Low-grade intraductal carcinoma of the salivary gland with prominent oncocytic change: a newly described variant.

AIMS: Low-grade intraductal carcinoma (LG-IDC) is a clinically indolent malignant tumour of the salivary glands. Because of its rarity, the histological variants of LG-IDC have not been well characterised. Herein, we describe five LG-IDC cases with prominent oncocytic change in the major salivary glands. METHODS AND RESULTS: We examined five cases, three males and two females (mean age = 63 years), of LG-IDC with oncocytic change. The sites affected by LG-IDC were the parotid and submandibular glands. The lesions were macroscopically unilocular or multilocular cysts with a solid tumour arising from the cyst wall. Smaller tumour cell nests were also observed. As with classic LG-IDC, the cyst wall was surrounded by myoepithelial cells with no invasive component. The tumour cells had abundant oncocytic cytoplasm and proliferated in a low-papillary, tubular or cribriform pattern. Immunohistochemically, the tumour cells were diffusely positive for pan-cytokeratin, S100, mammaglobin and antimitochondria antibody, and were negative for androgen receptor and gross cystic disease fluid protein-15. Unlike classic LG-IDC, some of these cases demonstrated focal invagination of myoepithelial cells in the intraductal tumour. CONCLUSION: Oncocytic LG-IDC should be recognised as a histologically unique variant of LG-IDC. Awareness of this entity is important to avoid erroneous diagnosis and inappropriate treatment for histological mimics.

Intra-abdominal mucinous adenocarcinoma of urachal origin: report of a case.

Intra-abdominal mucinous cystic tumors can be difficult to diagnose preoperatively. We report a case of histologically diagnosed primary urachal adenocarcinoma: a rare type of bladder tumor. This case report is interesting for clinicians. The patient was an 86-year-old man who presented with acute abdominal pain. Computed tomography (CT) showed a large cystic mass with calcification, near the apex of the urinary bladder. Laparotomy revealed a large intra-abdominal cystic mass adherent to the anterior abdominal wall and superior to the urinary bladder. We performed laparoscopic-assisted resection and partial cystectomy. The cystic mass measured approximately 15 × 14 × 11 cm and contained mucinous material. Histological examination revealed that it extended to the muscle of the bladder wall and that its epithelium was composed of atypical cells with increased papillary morphology. The mucinous material was glycoprotein with degenerative fatty tissue, and calcification was recognized partly in the specimen. Thus, we comprehensively diagnosed a mucinous cystic adenocarcinoma of urachal origin.

A surgical resection of hepatic granuloma mimicking intrahepatic cholangiocarcinoma: a case report.

Hepatic granuloma is relatively rare, and benign tumor of the liver. Herein, we report an unusual case of hepatic granuloma mimicking intrahepatic cholangiocarcinoma (ICC). An 82-year-old woman with a history of viral hepatitis B was admitted for investigation of liver mass in the left lobe. Dynamic computed tomography revealed a mostly hypo-enhancing main tumor with a peripheral ring enhancement, and positron emission tomography demonstrated localized an abnormal accumulation of fludeoxyglucose. Considering the possibility of malignant disease, extended left hepatectomy was performed. The resected tumor was macroscopically a periductal infiltrating nodular type, 4.5 × 3.6 cm in diameter. The pathological findings showed that granuloma and coagulative necrosis were present, and diagnosis of hepatic granuloma was confirmed. Pathological studies demonstrated that periodic acid-Schiff stain, Grocott-Gomori stain and Ziehl-Neelsen stain were all negative in the lesion.

Clear cell carcinoma of the seminal vesicle in a young adult.

Introduction: Most seminal vesicle malignancies are secondary to prostate or bladder cancer. Herein, we report a case of primary clear cell carcinoma of the seminal vesicle. Case presentation: A 27-year-old man was referred to our department for hematospermia and macroscopic hematuria. A digital rectal examination showed a soft elastic prostatic mass. Cystoscopy showed no bladder abnormalities, and tumor marker tests were unremarkable. Contrast-enhanced computed tomography and magnetic resonance imaging revealed a cystic tumor containing an enhanced nodule near the prostate and seminal vesicle. The tumor was removed en bloc with the prostate and seminal vesicle through a laparoscopic radical prostatectomy. A histopathologic examination confirmed the diagnosis, with the tumor likely arising from a remnant Müllerian epithelium. A 1-year follow-up revealed local tumor recurrence, prompting laparoscopy. Conclusion: A standard therapy for primary seminal vesicle carcinoma has not been established. Further studies are necessary to determine the optimal treatment strategy.

A case of primary carcinosarcoma of the liver with combined hepatocellular carcinoma and cholangiocarcinoma.

Primary carcinosarcoma of the liver is extremely rare. Here, we report an unusual case of carcinosarcoma of the liver containing combined hepatocellular carcinoma and cholangiocarcinoma. A 66-year-old man with a history of viral hepatitis B was admitted for investigation of multiple liver masses. Dynamic computed tomography revealed a mostly hypoenhancing main tumor with a peripheral ring enhancement and several satellite nodules. After transcatheter arterial chemoembolization and portal vein embolization, an extended right posterior sectionectomy was performed. The resected tumor was macroscopically a simple nodular type, 4.3 × 4.2 cm in diameter, with a dense fibrous capsule. The pathological findings showed that both carcinomatous and sarcomatous elements were present, and diagnosis of carcinosarcoma of the liver was confirmed. The carcinomatous element consisted of hepatocellular carcinoma and cholangiocarcinoma. The sarcomatous element was composed of spindle cells. Immunohistochemical studies demonstrated that cytokeratin AE1/AE3, human serum albumin, cytokeratin 7, and Arginase-1 were partially positive in tumor cells of the carcinomatous element but not in tumor cells of the sarcomatous element. Follow-up for 30 months after surgery has shown no signs of recurrence.

Renal metastasis from intrahepatic cholangiocarcinoma.

Metastases to the kidney are extremely rare and intrahepatic cholangiocarcinoma (ICC) is difficult to treat. In this study, we report a case of renal metastasis from ICC. A 72-year-old man who had been followed-up for chronic hepatitis C was diagnosed with ICC in the segment 8 and underwent S8 segmentectomy in 2014. During follow-up, the serum levels of carcinoembryonic antigen and carbohydrate antigen 19-9 were slightly elevated, and abdominal contrast-enhanced computed tomography revealed a low-density mass preceded by rim enhancement in the arterial phase measuring 1.5 × 1.5 cm in the segment 6, and a hypovascular mass measuring 2.2 × 2.0 cm in the upper pole of the left kidney in 2017. He underwent partial hepatectomy and partial nephrectomy. Based on postoperative histological findings combined with immunohistochemical analysis, the tumors both in the liver and kidney were diagnosed as recurrent ICC.

Langerhans cell histiocytosis confined to extrahepatic bile duct causing sclerosing cholangitis in child: a case report.

BACKGROUND: Langerhans cell histiocytosis (LCH) is an abnormal accumulation of Langerhans cells in various organs that sometimes induces organ dysfunction. LCH can affect the liver, resulting in sclerosing cholangitis and biliary cirrhosis. However, liver and bile duct involvement is usually observed in the disseminated form of LCH. We herein report a rare case of LCH localized only in the extrahepatic bile duct that resulted in severe liver cirrhosis. CASE PRESENTATION: A 3-year-old boy with elevated liver enzymes, obstructive jaundice, and dilation of the common bile duct was referred to our institution. Contrast-enhanced computed tomography showed atrophy of the right hepatic lobe, relative hypertrophy of the left hepatic lobe, choledocholiths, and biliary debris extensively with biliary duct dilation. Magnetic resonance cholangiopancreatography revealed dilation of the intrahepatic and extrahepatic bile ducts and multiple choleliths in the gallbladder and common bile duct. Laparoscopic cholecystectomy, intraoperative cholangiography, liver biopsy, and gastrointestinal fiberscopy were performed. A liver specimen showed severe biliary cirrhosis due to sclerosing cholangitis. The patient then underwent living-donor liver transplantation because of severe liver cirrhosis 3 months after the first surgery. The common bile duct was not suitable for duct-to-duct anastomosis and was resected because of severe inflammation. Histologic sections of the common bile duct showed histiocytic cell proliferation. Immunohistochemistry revealed histiocytoses that were positive for Langerin, S-100 protein, and CD1a. However, no histiocytic cell proliferation was noted in the liver tissue. The definitive diagnosis was LCH localized to the extrahepatic bile duct. LCH in the extrahepatic bile duct seemed to cause sclerosing cholangitis. The patient was discharged uneventfully 2 months after living-donor liver transplantation. CONCLUSIONS: LCH localized to the extrahepatic bile duct is extremely rare; however, LCH can still affect the extrahepatic bile ducts on occasion. LCH should be considered as a differential diagnosis if pediatric patients show the presence of sclerosing cholangitis.

Interdigitating dendritic cell sarcoma of the ileum recurred in multiple lymph nodes and duodenum three years after operation without chemotherapy.

Neoplasms derived from interdigitating dendritic cell are extremely rare. Here we describe a case of a 47-year-old man with interdigitating dendritic cell sarcoma (IDCS) in the ileum. He was admitted to a hospital due to ileus. The ileal tumor, measuring 2cm, was detected and resected with regional lymphadenectomy. At that time, a pathologic diagnosis of malignant peripheral nerve sheath tumor was made. The patient, who was not treated with chemotherapy, showed no signs of recurrence. After three years, we detected cervical lymphadenopathy and multiple duodenal masses in the patient in our hospital. Oval to spindle-shaped atypical cells, which resembled ileal tumor cells, infiltrated into the lymph node and duodenum. Immunohistochemical staining of these three lesions revealed positivity of S100 protein and several macrophage-related antigens. Based on the histologic and immunohistochemical analysis, the histopathologic diagnosis of IDCS was confirmed. To our knowledge, five cases of IDCS arising in the intestinal tract have been reported to date, and only one case, treated with both surgery and chemotherapy, led to remission. This is the first case that has a comparatively favorable prognosis without chemotherapy after surgery.