High-Output Stoma Leading to the Diagnosis of Antiphospholipid Syndrome.

Diverting stoma (DS) is widely created in colorectal surgery. High-output stoma (HOS) is a major complication of DS, which can lead to dehydration and thrombosis. Additionally, antiphospholipid syndrome (APS) is a risk factor for thrombosis, and it rarely occurs in men. Herein, we describe a case of multiple thromboses caused by chronic dehydration after HOS. A 48-year-old man visited our hospital with fever and lower abdominal pain; he was diagnosed with sigmoid diverticulitis. He underwent laparoscopic high anterior resection for relapsing diverticulitis and diverting ileostomy during the same operation. On postoperative day 1, an output of 3,000 mL/day was observed from the ileostomy. The stoma output exceeded 2,000 mL/day, which was diagnosed as HOS, and chronic dehydration persisted despite supplementation and restriction of oral water intake. Three months postoperatively, a computed tomography scan before ileostomy closure showed multiple thrombi in the inferior vena cava, right common iliac vein, and pulmonary artery. After antithrombotic therapy, ileostomy closure was performed. As lupus anticoagulant was positive twice and APS was diagnosed, antithrombotic therapy was changed from warfarin to direct oral anticoagulants. Thrombosis did not recur 6 months postoperatively. This is the first report of a case wherein APS was present in the background of thrombosis caused by HOS or chronic dehydration. It is important to be cautious about APS when there is thrombosis after HOS to select appropriate therapeutic agents.

A case of duodenal duplication cyst mimicking a pancreatic pseudocyst with intracystic hemorrhage.

BACKGROUND: Duodenal duplication cysts in adults are rare, and a preoperative diagnosis remains difficult because clinical manifestations are nonspecific and variable. We describe a case of a duodenal duplication cyst mimicking a pancreatic pseudocyst with repeated intracystic hemorrhage. CASE PRESENTATION: A 47-year-old male who complained of upper abdominal pain and vomiting was referred to our hospital. He was a heavy drinker and had a past history of hospitalization for alcoholic chronic pancreatitis. Plain abdominal computed tomography (CT) showed a cystic lesion of 7 cm in size in the lumen near the second part of the duodenum. The cystic lesion showed high density inside. Gastrointestinal endoscopy revealed that the lumen of the duodenum was deformed by a submucosal tumor-like mass and the endoscope could not pass through it, but active bleeding was not seen in the lumen of the duodenum. On the fourth day of hospitalization, anemia progressed and contrast-enhanced CT demonstrated a high-density spot on the wall of the cystic lesion. A pancreatic pseudocyst complicated with intracystic hemorrhage was preliminarily considered. Angiography was immediately performed, and a pseudoaneurysm was identified in the branch of the anterior superior pancreaticoduodenal artery (ASPDA). Transcatheter arterial embolization (TAE) was performed. Anemia did not progress after that, and follow-up CT showed reduction in the size of the cystic lesion. Afterward, the same symptoms recurred twice and surgical treatment was performed for the pancreatic pseudocyst with repeated intracystic hemorrhage. Macroscopically, a cystic mass of 5 cm in size was adjacent to the second part of the duodenum on the pancreas side. A pinhole-sized communication was identified between the cyst and the duodenum lumen. Microscopically, the cyst wall was composed of gastric mucosa and shared a common proper muscle layer with the duodenum. Chronic ulcers and erosions were seen in the cyst. Based on these findings, a diagnosis of duodenal duplication cyst was made. CONCLUSIONS: Duodenal duplication cysts in adults have seldom been reported and should be considered as a differential diagnosis for a patient with a cystic lesion adjoining the duodenum.

A Rare Case of Pylephlebitis as a Complication of Cholecystocolonic Fistula.

Pylephlebitis is defined as a septic thrombophlebitis of the portal vein and its tributaries that is associated with multiple suppurative abdominal infections. We report a case of pylephlebitis associated with a cholecystocolonic fistula (CCF). A 41-year-old man presented with upper abdominal pain and anorexia for 1 month. Abdominal contrast-enhanced computed tomography (CT) revealed thrombosis in the left and anterior branch of the portal vein and thickening of the walls of the portal vein and periside portals. The gallbladder was collapsed and pneumobilia was seen in the biliary tract. Blood culture was positive for Streptococcus anginosus. A diagnosis of thrombophlebitis of the portal vein associated with CCF was made, and the patient was immediately managed with an intravenous broad-spectrum antibiotic and anticoagulation. After the portal vein thrombosis (PVT) propagation and inflammation had subsided, cholecystectomy and partial resection of the transverse colon were performed. Pylephlebitis is rare but is a life-threatening complication of intra-abdominal infection. A high index of suspicion is required, and a CT scan should be performed immediately for an early diagnosis and appropriate treatment.

[A Case of Resected Esophageal Endocrine Cell Carcinoma That Responded to Combination Therapy Comprising Irinotecan and Cisplatin].

We report a case of resected esophagealcancer that responded wellto first-line combination therapy comprising irinotecan and cisplatin. The patient was a 71-year-old woman being treated for liver cirrhosis. She was admitted to our hospital in April 2015 because of dysphasia. Endoscopic examination revealed a tumor in the mid-thoracic esophagus, which was diagnosed as an endocrine cell carcinoma following pathological examination. Contrast-enhanced computed tomography and positron emission tomography did not show lymph node or distant metastases. She was treated with irinotecan and cisplatin combination therapy. After 6 courses of treatment, the tumor size had remarkably reduced. Subsequently, we performed subtotal esophagectomy and gastric tube reconstruction through the retroposterior mediastinalroute and the histologicaleffect was reported as a partial response. No viable tumor cells were observed in the extracted lymph nodes. However, bone metastasis and lymph node swelling occurred after 4 months. She received other therapeutic regimens, such as etoposide and carboplatin combination therapy. However, the tumor gradually progressed, and she died 18 months after the first treatment. Irinotecan and cisplatin combination therapy is a possible option for the management of esophageal endocrine cell carcinoma as a first-line treatment.

Acute diffuse peritonitis due to spontaneous rupture of a primary gastrointestinal stromal tumor of the jejunum: A case report.

INTRODUCTION: Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal tumors of the gastrointestinal tract. Overt peritonitis caused by GIST rupture is very uncommon. Three types of GIST rupture have been described: closed perforation due to abscess (abscess type), hemoperitoneum leading to rupture of the hematoma capsule in the tumor (hemoperitoneum type), and perforation of the digestive tract via a fistula leading to central necrosis of the tumor (bowel perforation type). This report describes a patient with spontaneous tumor rupture and diffuse peritonitis, a variant of the bowel perforation type of GIST rupture. PRESENTATION OF CASE: A 74-year-old man presented with symptoms of vomiting and abdominal pain. Computed tomography (CT) scan revealed an approximately 10×7-cm mass in the pelvis with free air and fluid collection. Emergency laparotomy revealed a tumor in the jejunum, which was ruptured with a hole measuring 5mm in diameter. The tumor and part of the jejunum were resected. Immunohistochemically, the mass was diagnosed as a GIST originating from the gastrointestinal tract. Despite chemotherapy with imatinib mesylate, the patient died 22 months after surgery. CONCLUSIONS: This report describes a patient with acute diffuse peritonitis due to spontaneous rupture of a primary GIST of the jejunum.

A colovesical fistula with a persistent descending mesocolon due to partial situs inversus: A case report.

INTRODUCTION: Situs inversus viscerum, a congenital condition in which the visceral organs are a mirror image of their normal physiological positions, could be total or partial. Persistent descending mesocolon (PDM) is a congenital anomaly that is asymptomatic because of its short length. PDM causing intestinal obstruction is a known clinical complication. PRESENTATION OF CASE: A 74-year-old woman presented with pneumaturia and enteruria for two months, and recurrent cystitis for a month. An enhanced computed tomography (CT) showed air in the bladder along with sigmoid colonic diverticula adherent to it, suspecting a fistula. The CT also showed partial situs inversus with the common hepatic artery, and left colic artery arising abnormally from the superior mesenteric artery (SMA). Minimally invasive endoscopic closure using the over-the-scope clipping system was difficult because of thickening and scar tissue due to chronic inflammation from diverticulitis. Thus, a sigmoidectomy was performed to close the fistula. Intraoperatively, we noted an abnormally fixed descending mesocolon. An emergency reoperation was performed on the sixth postoperative day owing to an anastomotic leak. Suture failure was attributed to these congenital abnormalities due to insufficient blood flow from an absent marginal vessel and a high endocolonic pressure by adhesions. Sigmoid colon re-resection and maturation of an ileostomy was performed. The patient had no specific postoperative complications, and the ileostomy was closed after three months. CONCLUSION: We report an extremely rare case of colovesical fistula due to a PDM in a patient having partial situs inversus with abnormal branches originating from the SMA.

A case of pancreatic schwannoma showing increased FDG uptake on PET/CT.

INTRODUCTION: Schwannomas are neoplasms originating from Schwann cells in nerve sheath, but pancreatic schwannomas are rare. PRESENTATION OF CASE: A 59-year-old female incidentally pointed out a 2-cm-sized solid mass in the pancreas head by computed tomography (CT) which was performed for other reasons. Magnetic resonance imaging (MRI) showed a mass with hypointensity on T1-weighted images and a heterogenous hyperintensity on T2-weighted images. Magnetic resonance cholangiopancreatography (MRCP) showed no abnormal findings in the main pancreatic duct. The mass of the pancreas head showed a significantly increased uptake on fluorin-18 fluorodeoxyglucose positron emission tomography CT (FDG-PET/CT). Based on the radiological findings, a malignant potential tumor was preliminarily considered. Subtotal stomach-preserving pancreatoduodenectomy (SSSPD) was performed to make a definite diagnosis. The resected specimen showed a well-encapsulated circumscribed mass, measuring 2.0cm, in the pancreas head. Histopathological examination revealed proliferation of spindle cells showing interlacing and palisading patterns in the tumor. The proliferating cells showed no atypia and few mitoses. Immunohistochemically, the spindle cells were positive for S-100 protein. The Ki67 labeling index was approximately 2%. Based on these findings, a diagnosis of benign schwannoma of the pancreas was made. CONCLUSION: Although pancreatic schwannoma is a rare neoplasm, it is important to take into account this tumor in the differential diagnosis of pancreatic tumors. In addition, we should be aware that FDG-PET/CT shows abnormal accumulation in a benign pancreatic schwannoma.

Laparoscopic operation after pre-operative reduction of an intussusception-induced inflammatory fibroid polyp.

Intussusception occurs mainly in children but rarely in adults. About 80-90% of adult cases of intussusception are due to benign or malignant neoplasms as a lead point. One of the causes is an inflammatory fibroid polyp (IFP), a rare, benign entity that occurs more frequently in the stomach than the ileum. We describe an uncommon case of a patient who presented with intussusception-induced IFP of the ileum. A 35-year-old woman presented with a 2-day history of abdominal pain. A computed tomography scan demonstrated ileocolic intussusception. During colonoscopy, reduction by pressured air easily released intussusception, and we suspected a submucosal tumour of the ileum. Successful pre-operative colonoscopic reduction was helpful for performing laparoscopic ileocecal resection 2 days later. Results of the histopathological examination confirmed a large IFP. The patient's post-operative course was uneventful. Pre-operative reduction by colonoscopy is effective for the diagnosis and treatment of intussusception-induced IFP.

[A Case of Resected Advanced Esophageal Cancer That Responded to Combination Therapy Comprising Docetaxel, Cisplatin, and 5-Fluorouracil].

We report a case of resected advanced esophagealcancer that responded wellto first-line combination therapy with docetaxel, cisplatin, and 5-fluorouracil(DCF therapy). A 72-year-old man was admitted to our hospital in January 2013 because of dysphagia. On the basis of the computed tomography(CT)and gastroendoscopy findings, he was diagnosed with advanced esophagealcancer with lymph node metastasis. The patient was treated with DCF therapy. After 2 courses of treatment, the primary tumor and lymph node metastasis were reduced on CT. After 3 courses of treatment, we performed subtotalesophagectomy and gastric tube reconstruction through the retroposterior mediastinalroute. No residualcancer cells were found in the esophagus or lymph nodes. The patient subsequently received oral administration of tegafur-uracilal one for 24 months. The post-operative course was uneventful, and there was no detectable lymph node metastasis 42 months after the originaldiagnosis. Therefore, DCF therapy is a possible option for the management of advanced esophagealcancer.

Sphincter-saving resection by cluneal arched skin incision for a gastrointestinal stromal tumor (GIST) of the lower rectum: a case report.

BACKGROUND: Planning the surgical strategy for a gastrointestinal stromal tumor (GIST) at the posterior wall of the lower rectum is difficult, as the procedures for the lower rectum are hampered by poor visualization and may cause anal dysfunction or discomfort. We report a novel procedure to resect a submucosal tumor of the rectum. CASE PRESENTATION: A 75-year-old woman presented with metrorrhagia. Endovaginal ultrasonography showed a low echoic tumor. Computed tomography showed an enhanced tumor, measuring 5.3 × 4.2 cm, behind the rectum. Magnetic resonance imaging revealed a submucosal tumor of the rectum, measuring 5.3 cm at its greatest dimension. Colonoscopy showed that the distal tumor margin was 1 cm above the dentate line. Core needle biopsy of the tumor revealed the rectal GIST. After receiving neoadjuvant imatinib treatment, the tumor size decreased to 3.5 cm. During the operation, we approached the rectum and resected the posterior rectal wall, including the 3.5 × 3.5 cm tumor with a safety margin, making an arched incision at the buttocks to form a skin flap with the patient in a jackknife position. The histopathological diagnosis was GIST of the rectum. Her anorectal sphincter function was well preserved. No recurrence was seen during the 2-year follow-up. CONCLUSIONS: This novel approach improves the operative field visibility in resecting a tumor with a safety margin and preserves a patient's anorectal sphincter function.

A solid pseudopapillary neoplasm of the pancreas in a man presenting with acute pancreatitis: A case report.

INTRODUCTION: A solid pseudopapillary neoplasm (SPN) of the pancreas is rare neoplasm that occurs predominantly in young women. The clinical presentation of SPNs is nonspecific, but acute pancreatitis is rare in the reported literature. PRESENTATION OF CASE: A 36-year-old man was referred to our hospital because of upper abdominal pain and elevation of serum amylase. A computed tomography (CT) scan showed swelling of the pancreas body and a poorly enhanced and indistinct mass in the pancreas body. He was diagnosed with acute pancreatitis. The symptom was improved with conservative treatment, but acute pancreatitis recurred twice during a period of 2 months. Magnetic resonance cholangiopancreatography (MRCP) and endoscopic retrograde cholangiopancreatography (ERCP) showed stenosis of the MPD adjacent to the mass. Distal pancreatectomy was performed because the mass in the pancreas body seemed to cause repeated acute pancreatitis and malignant pancreatic cancer could not be excluded. Immunohistochemically, a diagnosis of SPN of the pancreas was made from the resected specimen. DISCUSSION: To the best our knowledge, only 6 cases have been reported in the literature concerning the SPN presenting with acute pancreatitis. CONCLUSION: We report a man with a small SPN of the pancreas presenting with acute pancreatitis and mimicking pancreatic cancer. We should be aware that this rare pancreatic tumor can become a cause of acute pancreatitis.

A mixed adenoneuroendocrine carcinoma of the pancreas: a case report.

A tumor consisting of an adenocarcinoma component and a neuroendocrine carcinoma component, with each component accounting for at least 30% of the tumor, is defined as a mixed adenoneuroendocrine carcinoma (MANEC). We report a case of MANEC of the pancreas. A 63-year-old man presented with hyperglycemia and was referred to our hospital for further examination. Abdominal contrast-enhanced computed tomography (CT) revealed a mass of 2 cm in size in the pancreas head with portal vein narrowing. Fluorin-18 fluorodeoxyglucose-positron emission tomography (FDG-PET) CT revealed increased accumulation in the mass of the pancreas head. Endoscopic retrograde cholangiopancreatography (ERCP) showed severe narrowing of the main pancreatic duct. Cytological analysis by endoscopic ultrasonography-guided fine-needle aspiration (EUS-FNA) suggested a neuroendocrine tumor. Under the diagnosis of neuroendocrine tumor, pancreaticoduodenectomy with portal vein resection and regional lymph node dissection was performed with curative intent. Histological examination revealed that the tumor consisted of two cell populations. One was well- to moderately differentiated tubular adenocarcinoma. This cell component accounted for 45% of the whole tumor. The second component was non-adenocarcinoma cells arranged in a nest, and the cells had round nuclei, abundant cytoplasm, and coarse chromatin. The Ki67 labeling index was 40%. Immunohistochemically, the adenocarcinoma cells were positive for CEA but negative for chromogranin A (CgA) and synaptophysin (Syn), while the non-adenocarcinoma cells were positive for the expression of CgA and Syn but negative for CEA. Based on the findings, a diagnosis of MANEC of the pancreas was made. Postoperatively, lymph node metastasis and peritoneal dissemination developed rapidly and he died the 6 months after the operation. Due to the small number of reported cases of MANEC of the pancreas, its clinical behavior remains unclear and a standardized management protocol has not been established. Further investigation of more cases of this rare entity is necessary.

[A Case of HER2-Positive Advanced Gastric Cancer with Multiple Liver Metastases Effectively Treated with Trastuzumab, Capecitabine, and Cisplatin Combination Therapy].

We reported a case of human epidermal growth factor receptor 2(HER2)-positive advanced gastric cancer with multiple liver metastases that responded well to a combination of trastuzumab, capecitabine, and cisplatin(T-XP therapy)as first-line chemotherapy. A 73-year-old man was admitted to our hospital in December 2012 for liver dysfunction. Based on computed tomography(CT)and gastroendoscopy findings, he was diagnosed with advanced gastric cancer with multiple liver metastases. Because HER2 protein overexpression was observed in the primary tumor, he was treated with T-XP therapy. After 5 courses of treatment, the sizes of the primary tumor and multiple liver metastases were reduced on CT scans. In March 2013, a Billroth I distal gastrectomy with D2 lymph node dissection was performed. Liver metastasis was not detected. No residual cancer cells were found in the stomach or lymph nodes. The patient subsequently received oral administration of S-1 alone for 2 weeks followed by a 2-week rest period as 1 course. This was repeated for 19 courses. The postoperative course was uneventful, and there was no detectable liver metastasis 36 months after the original diagnosis. Therefore, T-XP therapy is an option for the management of HER2-positive advanced gastric cancer with liver metastasis.

Successful treatment of primary advanced gastric plasmacytoma using a combination of surgical resection and chemotherapy with bortezomib: A case report.

INTRODUCTION: Extramedullary plasmacytoma (EMP) is a plasma cell neoplasm that presents as a solitary tumor. EMP in the gastrointestinal organs are extremely uncommon. PRESENTATION OF CASE: A 36-year-old man was admitted to our hospital with advanced anemia. He had no specific medical history. Gastroendoscopic findings showed an 8.0-cm submucosal tumor with ulcer on the greater curvature of the gastric body. Fine-needle aspiration was performed, and the pathologic diagnosis of the submucosal tumor was a plasmacytoma. Therefore, the patient was diagnosed with gastric plasmacytoma. A total gastrectomy was performed with lymphadenectomy. The result of intraoperative peritoneal lavage cytology was positive. Histological examination revealed serosa-exposed plasmacytoma of the stomach with lymph nodes metastasis. Additionaly the patient received a three-drug chemotherapy regimen (bortezomib, cyclophosphamide, and dexamethasone [VCD]) from 3 weeks after the operation. After 4 cycles of chemotherapy, the patient received autologous peripheral blood stem-cell transplantation (auto-PBSCT). Eighteen months after diagnosis, the patient is in complete remission with no evidence of local relapse or evolution to multiple myeloma. CONCLUSIONS: This is the first reported case of advanced gastric plasmacytoma using adjuvant chemotherapy involving bortezomib and auto-PBSCT after the resection, and the patient has maintained a good course over a year. This protocol could be a new way to treat these tumors.

Heterotopic gastric mucosa in the hilar bile duct mimicking hilar cholangiocarcinoma: report of a case.

We herein report a case of heterotopic gastric mucosa in the hilar bile duct. An asymptomatic 58-year-old male was noted to have mild liver dysfunction in March 2009 during the follow-up for angina pectoris. Abdominal-enhanced CT revealed wall thickening from the upper common hepatic bile duct to the left hepatic bile duct. Endoscopic retrograde cholangiopancreatography (ERCP) showed stenosis at the junction of the left hepatic bile duct. Although the patient's serum tumor markers were all within the normal ranges, the possibility of malignant disease of the biliary tree could not be ruled out. Left hepatectomy with the caudate lobe and resection of the extrahepatic bile duct were performed. Histopathologically, the resected specimen showed a polypoid lesion measuring 2 × 2 cm in size that projected into the lumen of the left hepatic bile duct. Microscopic examination revealed this polypoid lesion to be composed of mucous glands resembling gastric fundic glands, with parietal and chief cells. We also review eight other reports of heterotopic gastric mucosa in the biliary tree previously published in the English literature.

[Significant regression of a cavernous hepatic hemangioma to a sclerosed hemangioma over 12 years: a case study].

A sixties woman was found to have diagnosed by abdominal ultrasonography with a tumor in the left lobe of the liver and was referred to our institution in 1998. Abdominal magnetic resonance imaging (MRI) showed a typical, 70×45 mm cavernous hemangioma, which was followed up by annual MRI. In 2006, 8 years after the initial diagnosis, the MRI showed that the tumor had reduced to 30×15 mm. Although atypical of hemangioma, review of the annual observations indicated a diagnosis of regressive hemangioma, which also accorded with clinical observations. In 2009, a liver biopsy was performed by laparotomy during gastrectomy for gastric cancer. Pathological examination of the biopsy revealed sclerosed hemangioma tissue, confirming the diagnosis of regression of a cavernous hemangioma to a sclerosed hemangioma over 12 years.

Angiomyolipoma of the colon: report of a case.

Angiomyolipomas are benign mesenchymal tumors mostly arising from the kidney. Angiomyolipoma of the colon is extremely rare. Here we report the findings of a 51-year-old man who presented with a submucosal tumor covered with normal mucosa and hemorrhage in the descending colon. He underwent a partial resection of the descending colon. A histopathological examination showed that the tumor of 5.7 cm in diameter included smooth muscle (spindle cell type), mature adipose tissue, and vessels, and therefore a diagnosis of angiomyolipoma was made. A submucosal type of angiomyolipoma of the colon is extremely rare. If a renal angiomyolipoma is larger than 4 cm, there is a risk of rupture. When colonoscopy shows a submucosal tumor of the colon with hemorrhage, angiomyolipoma should be considered. If an angiomyolipoma of the colon is large, surgical resection should be considered as a treatment option due to the risk of hemorrhage.