Isolated IgG4-related cholecystitis with localized gallbladder wall thickening mimicking gallbladder cancer: a case report and literature review.

BACKGROUND: IgG4-related cholecystitis, which is a manifestation of IgG4-related disease in the gallbladder, is associated with autoimmune pancreatitis or IgG4-related sclerosing cholangitis in most cases; isolated gallbladder lesions without systemic manifestations are very rare. Gallbladder wall thickening is often diffuse, but sometimes localized, in which case, differentiation from gallbladder cancer becomes difficult. The characteristic features of IgG4-related cholecystitis on imaging that would enable differentiation from gallbladder cancer remain poorly described. CASE PRESENTATION: We present a rare case of isolated IgG4-related cholecystitis with localized gallbladder wall thickening that was clinically difficult to distinguish from malignancy before resection. An 82-year-old man was referred to our hospital because of gallbladder wall thickening on abdominal ultrasonography without any symptoms. Dynamic computed tomography of the abdomen showed localized wall thickening from the body to the fundus of the gallbladder that was enhanced from an early stage with a prolonged contrast effect. There were no other findings, such as pancreatic enlargement and bile duct dilatation. Magnetic resonance cholangiopancreatography revealed neither dilatation nor stenosis of the bile duct and pancreatic duct. Endoscopic ultrasonography (EUS) showed a smooth layered thickening of the gallbladder wall with a maximum thickness of 6 mm and a well-preserved outermost hyperechoic layer in the same area. Laparoscopic cholecystectomy was performed because malignancy could not be completely ruled out. Pathological examination of a resected specimen revealed IgG4-positive plasma cell infiltration, fibrosis, and phlebitis. Although the serum IgG4 level measured after resection was normal, the condition was ultimately diagnosed as probable IgG4-related cholecystitis according to the 2020 revised comprehensive diagnostic criteria for IgG4-related disease. The EUS images reflected the pathological findings, in which lymphocytic infiltration was distributed in a laminar fashion in the gallbladder wall. CONCLUSIONS: Although rare, isolated IgG4-related cholecystitis with localized wall thickening mimicking gallbladder cancer remains a clinical problem. A smooth laminar thickening of the gallbladder wall on EUS imaging could be one of the most informative characteristics for differentiating IgG4-related cholecystitis from gallbladder cancer.

Successful outcomes after laparoscopic spleen-preserving pancreatic resection for a desmoid tumor: A case report.

INTRODUCTION: Desmoid tumors are slowly growing neoplasms that arise from fibroblasts. These tumors are locally aggressive and have a high rate of recurrence after surgery. Pancreatic desmoid tumors are extremely rare. The indications and outcomes of laparoscopic surgery for pancreatic desmoid tumors have not been fully elucidated. This report therefore aimed to describe a rare case of a pancreatic desmoid tumor in a patient who was successfully treated with laparoscopic spleen-preserving pancreatic resection. PRESENTATION OF CASE: We report a case of a 60-year-old man who presented with back pain. Contrast-enhanced computed tomography scan revealed a circumscribed tumor in the pancreatic tail measuring 3 cm. The patient underwent laparoscopic spleen-preserving distal pancreatectomy. Pathological analysis revealed a desmoid tumor infiltrating the pancreatic parenchyma. There was no evidence of recurrence at 36 months of follow-up. DISCUSSION AND CONCLUSION: Isolated sporadic pancreatic desmoid tumors are extremely rare. Laparoscopic radical resection could be a safe and effective treatment for this benign but locally aggressive tumor. To the best of our knowledge, this is the first study to report a patient with a pancreatic desmoid tumor who was successfully treated with laparoscopic surgery.

A prospective feasibility study of one-year administration of adjuvant S-1 therapy for resected biliary tract cancer in a multi-institutional trial (Tokyo Study Group for Biliary Cancer: TOSBIC01).

BACKGROUND: Although surgery is the definitive curative treatment for biliary tract cancer (BTC), outcomes after surgery alone have not been satisfactory. Adjuvant therapy with S-1 may improve survival in patients with BTC. This study examined the safety and efficacy of 1 year adjuvant S-1 therapy for BTC in a multi-institutional trial. METHODS: The inclusion criteria were as follows: histologically proven BTC, Eastern Cooperative Oncology Group (ECOG) performance status 0 or 1, R0 or R1 surgery performed, cancer classified as Stage IB to III. Within 10 weeks post-surgery, a 42-day cycle of treatment with S-1 (80 mg/m2/day orally twice daily on days 1-28 of each cycle) was initiated and continued up to 1 year post surgery. The primary endpoint was adjuvant therapy completion rate. The secondary endpoints were toxicities, disease-free survival (DFS), and overall survival (OS). RESULTS: Forty-six patients met the inclusion criteria of whom 19 had extrahepatic cholangiocarcinoma, 10 had gallbladder carcinoma, 9 had ampullary carcinoma, and 8 had intrahepatic cholangiocarcinoma. Overall, 25 patients completed adjuvant chemotherapy, with a 54.3% completion rate while the completion rate without recurrence during the 1 year administration was 62.5%. Seven patients (15%) experienced adverse events (grade 3/4). The median number of courses administered was 7.5. Thirteen patients needed dose reduction or temporary therapy withdrawal. OS and DFS rates at 1/2 years were 91.2/80.0% and 84.3/77.2%, respectively. Among patients who were administered more than 3 courses of S-1, only one patient discontinued because of adverse events. CONCLUSIONS: One-year administration of adjuvant S-1 therapy for resected BTC was feasible and may be a promising treatment for those with resected BTC. Now, a randomized trial to determine the optimal duration of S-1 is ongoing. TRIAL REGISTRATION: UMIN-CTR, UMIN000009029. Registered 5 October 2012-Retrospectively registered, https://upload.umin.ac.jp/cgi-open-bin/ctr_e/ctr_view.cgi?recptno=R000009347.

Comparison of olanexidine versus povidone-iodine for preventing surgical site infection in gastrointestinal surgery: study protocol for a multicentre, single-blind, randomised controlled clinical trial.

INTRODUCTION: The prevalence of surgical site infection (SSI) remains higher in gastrointestinal surgery than in other surgeries. Although several guidelines have indicated the efficacy of chlorhexidine and povidone-iodine in reducing the SSI rate, the optimal recommendation has still not been established. Therefore, it is necessary to determine the more effective antiseptic for surgical site preparation. Olanexidine (1.5% olanedine, Otsuka Pharmaceutical Factory, Tokushima, Japan), which is a new antiseptic in Japan, has antimicrobial activity against a wide range of bacteria, including Gram-positive and Gram-negative bacteria. Our study will contribute to determining a new antiseptic for use in gastrointestinal and other surgeries. METHODS AND ANALYSIS: We propose a multicentre, randomised controlled clinical trial for comparing two treatments, that is, 1.5% olanexidine or 10% povidone-iodine, for surgical skin preparation to prevent SSI in clean-contaminated gastrointestinal surgeries with surgical wounds. Patients aged ≥20 years at the time of consent will be included. The primary outcome measure is the 30-day postoperative SSI rate. For the primary analysis, which is aimed at comparing the treatment effects, the adjusted risk ratio and its 95% CI will be estimated using the Mantel-Haenszel method. ETHICS AND DISSEMINATION: The protocol was first approved by the Institutional Review Board of Keio University School of Medicine, followed by the institutional review board of each participating site. Participant recruitment began in June 2018. The final results will be published in international peer-reviewed medical journals. TRIAL REGISTRATION NUMBER: UMIN 000031560; Pre-results.

Mass-forming type 1 autoimmune pancreatitis mimicking pancreatic cancer.

We reported three cases of mass-forming type 1 autoimmune pancreatitis (AIP) that were preoperatively suspected to be pancreatic cancer, and reviewed their clinicopathological features. Radiological findings in the patients revealed hypoattenuating masses in the early phase or a stricture of the main pancreatic duct with upstream dilatation, which was consistent with the diagnosis of pancreatic cancer. Histopathologically, the lesions were well demarcated and met all diagnostic criteria for immunoglobulin G4 (IgG4)-related AIP, including the presence of periductal lymphoplasmacytic infiltration, obliterative phlebitis, storiform fibrosis and abundant IgG4-positive plasma cells. However, the adjacent uninvolved pancreatic duct and lobular structures were well preserved. And in all patients, none or some of the aforementioned characteristics were observed. We suggest that some cases of focal AIP may progress to more severe grades and exhibit mass formation, although remaining localized. These focal cases of AIP are difficult to distinguish from pancreatic cancer. To our knowledge, this report is the first to present a histopathological comparison of mass-forming AIP with the adjacent uninvolved pancreatic tissues.

Successful resolution of a hemorrhagic pancreatic pseudocyst ruptured into the stomach complicating obstructive pancreatitis due to pancreatic cancer: a case report.

BACKGROUND: Hematemesis is uncommon as an initial presenting symptom in pancreatic cancer. We present herein a case of a pseudoaneurysm that ruptured and fistulized into the stomach. The pseudoaneurysm was secondary to a pancreatic pseudocyst complicating obstructive pancreatitis due to pancreatic cancer. The patient was successfully treated using trans-arterial embolization followed by curative surgery. CASE PRESENTATION: A 61-year-old man presented to the emergency room with hematemesis. Laboratory examinations revealed a low level of hemoglobin (5.0 g/dl). The patient had presented to another hospital due to hematemesis 1 month before presenting to our hospital. A low-density mass in the pancreatic body with dilatation of the distal main pancreatic duct and a pseudocyst in the pancreatic tail had been observed by radiology at the previous hospital. Further investigation had been planned. Abdominal computed tomography on admission to our hospital demonstrated a pseudoaneurysm in close contact with the wall of the pseudocyst of the pancreatic tail, compressing the stomach. The pseudoaneurysm had not been detected by abdominal computed tomography at the previous hospital. Emergency selective angiography revealed that the pseudoaneurysm arose from the left gastroepiploic artery branching from the splenic artery. Trans-arterial embolization of the left gastroepiploic artery through the splenic artery was successfully performed. Elective distal pancreatectomy and splenectomy with regional lymph node dissection combined with partial resection of the stomach was performed 3 weeks after coil embolization. Pathological examination revealed a moderately differentiated tubular adenocarcinoma in the pancreatic body with regional lymph node metastasis and revealed the pseudoaneurysm rupturing into the pancreatic pseudocyst. The patient has experienced no tumor recurrence or metastasis during 1 year of follow-up. CONCLUSIONS: Spontaneous rupture of a pseudoaneurysm is a rare and potentially lethal complication of a pancreatic pseudocyst. Most affected patients have a history of alcoholism and suffer from acute or chronic pancreatitis. To our knowledge, this is the first reported case of a hemorrhagic pancreatic pseudocyst complicating obstructive pancreatitis due to pancreatic cancer.

Adenosquamous carcinoma of the ampulla of Vater: a case report and literature review.

BACKGROUND: Adenosquamous carcinoma of the ampulla of Vater is extremely rare, and its clinicopathological features are limited and described in few previous case reports. Here, we report curative resection of adenosquamous carcinoma of the ampulla of Vater at an early stage. CASE PRESENTATION: An 81-year-old woman was referred to our hospital for investigation of the frequent elevation of hepatic and biliary enzymes and dilatation of the intrahepatic bile ducts. Preoperative examinations revealed an exposed reddish tumor in the ampulla of Vater, which was diagnosed on biopsy to be adenocarcinoma with squamous cell carcinoma component. Pylorus-preserving pancreaticoduodenectomy with regional lymph node dissection was performed. Pathological examinations revealed the presence of two malignant components in the lesion, including poorly differentiated tubular adenocarcinoma and squamous cell carcinoma, without invasion beyond the sphincter of Oddi or into the duodenal submucosa. These squamous cell carcinoma and adenocarcinoma components in the tumor comprised approximately 30 and 70% of the lesion, respectively. No metastasis into regional lymph nodes was observed, and the patient experienced no tumor recurrence or metastasis until 20 months after surgery. CONCLUSION: We identified only six reported cases of adenosquamous carcinoma of the ampulla of Vater in the English literature, and all of these patients died of recurrence within 14 months after surgery. To the best of our knowledge, this is the first report of adenosquamous carcinoma of the ampulla of Vater that was curatively resected at an early stage. Although more number of studies on clinicopathological findings are required to determine the appropriate surgical indication, we suggest that surgery remains the mainstay therapy for adenosquamous carcinoma of the ampulla of Vater detected at an early stage.

Dieulafoy lesion in the ileum of a child: a case report.

A 14-year-old girl had massive bleeding from a Dieulafoy lesion of the ileum. A preoperative dynamic computed tomography scan detected the point of bleeding. Selective arteriography with embolization using microcoils could not stop the bleeding, but the microcoils were useful as markers of the location of the bleeding point. The position of the microcoils was confirmed by intraoperative fluoroscopy. A partial resection of the ileum that included the lesion was performed. The pathologic finding was Dieulafoy lesion of the ileum. Dieulafoy lesion is a rare condition that usually presents in the stomach. Dieulafoy lesion of the ileum is extraordinarily rare, and to our knowledge, this is only the second case report of an ileal lesion in a child.

Successful endoscopic submucosal dissection for mucosal cancer of the duodenum.

We report a case of mucosal duodenal cancer in a 62-year-old woman, which was successfully removed en bloc by endoscopic submucosal dissection (ESD). The patient underwent an upper gastrointestinal endoscopy at our hospital, which revealed an elevated flat mucosal lesion (type IIa) measuring 10 mm in diameter in the second portion of the duodenum. Histopathological examination of a biopsy specimen revealed features suggestive of a tubulovillous adenoma with severe atypia. As the findings suggested that the lesion had an adenocarcinoma component but was confined to the mucosal layer, we decided to carry out ESD and successfully removed the tumor in one piece. The resected tumor was 20 x 15 mm in size. Histopathological examination revealed that the lesion was a well-differentiated mucosal adenocarcinoma with no lymphovascular invasion. Mucosal duodenal cancer is extremely rare, and ESD of a lesion in the duodenum requires a high level of skill. To the best of our knowledge, this case is the first report of successful ESD carried out in a case of mucosal duodenal cancer.

Borderline cases between benignancy and malignancy of the duodenum diagnosed successfully by endoscopic submucosal dissection.

OBJECTIVE. Due to advances in endoscopic equipment, primary duodenal tumors are found more frequently than in the past. We performed endoscopic submucosal dissection (ESD) to diagnose and treat four non-ampullary duodenal tumors. MATERIAL AND METHODS. During endoscopic treatment, marks were placed around the circumference of the tumor and sufficient amounts of physiological saline with epinephrine were injected into the submucosal layer to elevate the lesion. An incision was made around the lesion using a long-type needle knife and the isolated lesion was resected completely "en bloc". In this procedure, a cylindrical transparent hood was attached to the endoscopy apparatus to allow for satisfactory visualization of the procedure. RESULTS. The mean age of the patients was 69 years. The patients consisted of two males and two females. Gross examination showed three flat, elevated lesions and one polypoid lesion. Tumor size ranged from 10 to 31 mm in maximum diameter. Histological examination revealed two cases of well-differentiated adenocarcinomas and two cases of tubular adenomas with severe atypia. Procedure-related complications consisting of perforation occurred in two cases and were resolved under close postoperative observation including antibiotics, use of a nasogastric tube and nil per oral feeding status. The mean follow-up period was 18 months and none of the patients experienced tumor recurrence after the treatment. CONCLUSIONS. Since tissue obtained from endoscopic biopsies can sometimes prove difficult for definitive histological diagnosis, ESD may play an important role in the management of cases appearing to border on malignancy. In addition, ESD allows for minimally invasive treatment without sacrificing the possibility of cure for duodenal carcinoma.

Primary non-Hodgkin's lymphoma of the main hepatic duct junction.

A rare case of primary non-Hodgkin's lymphoma of the main hepatic duct junction is reported. A 71-year-old man was admitted for treatment of obstructive jaundice. Radiological examination revealed stenosis of the main hepatic duct junction. Biliary drainage was not necessary because total bilirubin decreased spontaneously. A left hepatic and caudate lobectomy, combined with resection of bile ducts and lymph node dissection, was performed with the preoperative diagnosis of cholangiocarcinoma of the main hepatic duct junction. Macroscopic examination of the resected specimen revealed tumorous growth in the main hepatic duct junction. Histological and immunochemistry findings disclosed a mucosa-associated lymphoid tissue (MALT) lymphoma. The patient received three courses of combination chemotherapy [cyclophosphamide, doxorubicin, vincristine and prednisolone (CHOP)], and there was no evidence of recurrence 45 months after the surgery. Although primary non-Hodgkin's lymphoma of the main hepatic duct junction is extremely rare and difficult to diagnose preoperatively, aggressive surgery followed by chemotherapy, as here, is a possible curative option.

Efficacy of an extracorporeal flat-plate bioartificial liver in treating fulminant hepatic failure.

BACKGROUND: Fulminant hepatic failure is associated with a high mortality rate. Orthotopic liver transplantation is the only established treatment for patients who do not respond to medical management. A major limitation of this treatment is a shortage of donor organs, resulting in many patients dying while waiting for a transplant. An extracorporeal bioartificial liver (BAL) has the potential to provide temporary support for patients with fulminant hepatic failure (FHF) and for patients awaiting orthotopic liver transplantation. We developed a flat-plate BAL with an internal membrane oxygenator in which porcine hepatocytes were cultured as a monolayer. MATERIALS AND METHODS: Twenty-four hours after cannulation of the left carotid artery and right jugular vein, FHF was induced in rats by administering 2 intraperitoneal injections of D-galactosamine (GalN) (1.2 g/kg) at a 12-h interval. The rats were connected to a BAL device 24 h after the first GalN injection and underwent extracorporeal perfusion for a duration of 10 h. Liver histology, liver-specific markers, and animal survival up to 168 h (7 days) were examined. RESULTS: Histologically, liver damage was reduced in the animal group treated with the hepatocyte-based BAL device. Significant reductions occurred in the plasma ammonia levels and prothrombin times in the group treated with the seeded BAL device. Animal survival in the group treated with the seeded BAL device was significantly higher (50.0%) than in the control animal group treated with an unseeded BAL device (11.1%). CONCLUSIONS: This flat-plate BAL with an internal membrane oxygenator and cultured porcine hepatocytes has yielded encouraging results in the treatment of rats with GalN-induced FHF.