is especially difficult without bone marrow involvement. Immunohistochemical studies using an appropriate panel of markers are extremely helpful to make a correct diagnosis. The strategy for the management of nonleukemic GS should be individualized. Aggressive chemotherapy is beneficial and may induce complete remission. Early diagnosis and strict followup of these patients are essential for a better outcome in GS.Written informed consent was obtained from the patient's father for publication of this case report. A copy of the written consent is available for review with the Editor-in-Chief of this journal.The authors declare that they have no conflict of interests.I. Elghissassi conceived the case report, analyzed and interpreted the patient data regarding the haematological disease H. Innrhaoun analyzed and interpreted the patient data and was a major contributor in the literature review H. Mrabti analyzed and interpreted the patient data and was a major contributor in the literature review H. Errihani contributed to revision, supervision and approval of the work. All authors read and approved the final manuscript.


Introduction. Ten cases of dome-type adenocarcinoma of the colon have been reported so far. Most of them were presented as early lesions, with endoscopic and microscopic distinguishing features. Methods and Results. A raised plaque was removed from the right colon during colonoscopy in a 56-year-old man. Histopathological examination showed a cancerized adenoma invading the submucosa with several typical features of dome-type adenocarcinoma, in particular the associated prominent lymphoid tissue. Immunohistochemistry showed retention of the mismatch repair proteins MLH-1, MSH-2, MLH-6, and PMS-2. Conclusion. We report an additional case of dome-type adenocarcinoma of the colon as an early, low-risk, and microsatellite stable tumor, indicating that this particular histotype may deserve specific consideration for both classification and management. Since the initial reports back in late 90's & 2000's by De Petris et al.  and JassOriginating from this specific microenvironment, the most important morphological feature of DC is the association with a prominent lymphoid stroma.We would like to add an additional case of this particular histotype that was recently diagnosed in our institute.A 56-years male with amyotrophic lateral sclerosis was colonoscoped because of painful constipation. There was no case of colorectal cancer in his family history.An 8-mm raised plaque was seen in the right flexure  and remoRoutine histopathological examination showed a cancerized adenoma invading the submucosa associated with expanded lymphoid tissue encompassing several reactive germinal centres Figures .The cancer, arisen in a flat adenoma with high-grade dysplasia, was ulcerated superficially and the advancing edge appeared quite well circumscribed except for a more submucosa-invading tongue of neoplastic glands where some low-grade budding was observed Figures .The glands were in part cystically dilated containing a pink eosinophilic material, in part cribriform arranged Figures .A clear space often separates the glandular epithelium from the intraglandular material.Neoplastic cells lining the glands were columnar, single-layered, well-differentiated, eosinophilic . No tumor infiltrating lymphocytes nor goblet cells were observed. Necrosis and desmoplasia also were absent.The T1 adenocarcinoma was considered a low-risk lesion because of the absence of lymphovascular invasion, the low-differentiation grade, and the negative resection margin, therefore no hemicolectomy was performed.Immunohistochemistry showed retained expression of the mismatch repair proteins MLH-1, MSH-2 MSH-6, and PMS-2 in the neoplastic cells as well in the internal control Figures , resp..The patient underwent a follow-up colonoscopy one year later: the mucosal biopsies from the polypectomy site showed at histology mild fibroinflammatory changes.DC may develop as sporadic-type colon cancer or in association with ulcerative colitis,  familialSome distinguishing features both macroscopic and microscopic are constantly present: the nonpolypoid appearance, the cell architecture, the cytology, and the presence of prominent lymphoid tissue; other features such as the tumor infiltrating lymphocytes, the intra-acinar necrosis, the remnants of a preexisting adenoma and foci of usual-type adenocarcinoma of the colon may be present or absent.Most cases are reported in the early growth phase: eight over ten case reported are T1N0,  one is TThe case reported here of DC is another early and low-grade lesion, lacking features of biological aggressiveness, microsatellite stable tumor, suggesting that this particular histotype may deserve a space in the classification of tumors of the colon and rectum.per se may identify a low-risk malignant lesion, influencing the treatment decision-making process.In the management of neoplastic colonic polyps DC


Spermatocytic seminoma (SS) is a rare testicular neoplasm characterized by a palpable, painless, slowly enlarging mass in the testis. Even more rare is a synchronous bilateral presentation. Only eight cases of bilateral SS have been reported in the literature, of which three cases were present with synchronous testis enlargement, and five were sequential. Here, we report an additional case of synchronous bilateral SS and present a comprehensive relevant literature review concerning clinical features, histopathology, and treatment. Spermatocytic seminoma (SS), a rare testicular neoplasm differing from classical seminoma, usually presents in older men (>50 years of age) and occurs only sporadically in younger men (<30 years of age) -3. SS haA 48-year-old man presented in June 2011 complaining of gradually increasing left testicular painless swelling for 2 years. For the immediate 2 previous months, he found his right testicle enlarging rapidly. There was no history of cryptorchidism, bilateral scrotal pain, voiding complaints, local trauma, weight loss, or hereditary disease. A comprehensive physical examination revealed bilateral testis enlargement and displayed firm consistencies to palpation. A huge, well-defined, non-tender, left testicular mass was palpable in the scrotum. The patient’s right testicle had similar palpatory findings as his left testicle, while his inguinal lymph nodes were not palpable. Scrotal ultrasonography  revealed a well-defined 75 × 45 × 40 mm left testicular multilocular tumor and an 80 × 50 × 45 mm right testicular solid tumor with heterogeneous echogenicity  was performed. The cytological smear revealed malignant tumor cells present in both testes with bloody fluid background Figure . The tumOn gross examination, the left testicle measured 7 × 4 × 3 cm and weighed 108 g, while the right testicle measured 7 × 4 × 4 cm and weighed 118 g. Both masses had fleshy, pale-grey cut surfaces and were confined to the testis with no invasion or penetration of the tunica. There were no overlying skin changes. A histological examination confirmed the SS Figures . The datFollowing the operation, the patient was followed closely without any adjuvant therapy and was in good condition with no evidence of metastasis 18 months after the operation.SS is a distinctive, uncommon testicular germ cell neoplasm, first described by Masson in 1946  and 1% oDMRT1, a male-specific transcriptional regulator, was a likely candidate gene for involvement in the development of SS [SS is found exclusively in the testis and is not associated with any known risk factors for germ cell tumors including cryptorchidism, subfertility, or gonadal dysgenesis . These tnt of SS . Most cant of SS . SS usuant of SS . Raiss ant of SS  and Carrnt of SS . Our revnt of SS . Therefont of SS  and is gnt of SS . Matoskant of SS . In our Histopathologically, the presence of sheets and cords of a mixture of different sized cells characterize SS with similarities to spermatogonia and spermatocytes . These oIn most cases, classical seminomas show diffuse positivity for CD117, PLAP, and OCT4. However, many of the markers useful in other types of germ cell tumor are generally negative in SS . PositivA preoperational SS diagnosis is unlikely due to its rarity and similarity with classical seminoma. Saran and colleagues applied FNAC to diagnose a case of SS and confirmed it by histology . The autRadical orchiectomy via an inguinal approach followed by adjuvant is a typical malignant testicular tumor therapy treatment. Although sporadic cases of metastatic SS have been reported, SS is an indolent neoplasm that rarely metastasizes and bears an excellent prognosis . Hence, SS is a rare testicular tumor, with bilateral sequential SS presentation being even more rare. It differs from classical seminoma especially by its behavior, characterized by an almost complete inability to metastasize with only very few examples described with metastatic behavior. FNAC is a procedure used for precluding malignant tumors. However, histopathology is essential to confirm the diagnosis. In spite of scarce evidence of bilateral SS metastasis, long-term periodic surveillance remains necessary.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.FNAC: Fine-needle aspiration cytology; H&E: Hematoxylin and eosin; PLAP: Placental-like alkaline phosphatase; SS: Spermatocytic seminoma.The authors declare that they have no competing interests.NX and FL performed the operation; RT and MS collected relevant literature and modified the draft; KG wrote the initial draft; LL checked the manuscript; all authors read and approved the final manuscript.


Ischemic colitis is one of the most common intestinal ischemic injuries. It results from impaired perfusion of blood to the bowel and is rarely caused by vasculitis. We report a case of ischemic colitis revealing polyarteritis nodosa (PAN) in a 55-year-old man. Histological examination of the resected colon led to the diagnosis of PAN. A 55-year-old man admitted with acute abdomen surgery proved to be necessary. Histological examination of the pathologic specimens revealed fibrinoid necrosis and destruction of the internal lamina in small and medium size arteries. Although the gastrointestinal tract is frequently involved, it is rare for PAN to be accompanied by severe ischemic colitis as initial presentation , 2.A 55-year-old man, with recurrent abdominal pain since 10 months treated with antispasmodic drugs, was admitted in Rabta's Department of Surgery (A), because of fever, abdominal pain, and bloody stool. Abdominal examination revealed mild rebound tenderness.Laboratory tests revealed severe inflammation. Upper gastrointestinal endoscopy was normal. Colonoscopy showed “ulcerative and bleeding mucosa in sigmoid colon.”CT-scan revealed “dilatation of sigmoid colon, wall thickening, and hyperattenuation. Colon was distended upstream.”At laparotomy, the colon appeared cyanotic. Gentle palpation of sigmoid colon caused violaceous discoloration of the bowel. Pulsations were left in the celiac, superior mesenteric, and inferior mesenteric arteries. The entire colon was resected and ileostomy was created.Histological examination of the pathologic specimens revealed “fibrinoid necrosis and destruction of the internal lamina in small and medium-size arteries which are rich in plasma cells, lymphocytes, and neutrophils” Figures , and 4.He was diagnosed with PAN and was admitted to our department.Physical examination revealed reduction in tactile sensitivity in the territory of the right peroneal nerve and both ulnar nerves; the electromyography showed multiple mononeuropathy.Urinalysis, coagulation profile, serum electrolytes and enzymes, serology of hepatitis B and C, antineutrophil cytoplasmic antibody (ANCA), and serum tests for rheumatic diseases were normal.Computed tomography did not reveal microaneurysms.Treatment included 3 intravenous pulses of methylprednisolone, 60 mg/day of prednisone, and 12 monthly intravenous pulses of cyclophosphamide. After discharge, a progressive withdrawal of prednisone was accomplished. One year after, PAN has not relapsed.We have presented a case of PAN revealed by ischemic colitis necessitating acute surgical intervention.Polyarteritis nodosa (PAN), first described in 1866 by Kussmaul and Maier .PAN involves the gastrointestinal (GI) tract in more than 50% of patients at some time during its course , 4.Clinically apparent ischemic disease of the small bowel is the frequent site of involvement. The colon is less commonly involved .The presentation of colonic PAN may mimic inflammatory bowel disease in young patients and atherosclerotic ischemic colitis in older ones . Thus, aP < 0.001) [When studying 342 patients with PAN, Pagnoux found that GI involvement was present in 132 cases (37.9%) and was significantly more frequent in HBV related PAN (< 0.001) .Although GI involvement is frequent in PAN, the isolated abdominal initial presentation is uncommon, and since 1975, only 9 cases have been published in the English language literature. They are summarized in In the presence of acute abdominal signs, early surgery is warranted. In their absence, medical treatment should suffice. In all cases, close observation by a surgical team must be undertaken.For many years, PAN treatment has involved administration of high-dose steroid with an additional cytotoxic agent, such as cyclophosphamide, to induce remission. In most patients, it is appropriate to treat aggressively. Once remission is achieved, maintenance therapy with daily or alternate-day low-dose prednisolone and oral azathioprine is frequently used for up to 18 months .PAN, unlike some other vasculitides such as Wegener granulomatosis, appears to be a condition in which permanent remission can be achieved. Relapses can occur, but despite these, a real possibility of cure can be anticipated. However, if treatment is delayed or inadequate, life-threatening complications can occur due to the vasculitic process.Although the gastrointestinal tract is frequently involved with PAN, it is extremely rare for PAN to be accompanied by ischemic colitis, particularly at the initial presentation.


Malignant Müllerian mixed tumors (MMMTs) of the uterine cervix are extremely rare, accounting for 0.005% of all cervical malignancies. To date, only approximately 50 well-documented cases have been reported. Although several epithelial components have been described in cervical MMMTs, small cell neuroendocrine carcinoma (SCC) has not appeared in the English literature. We present a 43-year-old woman, para 2 gravida 2, who had MMMT with SCC and rhabdomyosarcoma components in the uterine cervix. She was referred to our hospital because of a cervical mass with an abnormal Pap smear result. Cervical biopsy revealed SCC. After neoadjuvant chemotherapy with balloon-occluded arterial infusion, she underwent type II radical hysterectomy with pelvic lymphadenectomy. Histological analysis revealed that the cervical tumor comprised SCC and rhabdomyosarcoma components. Genotype analysis indicated human papillomavirus type 18. She underwent concurrent chemoradiation therapy. The patient had been free of the disease and showed no evidence of recurrence 38 months after operation. Malignant Müllerian mixed tumors  of the uterine cervix are extremely rare, accounting for 0.005% of all cervical malignancies  and are  Histologically, MMMTs comprise epithelial and stromal components, with many combinations. However, to our knowledge, no case of combined small cell neuroendocrine carcinoma (SCC) and rhabdomyosarcoma has been reported yet. We report a case of an MMMT with rare histological features that was successfully treated with neoadjuvant chemotherapy using balloon-occluded arterial infusion (BOAI), surgery, and concurrent chemoradiation therapy. A 43-year-old woman, para 2 gravida 2, was referred to the Department of Obstetrics and Gynecology at the Osaka Minami Medical Center because of a uterine cervical mass with an abnormal Pap smear result. On magnetic resonance imaging, the cervical mass measured 67 × 54 mm at presentation. Based on the result of the histological examination, using a punch biopsy specimen, SCC was diagnosed, and neoadjuvant chemotherapy with BOAI was performed twice with cisplatin (100 mg), mitomycin C (10 mg), and pirarubicin (50 mg). The tumor was downsized to 24 × 16 mm in 2 months. She subsequently underwent type II radical hysterectomy with pelvic lymphadenectomy. For adjuvant therapy, she underwent 5 courses of chemotherapy with 50 mg cisplatin and 40 Gy whole pelvic irradiation. Her follow-up examination results obtained 38 months after the operation were unremarkable. Grossly, the anterior lip of the uterine cervix was occupied by the 27 × 23 mm hard round mass. On cross-sectional examination, the tumor was whitish yellow and solid. Microscopically, the tumor was located in the stroma of the uterine cervix . The tumα-smooth muscle actin, S-100, MyoD1, myogenin, neuron-specific enolase (NSE), synaptophysin, chromogranin A, CD56, CD10, CD99, carcinoembryonic antigen, p53, p63, p16, and Ki-67. In component A, the tumor cells were positive for EMA, NSE, synaptophysin  . Immunohtophysin , chromogtophysin , p53, p6tophysin , CD56 (Ftophysin , and CD1tophysin . Human p MMMTs of the uterine cervix are extremely rare. To date, only approximately 50 well-documented cases have been reported –6, in wh The prognosis in cervical MMMTs depends on the clinical stage of the disease and is better than that in the uterine counterpart because of their early detection in many cases , 6. Howe MMMT and SCC have an aggressive nature. Fortunately, our case with pelvic lymph node metastasis was diagnosed at its early clinical stage and was well managed with neoadjuvant chemotherapy with BOAI, which several authors , 17 repo In summary, we present a case of cervical MMMT with SCC and rhabdomyosarcoma components. To our knowledge, this is the first paper of cervical MMMT with an extremely rare association. Despite its aggressive nature, our case was successfully managed with the combination of neoadjuvant chemotherapy using BOAI, surgery, and concurrent chemoradiation therapy. However, further follow-up study is necessary.


Askin tumors are rare malignant neoplasms that are localized in the thoracopulmonary region and mainly occur in children and adolescents. Further investigation with regard to the effective treatment of this disease is required, since the disease has a low incidence and limited knowledge is available on the biological activity and prognostic factors of this type of tumor. The present study describes the case of a 30-year-old male patient with a histologically and morphologically proven Askin tumor who was treated in Shandong Cancer Hospital and Institute . A chest computed tomography (CT) scan demonstrated a large mass filling the entire left lung, associated with mediastinum and right lung compression and accompanied by destruction of the 2nd rib. The patient accepted chemotherapy and radiotherapy instead of a radical mass resection since the mass was irresectable. A good clinical response was achieved to the chemotherapy and radiotherapy. The diagnosis and treatment of Askin tumors remains a challenge for clinicians and surgeons due to the absence of standard therapeutic guidelines for the treatment of this disease. According to the experience obtained from the cases encountered to date, treatment strategies should aim to reduce local recurrence and distant metastasis. Moreover, surgery, chemotherapy and radiotherapy or a combination of these methods appears to constitute an effective treatment strategy for Askin tumors. This group of malignancies includes classic Ewing sarcoma of the bone, extraskeletal Ewing sarcoma, peripheral primitive neuroectodermal tumors (PNETs) and Askin tumors. The tumors that occur in the thoracopulmonary region have been referred to as Askin tumors since the first report of a PNET of the chest wall by Askin et al in 1979.An Askin tumor is a soft tissue sarcoma belonging to the ESFTs that is localized in the thoracopulmonary region. This type of tumor mainly occurs in children and adolescents. Although rare, individual cases have been occasionally reported in older patients  and newb2. Compression of the mediastinum and right lung was evident. The large mass extended into the lung, chest wall and axilla, accompanied by destruction of the 2nd rib  for further examination. A physical examination indicated swelling over the left chest wall and supraclavicular region, with decreased breath sounds over the left lung and dull percussion notes in the left hemithorax. No lymph nodes were palpable. Horner syndrome was also observed, as characterized by the symptoms of vertigo and left-sided anhidrosis and hypothermia without ptosis of the face and body, as well as miosis and conjunctival congestion in the left eye. Contrast-enhanced computed tomography (CT) showed a large, heterogeneous density compatible with areas of non-enhancing necrosis; the maximum cross-sectional area of the mass was 15×22.5 cm 2nd rib . A needl 2nd rib . The diaThe patient underwent chemoradiotherapy instead of surgery due to the anatomical complexities of the involved structures. The patient was administered 4 cycles of combined chemotherapy (1 cycle every 3 weeks), including 2 mg vincristine, 30 mg epirubicin and 60 mg cisplatin; the 4th cycle was administered in combination with radiotherapy at a dose of 1.8 Gy per fraction. The patient refused further treatment when 20 fractions of radiotherapy were completed. The patient response to the aforementioned treatment was good and a clinical response was achieved. A physical examination indicated disappearance of the swelling in the chest wall and relief of the symptoms, although a CT scan indicated that the limited reduction in tumor size was not ideal. At the time of writing this manuscript, the patient was well with no evidence of metastasis.et al 81 and 86 and European Intergroup Cooperative Ewing’s Sarcoma Study (EICESS) 92, radiotherapy was used in ~87% of patients as a pro- or post-operative adjuvant therapy or as radical radiotherapy for unresectable tumors ,23. Baseet al concludeet al. The impet al. Althouget al,24,25. Eet al.et al, but also in prolonged survival rates. Demir et al. In the case of patients with a high risk of recurrence , the chemotherapeutic regimen used included vincristine, actinomycin D, ifosfamide (IFM) and ADM (VAIA)  . SubsequM (VAIA) . Grier eA) . Autologous bone marrow transplantation has been suggested to be an effective rescue treatment, but this remains controversial. It has been demonstrated that the use of high-dose chemotherapy followed by either autologous bone marrow transplantation or peripheral stem cell rescue is valuable in the treatment of Ewing sarcoma . This poet al reportedIn conclusion, Askin tumors/PNETs develop in soft tissues and the disease is detectable only when the tumor is large. Thus, Askin tumors are mostly diagnosed at an advanced stage, which is one of the most important reasons for a poor prognosis. Moreover, due to the rarity of this disease, relevant studies are mostly small-scale, single-institution clinical trials. The diagnosis and treatment of Askin tumors/PNETs remains a challenge for clinicians and surgeons due to the absence of standard guidelines. Notably, a wide resection combined with polychemotherapy has been shown to result in satisfactory local control. The results of local control and decreased distant relapse in patients administered neoadjuvant chemotherapy followed by a complete resection are superior compared with the results in patients who undergo primary surgery followed by chemotherapy and/or radiotherapy. Consequently, large-scale randomized trials are required for the development of effective treatment strategies for Askin tumors.


Gastric cancer is one of the most common malignancies in the world and is the second most common cause of cancer-related death in Korea. Colorectal metastases from gastric adenocarcinoma are known to be very rare. We report an unusual case of rectal metastasis of gastric adenocarcinoma.th postoperative day with no adverse events.We report a case of a 43-year-old female patient with gastric cancer who first presented with epigastric pain. The endoscopic and radiologic findings were suggestive of Borrmann type III advanced gastric cancer with linitis plastica. Radical total gastrectomy with D2 lymph node dissection was performed. The pathology report was AJCC TNM Stage II gastric adenocarcinoma (T3N0M0). On follow up at 34 months after surgery, the patient complained of difficulty in defecation. On colonoscopy, a hard, indurated extraluminal mass was detected 7 cm proximal to the anal verge. The biopsy demonstrated chronic nonspecific colitis. Abdominal CT, rectal MRI and PET-CT revealed rectal metastasis from gastric cancer. Laparoscopic ultralow anterior resection with diverting ileostomy was performed. The pathology report was metastatic adenocarcinoma, and this diagnosis was identical to the gastric pathology reported in the previous pathology report. The patient was discharged after the 11Rectal metastasis from gastric cancer is known to be very rare. However, metastatic gastric adenocarcinoma should be considered as a differential diagnosis for patients presenting with a colorectal mass and a past history of gastric cancer. Gastric cancer is one of the most common malignancies in the world and is the second most common cause of cancer-related death in Korea . The knoColonic metastases from gastric adenocarcinoma are known to be very rare and such a hematogenous recurrence pattern of advanced gastric cancer with lymph node-negative and absence of lymphovascular invasion in the pathology after curative R0 resection has rarely been described.We report an unusual case of rectal metastasis from gastric adenocarcinoma.A 43-year-old woman who first presented with epigastric pain was diagnosed with gastric cancer. She had no family history of gastric cancer. Her past medical history revealed that she had undergone Cesarean section once. Laboratory findings showed mild anemia, with a hemoglobin level of 10.9 g/dL (reference range 12.3-16.5 g/dL). The endoscopic and radiologic findings revealed that the gastric mass was Borrmann type III advanced gastric cancer with linitis plastica.th edition American Joint Committee on Cancer (AJCC) was stage II (T3N0M0)[12 with monitoring every 3 months, abdominal CT every 6 months, and gastroduodenoscopy annually for 3 years. She had shown no evidence of disease recurrence in the 33 months prior to this episode. At thirty-four months after surgery, during a routine check-up, the patient complained of difficulty with defecation and constipation since the last.She had undergone radical total gastrectomy with D2 lymph node dissection, and splenectomy in December 2007. The final pathology report after surgery demonstrated a diffuse, poorly differentiated adenocarcinoma. The surgical resection margins were clear. Thirty-five lymph nodes were harvested, and were found to be tumor-free, and spleen was not involved. Lymphovascular invasion was absent, although, perineural invasion was noted. The TNM staging according to the 6 (T3N0M0). She was1 month. On digital rectal examination and colonoscopy, hard induration and stenosis of the rectum, with an extraluminal mass 7 cm proximal to the anal verge, with edematous, erythematous, and nodular mucosa was detected  level was 3.3 ng/mL . The other serum tumor markers, including CA 19-9, were also within their normal ranges.Abdominal computed tomography (CT) and rectal MRI revealed a 5 cm in length concentric wall thickening with enhancement in the mid-rectum about 6 cm proximal to the anal verge, and it had a target-like appearance Figure . No evidHer past medical history and radiologic findings suggested that metastasis to the rectum from gastric malignancy was more likely than primary rectal malignancy.On subsequent F-18-fluoro-deoxy-glucose (FDG) positron emission tomography (PET) scan, focal hypermetabolic lesion with the standardized uptake value (SUV) of 7.6 was detected in the upper rectum Figure . No abnoFinally, it was postulated that the rectal tumor was a metastatic tumor from gastric malignancy and peritoneal seeding in the pelvic cavity. Laparoscopic low anterior resection with diverting ileostomy was performed.2 gas to a pressure of 12 mmHg. The moderate adhesions resulting from the previous gastrectomy were dissected and lysed with an ultrasonic dissector. After careful inspection of the liver and entire abdomen, a laparoscopic biopsy of the peritoneal mass was performed, and frozen sections were obtained for analysis. For operation of the rectum, a medial-to-lateral approach for laparoscopic colectomy was chosen and a high ligation of the inferior mesenteric artery and vein was performed, and the dissection was begun. Laparoscopic total mesorectal excision up to 2 cm distal to the rectal tumor was accomplished, and double stapling using endoscopic linear staplers and a circular stapler was performed for rectal transection and primary anastomosis. All procedures were in keeping with the oncologic principles.Briefly, the abdomen was entered using open (Hasson) method, and an 11 mm supraumbilical optic port was placed. A 12-mm port was placed in both the right and left lower quadrants and two 5-mm ports were placed about 10 cm proximal to each 12-mm port. The abdomen was insufflated with COThe OCTO port wound protector and retractor system  and cylindrical vinyl film were used to cover the minilaparotomy for protection against cancer cell dissemination during specimen extraction. A diverting loop ileostomy was performed laparoscopically after reestablishing a pneumoperitoneum Figure .Intraoperative fresh frozen sections of paraaortic lymph nodes and peritoneal mass were obtained for analysis, and the findings were negative for malignancy.2 in size. The configuration of the lesion was of an ulceroinfiltrative mass. The safety resection margins were 10 cm proximally, 2 cm distally. Both the resected surgical margins were free from tumor cells. The circumferential margin after excision of the rectal tumor was 1.1 mm. The type of differentiation was of a poorly differentiated metastatic gastric cancer, invading all layers of the rectal wall up to pericolic soft tissue with focal mucosal invasion. Thirty-five lymph nodes were harvested, and all of them were found to be free from tumor cell invasion.The fresh specimen was about 17 cm in length on gross examination, and the rectal tumor size was about 4 × 4.5 cmThe immunohistochemical staining for cytokeratin 7 (CK7), cytokeratin 20 (CK20), c-erb B2 and CDX2 were negative Figure . The patrd postoperative day, and the patient was discharged after the 11th postoperative day with no adverse events. After rectal resection, the patient was still being followed up for chemotherapy with the FOLFOX-4 regimen  starting from December 2010. Her clinical course is being observed and she is exhibiting stable disease while the 12 cycles of the FOLFOX-4 regimen are being continued.Oral feeding was started on the 3The incidence of gastric cancer throughout the world is still high, and the incidence rate is increasing, especially in many Asian countries including Korea . In clinIntestinal metastasis from gastric cancer is not common, although the most common cause of secondary neoplastic infiltration of the colon is gastric linitis plastica ,8. The mAlso, the patterns of metastases in the intestinal and diffuse types of gastric cancer are known to be different, and the diffuse type of gastric cancer demonstrated a wider dissemination than the intestinal type. The intestinal type involved the liver more frequently . PeritonThe endoscopic features of luminal metastases are variable, but the typical characteristic features are diffuse circumferential wall thickening and stiffness which mimick those of linitis plastica such that the term secondary linitis plastica of the rectum has been used . The oveCT appears to be the most feasible technique for detecting intestinal metastasis from gastric adenocarcinoma . IntestiThe laparoscopic surgery has been an alternative to conventional open surgery in the treatment of colorectal carcinoma without compromising the oncologic outcome of laparoscopic colectomy ,15. In tThe clinical utility of CDX2 was confirmed to be fairly specific for the identification of adenocarcinoma of the gastrointestinal tract, particularly colorectal adenocarcinoma, in the primary and metastatic setting . In 55% After rectal resection, we used the FOLFOX regimen for systemic therapy due to our institutional preference. Other regimens such as DCF , ECF , irinotecan plus cisplatin, with other modifications could also be used.In conclusion, we experienced a rare case of hematogenous rectal metastatic carcinoma from gastric adenocarcinoma which was treated successfully by laparoscopic surgery.Colorectal metastasis from gastric cancer may occur, although it is very rare. When the symptoms of change in bowel habits such as abrupt hematochezia, or constipation are noticed, the possibility of metastases to the colon and rectum should be suspected, and the importance of endoscopic and CT surveillance for metastatic sites in the colon and rectum should be emphasized at postoperative check up during the follow-up period.Written informed consent was obtained from the patients for publication of this report and any accompanying images. A copy of the written consent is available for review with the Editor-in-Chief of this journal.The authors declare that they have no competing interests.SWL wrote the main manuscript and HRK performed the operation, revised the manuscript for important intellectual content, and gave the final approval for the version to be submitted for publication. All authors read and approve the final manuscript.