Atsuya Yonemori

Intraductal oncocytic papillary neoplasm of the bile duct: the first case of peribiliary gland origin

We report herein the first case of intraductal oncocytic papillary neoplasm of the bile duct arising from a peribiliary gland of the left hepatic duct. The patient was a 63-year-old Japanese man. Radiological and cholangioscopic examinations revealed intraductal tumor of the left hepatic duct. After pathological diagnosis of adenocarcinoma by cholangioscopic biopsy, a surgical hepatobiliary resection was performed. Pathological examination revealed papillary tumor in the left hepatic duct. Histologically, the tumor was identified as papillary neoplasm comprising oncocytic cells and delicate fibrovascular cores. Interestingly, this tumor originated from the cystic space in the bile duct wall. This cystic space was histologically identified as a cystically dilated peribiliary gland. Carcinoma in situ was observed in this cystic peribiliary gland at the bottom of the tumor, but not on any areas of biliary epithelium. This case suggests that intraductal papillary neoplasm can arise from both biliary epithelium and peribiliary glands.

Prognostic Impact of Regional Lymph Node Micrometastasis in Patients With Node-Negative Biliary Cancer

Objective:To immunohistochemically identify regional lymph node micrometastases in patients with regional node-negative biliary cancer who underwent curative resection, and to evaluate their clinical significance. Summary Background Data:The clinical significance of immunohistochemically detected lymph node micrometastasis has recently been evaluated in various tumors. However, few reports have focused on this issue with regard to biliary cancer. Methods:A total of 1421 regional lymph nodes from 151 patients with biliary cancer with negative regional nodes (as determined by conventional methods) were immunostained with antibody against cytokeratins 7 and 8 (CAM5.2). Prognostic impact was evaluated among patients with no metastasis, micrometastasis, and obvious metastasis detected by hematoxylin and eosin staining. Immunostained tumor foci were classified as small micrometastasis or large micrometastasis according to size (above or below 0.2 mm). Results:CAM5.2-positive occult carcinoma cells in regional lymph nodes were detected in 33 (22%) of 151 patients and 49 (3%) of 1421 regional lymph nodes. Small micrometastases were detected in 23 patients, whereas large micrometastases were found in 10 patients. Survival for patients with micrometastasis was significantly worse than that for patients without (P = 0.0051), but was significantly better than that for patients with overt metastasis (P = 0.0092). No significant difference in postoperative survival was seen between patients with small and large micrometastases (P = 0.4221). Conclusions:Occult cancer cells were present in regional lymph nodes of 22% patients with regional node-negative biliary cancer, and were associated with significantly worse survival. Patients with micrometastases should be treated as carefully as node-positive patients.

Prognostic impact of para‐aortic lymph node micrometastasis in patients with regional node‐positive biliary cancer

The presence of para‐aortic lymph node metastasis in biliary cancer has a negative impact on prognosis. The relevance of para‐aortic lymph node micrometastasis is unknown.

Spindle Cell-Type Undifferentiated Carcinoma of the Common Bile Duct of the Hepatic Hilus: Report of a Case

Spindle cell-type undifferentiated carcinoma arising from the extrahepatic bile duct is extremely rare. We herein report a case of this type of carcinoma in the common bile duct of the hepatic hilus. A 59-year-old man was admitted to our hospital complaining of jaundice. The laboratory data revealed an elevation of the serum carbohydrate antigen 19–9 level. Cholangiography revealed a complete obliteration of the left hepatic bile duct and stenosis of the bile duct from the superior to the right hepatic bile duct. Computed tomography showed the tumor to measure 15 × 12 mm in the hepatic hilus, with the obliteration of the right to main trunk of the portal vein and a swollen lymph node in the hepato-duodenum ligament. Arteriography revealed a kink of the right hepatic artery; therefore an encasement of the right hepatic artery was suspected. We preoperatively diagnosed hilus bile duct carcinoma and scheduled a right trisection hepatectomy. Intraoperative frozen sections taken from the tumor and tissues around the hepatic arteries showed spindle and inflammatory cells; therefore an inflammatory pseudotumor was diagnosed intraoperatively. As the right hepatic bile duct was occluded, a right lobe hepatectomy was performed. However, a permanent section revealed both spindle cells and poorly differentiated tubular adenocarcinoma cells positive for CAM5.2, AE1/AE3, and vimentin. On the basis of these findings, the tumor was finally diagnosed to be spindle cell-type undifferentiated carcinoma. Unfortunately, the patient died of pulmonary infarction 11 days after the operation.

Para-Aortic Lymph Node Micrometastasis in Patients with Node-Negative Biliary Cancer

Background/Aims: The presence of para-aortic lymph node metastasis in biliary cancer negatively impacts prognosis. The present study aims to immunohistochemically identify and evaluate the clinical significance of para-aortic lymph node micrometastases in 66 patients who had undergone curative resection of biliary cancer. Methods: We used an antibody against cytokeratins 7 and 8 (CAM5.2) to immunostain 529 para-aortic lymph nodes that were negative according to conventional analysis from 66 patients with biliary cancer. Results: We detected CAM5.2-positive occult carcinoma cells in para-aortic lymph nodes from 3 (5%) of the 66 patients and in 3 (0.6%) of the 529 para-aortic lymph nodes. One of the 3 patients also had micrometastasis in the regional lymph nodes. All 3 patients with para-aortic lymph node micrometastasis are alive at 45, 48 and 90 months after surgery despite having locally advanced cancer. Conclusions: Occult cancer cells were identified in para-aortic lymph nodes from 5% of patients with node-negative biliary cancer, yet these patients have survived over the long term. The presence of para-aortic nodal micrometastasis might not have an influence on survival. However, further studies using a greater number of patients are required to support this notion.

Gastrointestinal Obstruction due to Solitary Lymph Node Recurrence of Alpha-Fetoprotein-Producing Gastric Carcinoma with Enteroblastic Differentiation

We report an unusual case of alpha-fetoprotein (AFP)-producing gastric carcinoma with enteroblastic differentiation. A 75-year-old woman was admitted to our hospital with occasional upper abdominal discomfort. We performed gastroscopy and observed a type 2 tumor, primarily in the pyloric region. Histological examination of biopsies confirmed gastric adenocarcinoma. Based on these findings, we diagnosed gastric adenocarcinoma and performed laparoscopic distal gastrectomy with lymph node dissection. Histological examination revealed an invasive lesion composed of adenocarcinoma with a tubulopapillary growth pattern. Tumor cells were cuboidal in shape with characteristically clear cytoplasm rich in glycogen. Two regional lymph node metastases were seen microscopically. Immunohistochemically these cells were positive for AFP, carcinoembryonic antigen, caudal-type homeobox transcription factor 2 and common acute lymphoblastic leukemia antigen. The final diagnosis was AFP-producing gastric carcinoma with enteroblastic differentiation. 26 months after initial surgery, the patient was readmitted to our hospital for gastrointestinal obstruction due to lymph node recurrence.

Goblet cell carcinoid of the rectum with lymph node metastasis: report of a case.

We report an unusual case of goblet cell carcinoid (GCC) of the rectum. A 75-year-old man was admitted to our hospital with anal bleeding, and a hard tumor was felt on the anterior wall of the lower rectum during rectal examination. We performed colonoscopy, and found a 30-mm type 2 tumor in the lower rectum and anal canal. Histological examination of biopsies revealed rectal adenocarcinoma. Based on these findings, we diagnosed rectal adenocarcinoma and performed Miles’ operation with lymph node dissection. Histological examination revealed an invasive lesion composed of signet-ring-like cells. Seven regional lymph node metastases were seen microscopically. The tumor produced copious mucin, which was stained with Alcian blue. Immunohistochemistry was positive for synaptophysin, chromogranin A, CD56, carcinoembryonic antigen, p53, Ki-67, E-cadherin, and cytokeratin 20. The final diagnosis was GCC of the rectum.

Ganglioneuroblastoma arising within a retroperitoneal mature cystic teratoma.

We discuss an extremely rare case of ganglioneuroblastoma arising within a retroperitoneal mature cystic teratoma. Radiological examinations showed a cystic tumor sandwiched between the pancreas and left kidney. Surgery was scheduled because the tumor seemed to have originated from the pancreas. En-block resection of the tumor with distal pancreatectomy, splenectomy, and left adrenalectomy was performed. In terms of macroscopic appearance, the tumor mainly consisted of a unilocular cystic mass, but the presence of a smaller, solid mass was also noted within the tumor. Histopathologic examination confirmed that the cystic mass was consistent with a mature cystic teratoma of the retroperitoneum, and in addition, a ganglioneuroblastoma was evident in the solid component. Histopathologically, the ganglioneuroblastomatous area was intimately associated with dermoid tissue of the mature cystic teratoma, thus this case was diagnosed to be a mature cystic teratoma with malignant transformation. To best of our knowledge, this is the first reported case of ganglioneuroblastoma arising in a mature cystic teratoma.