Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 21, 2022; 10(3): 1000-1007
Published online Jan 21, 2022. doi: 10.12998/wjcc.v10.i3.1000
Multicentric recurrence of intraductal papillary neoplasm of bile duct after spontaneous detachment of primary tumor: A case report
Hiroki Fukuya, Akifumi Kuwano, Shigehiro Nagasawa, Yusuke Morita, Kosuke Tanaka, Masayoshi Yada, Akihide Masumoto, Kenta Motomura
Hiroki Fukuya, Akifumi Kuwano, Shigehiro Nagasawa, Yusuke Morita, Kosuke Tanaka, Masayoshi Yada, Akihide Masumoto, Kenta Motomura, Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
Author contributions: Fukuya H, Kuwano A, Nagasawa S, Morita Y, Tanaka K, Yada M, Masumoto A and Motomura K reviewed the literature and contributed to writing a draft of the manuscript; all authors issued final approval for the submitted version.
Informed consent statement: Informed consent was obtained from the patient allowing publication of this report and accompanying images.
Conflict-of-interest statement: The authors declare no potential conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Kenta Motomura, MD, PhD, Chief Doctor, Department of Hepatology, Iizuka Hospital, 3-83 Yoshio-machi, Iizuka 820-8505, Fukuoka, Japan. kmotomurah2@aih-net.com
Received: April 22, 2021
Peer-review started: April 22, 2021
First decision: June 24, 2021
Revised: July 7, 2021
Accepted: December 21, 2021
Article in press: December 21, 2021
Published online: January 21, 2022
Abstract
BACKGROUND

Intraductal papillary neoplasm of the bile duct (IPNB) rarely recurs in a multicentric manner. We encountered a patient with multiple recurrences of the gastric subtype of IPNB one year after spontaneous detachment of the primary tumor during peroral cholangioscopy (POCS).

CASE SUMMARY

A 68-year-old woman on maintenance hemodialysis because of lupus nephritis had several cardiovascular diseases and a pancreatic intraductal papillary mucinous neoplasm (IPMN). She was referred to our department for dilation of the common bile duct (CBD) and a tumor in the lumen, detected using ultrasonography. She had no complaints, and blood tests of hepatobiliary enzymes were normal. Magnetic resonance cholangiopancreatography (MRCP) showed a papillary tumor in the CBD with a filling defect detected using endoscopic retrograde cholangiography (ERC). Intraductal ultrasonography revealed a papillary tumor and stalk at the CBD. During POCS, the tumor spontaneously detached with its stalk into the CBD. Pathology showed low-intermediate nuclear atypia of the gastric subtype of IPNB. After 1 year, follow-up MRCP showed multiple tumors distributed from the left hepatic duct to the CBD. ERC and POCS showed multicentric tumors. She was alive without hepatobiliary symptoms at least two years after initial diagnosis of IPNB.

CONCLUSION

The patient experienced gastric subtype of IPNB without curative resection. Observation may be reasonable for patients with this subtype.

Keywords: Bile duct neoplasm, Neoplasm Recurrence, Pancreatic intraductal neoplasms, Magnetic resonance cholangiopancreatography, Endoscopic retrograde cholangiography, Peroral cholangioscopy, Case report

Core Tip: Multiple occurrences of intraductal papillary neoplasm of bile duct (IPNB) are rare. Here we present the case of a patient with multicentric recurrence of IPNB after spontaneous detachment of the primary tumor. She harbored an asynchronous intraductal papillary mucinous neoplasm and experienced gastric subtype of IPNB without complete resection.