Surgical Case of Metachronous Occurrence of Intracholecystic Papillary Neoplasm in the Remnant Cystic Duct 19 Years after Cholecystectomy

Abstract

INTRODUCTION: Intracholecystic papillary neoplasm (ICPN) is a recently identified disease characterized by papillary pre-invasive neoplasm of the gallbladder. Despite its characterization, the natural history of ICPN remains elusive. Furthermore, a few cases of metachronous ICPN in the remnant biliary system have been documented. Here, we report a surgical case involving metachronous ICPN in the remnant cystic duct 19 years post-cholecystectomy for primary ICPN.

CASE PRESENTATION: A 77-year-old man presented to our hospital with general fatigue and jaundice. He had previously undergone an open cholecystectomy and lithotomy for gallbladder cancer and common bile duct stones 19 years earlier. Blood tests revealed elevated levels of hepatobiliary enzymes and tumor markers. Both computed tomography and magnetic resonance imaging indicated dilatation of the intrahepatic and common bile ducts, and an enhanced nodule was observed in the common hepatic duct. Intraductal ultrasonography identified a papillary tumor infiltrating the distal bile duct from the common hepatic duct. Brush cytology subsequently helped confirm adenocarcinoma. Consequently, the patient was diagnosed with Bismuth type 1 perihilar cholangiocarcinoma and underwent subtotal stomach-preserving pancreaticoduodenectomy. Histological examination revealed the tumor as pancreatobiliary-type ICPN associated with invasive carcinoma, which had originated in the remnant cystic duct and invaded the common hepatic duct. A retrospective review of the resected gallbladder specimens from 19 years earlier confirmed ICPN according to the current classification, establishing this as a metachronous occurrence of ICPN.

CONCLUSIONS: These findings suggest that ICPN can recur as metachronous lesions in the remnant biliary system after resection of primary lesion, highlighting the necessity of sustained, long-term biliary surveillance following primary lesion resection.