A case of gallbladder adenocarcinoma arising in association with intracystic papillary neoplasm (ICPN) with abundant muc

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A case of gallbladder adenocarcinoma arising in association with intracystic papillary neoplasm (ICPN) with abundant mucin production Takuya Oba1   · Norihiro Sato1 · Toshihisa Tamura1 · Masahiro Komeda1 · Yasuhiro Adachi1 · Atsuji Matsuyama2 · Keiji Hirata1 Received: 3 June 2020 / Accepted: 7 September 2020 © Japanese Society of Gastroenterology 2020

Abstract Intracystic papillary neoplasm (ICPN) of the gallbladder is a rare clinicopathological entity with a wide range of malignant potentials. Here, we report a case of mucin-producing gallbladder carcinoma possibly derived from ICPN. A 78-year-old female patient was referred to our hospital for examination of jaundice. Abdominal CT showed dilated biliary trees and a contrast-enhanced large polypoid mass in the gallbladder. Duodenoscopy showed a large amount of mucin extravasating from the ampulla of Vater. Bile cytology showed no evidence of malignancy. Under the diagnosis of mucin-producing gallbladder tumor, we performed laparoscopic cholecystectomy. Macroscopically, there was a large papillary tumor throughout the entire gallbladder mucosa. Pathological examinations showed a gallbladder adenocarcinoma localized to the mucosa in association with ICPN. Immunohistochemical analysis of the tumor revealed positive staining for MUC2 and MUC5AC but negative for MUC1 and MUC6, suggestive of the intestinal type. Keywords  Mucin-producing gallbladder carcinoma · Intracystic papillary neoplasm · Immunohistochemical analysis Abbreviations ICPN Intracholecystic papillary–tubular neoplasm IPNB Intraductal papillary neoplasm of the bile duct IPMN Intraductal papillary mucinous neoplasm US Ultrasonography CT Computed tomography ERC Endoscopic retrograde cholangiography CEA Carcinoembryonic antigen CA19-9 Carbohydrate antigen 19-9

* Takuya Oba t‑[email protected]‑u.ac.jp 1



Department of Surgery 1, School of Medicine, University of Occupational and Environmental Health, Kitakyushu 807‑8555, Japan



Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, Kitakyushu, Japan

2

Introduction Intracystic papillary neoplasm or intracholecystic papillary–tubular neoplasm (ICPN) of the gallbladder is a newly established histopathological entity, which was first defined in the 2010 World Health Organization (WHO) classification [1]. Because of the limited number of reported cases, the terminology of ICPN has not been widely recognized. ICPN has been considered as a counterpart of intraductal papillary neoplasm of the bile duct (IPNB) and intraductal papillary mucinous neoplasm of the pancreas (IPMN). ICPN includes a wide spectrum of neoplasms with different malignant potentials, ranging from low-grade dysplasia to invasive carcinoma. In a large series of 123 ICPN cases, more than half (55%) of the cases had an associated invasive carcinoma [2]. Here, we report a rare case of mucin-producing gallbladder carcinoma that arose in association with ICPN. In our present case, pathological and immunohistochemical exam