Tamura H, Ozono Y, Uchida K, Uchiyama N, Hatada H, Ogawa S, Iwakiri H, Kawakami H. Multiple intrahepatic artery aneurysms during the treatment for IgG4-related sclerosing cholangitis: A case report. World J Hepatol 2024; 16(12): 1505-1514 [DOI: 10.4254/wjh.v16.i12.1505]
Corresponding Author of This Article
Hiroshi Kawakami, MD, PhD, Professor, Division of Gastroenterology and Hepatology, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, 5200 Kihara, Kiyotake, Miyazaki 889-1692, Japan. hiropon@med.miyazaki-u.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/
Hotaka Tamura, Yoshinori Ozono, Keisuke Uchida, Naomi Uchiyama, Hiroshi Hatada, Souichiro Ogawa, Hisayoshi Iwakiri, Hiroshi Kawakami, Division of Gastroenterology and Hepatology, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Miyazaki 889-1692, Japan
Author contributions: Tamura H and Ozono Y contributed to manuscript writing and editing, and data collection; Kawakami H contributed to manuscript editing and data collection; Uchida K, Uchiyama N, Hatada H, Ogawa S, Iwakiri H, Kawakami H contributed to conceptualization and supervision; all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest to disclose.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Hiroshi Kawakami, MD, PhD, Professor, Division of Gastroenterology and Hepatology, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, 5200 Kihara, Kiyotake, Miyazaki 889-1692, Japan. hiropon@med.miyazaki-u.ac.jp
Received: May 21, 2024 Revised: October 1, 2024 Accepted: October 31, 2024 Published online: December 27, 2024 Processing time: 192 Days and 3.1 Hours
Abstract
BACKGROUND
The purpose of this case report is to describe a case of multiple intrahepatic artery aneurysms during treatment for IgG4-related sclerosing cholangitis (IgG4-SC) and to provide information for daily practice.
CASE SUMMARY
A 64-year-old Japanese woman was diagnosed with IgG4-SC five years prior and was receiving maintenance treatment with prednisolone 7.5-10 mg/day. She developed abdominal pain and a sudden onset of black stool and was admitted to our hospital. Abdominal contrast-enhanced computed tomography (CT) and ultrasonography (US) revealed multiple intrahepatic artery aneurysms that developed during the treatment for IgG4-SC. Emergency transarterial embolization for multiple hepatic artery aneurysms was performed. Hepatic artery aneurysms disappeared on contrast-enhanced CT and US, the progression of anemia ceased, and the melena resolved. Thus, hemostasis was achieved.
CONCLUSION
Hepatic artery aneurysms should be considered poor prognostic complications of IgG4-SC.
Core Tip: Hepatic artery pseudoaneurysm is a disease with poor prognosis due to high rupture and mortality rates. IgG4-related sclerosing cholangitis (IgG4-SC) is a disease caused by an autoimmune mechanism. we report a case of multiple ruptured intrahepatic artery pseudoaneurysms that occurred during the treatment of IgG4-SC. We consider the cause is complex: IgG4-related vasculitis, severe acute obstructive cholangitis and arteriosclerosis. Hepatic artery aneurysms should be considered poor prognostic complications of IgG4-SC.
