Miyazu T, Ishida N, Asai Y, Tamura S, Tani S, Yamade M, Iwaizumi M, Hamaya Y, Osawa S, Baba S, Sugimoto K. Intrahepatic cholangiocarcinoma in patients with primary sclerosing cholangitis and ulcerative colitis: Two case reports. World J Gastrointest Surg 2023; 15(6): 1224-1231 [PMID: 37405109 DOI: 10.4240/wjgs.v15.i6.1224]
Corresponding Author of This Article
Ken Sugimoto, MD, PhD, Professor, First Department of Medicine, Hamamatsu University School of Medicine, 1-20-1 Handayama, Higashi-ku, Hamamatsu 431-3192, Japan. sugimken@hama-med.ac.jp
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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/
Takahiro Miyazu, Yusuke Asai, Satoshi Tamura, Shinya Tani, Mihoko Yamade, Yasushi Hamaya, Ken Sugimoto, First Department of Medicine, Hamamatsu University School of Medicine, Hamamatsu 431-3192, Japan
Natsuki Ishida, Satoshi Osawa, Department of Endoscopic and Photodynamic Medicine, Hamamatsu University School of Medicine, Hamamatsu 431-3192, Japan
Moriya Iwaizumi, Department of Laboratory Medicine, Hamamatsu University School of Medicine, Hamamatsu 431-3192, Japan
Satoshi Baba, Department of Diagnostic Pathology, Hamamatsu University School of Medicine, Hamamatsu 431-3192, Japan
Author contributions: Miyazu T and Sugimoto K wrote the manuscript; Ishida N, Asai Y, Tamura S, Tani S, Yamade M, Iwaizumi M, Hamaya Y, and Osawa S contributed to the manuscript design and coordination; Baba S contributed to the pathological examination.
Informed consent statement: The manuscript and all images were published with the informed consent of the patient or their families.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read CARE Checklist (2016), and the manuscript was prepared and revised according to 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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Ken Sugimoto, MD, PhD, Professor, First Department of Medicine, Hamamatsu University School of Medicine, 1-20-1 Handayama, Higashi-ku, Hamamatsu 431-3192, Japan. sugimken@hama-med.ac.jp
Received: December 25, 2022 Peer-review started: December 25, 2022 First decision: February 28, 2023 Revised: March 10, 2023 Accepted: April 12, 2023 Article in press: April 12, 2023 Published online: June 27, 2023 Processing time: 172 Days and 2.9 Hours
Abstract
BACKGROUND
Primary sclerosing cholangitis (PSC) is an extraintestinal manifestation of ulcerative colitis (UC). PSC is a well-known risk factor for intrahepatic cholangiocarcinoma (ICC), and ICC is known to have a poor prognosis.
CASE SUMMARY
We present two cases of ICC in patients with PSC associated with UC. In the first case, a tumor was found by magnetic resonance imaging (MRI) in the liver of a patient with PSC and UC who presented to our hospital with right-sided rib pain. The second patient was asymptomatic, but we unexpectedly detected two liver tumors in an MRI performed to evaluate bile duct stenosis associated with PSC. ICC was strongly suspected by computed tomography and MRI in both cases, and surgery was performed, but unfortunately, the first patient died of ICC recurrence 16 mo postoperatively, and the second patient died of liver failure 14 mo postoperatively.
CONCLUSION
Careful follow-up of patients with UC and PSC with imaging and blood tests is necessary for early detection of ICC.
Core Tip: Intrahepatic cholangiocarcinoma (ICC) commonly develops on top of primary sclerosing cholangitis (PSC) associated with ulcerative colitis (UC). Both of our patients died, although they were asymptomatic or mildly symptomatic at the time the ICC was discovered. Patients with long-term PSC coexisting with UC require regular follow-up with imaging such as magnetic resonance imaging even if they are asymptomatic.
