Primary Sclerosing Cholangitis Complicated with Aphthoid Colitis Difficult to Diagnose from IgG4-Related Sclerosing Cholangitis

Jin Imai; Hitoshi Ichikawa; Motoki Kaneko; Hiroyuki Ito; Shinji Takashimizu; Takayuki Shirai; Takuma Tajiri; Norihito Watanabe; Hidekazu Suzuki

doi:10.1159/000521538

Case Reports in Gastroenterology (Feb 2022)

Primary Sclerosing Cholangitis Complicated with Aphthoid Colitis Difficult to Diagnose from IgG4-Related Sclerosing Cholangitis

Jin Imai,
Hitoshi Ichikawa,
Motoki Kaneko,
Hiroyuki Ito,
Shinji Takashimizu,
Takayuki Shirai,
Takuma Tajiri,
Norihito Watanabe,
Hidekazu Suzuki

Affiliations

Jin Imai: Department of Gastroenterology, Tokai University School of Medicine, Kanagawa, Japan
Hitoshi Ichikawa: Department of Gastroenterology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Motoki Kaneko: Department of Gastroenterology, Tokai University School of Medicine, Kanagawa, Japan
Hiroyuki Ito: Department of Gastroenterology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Shinji Takashimizu: Department of Gastroenterology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Takayuki Shirai: Department of Gastroenterology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Takuma Tajiri: Department of Pathology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Norihito Watanabe: ORCiD; Department of Gastroenterology, Tokai University School of Medicine Hachioji Hospital, Tokyo, Japan
Hidekazu Suzuki: Department of Gastroenterology, Tokai University School of Medicine, Kanagawa, Japan

DOI: https://doi.org/10.1159/000521538
Journal volume & issue: Vol. 16, no. 1
pp. 23 – 28

Abstract

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A 19-year-old man visited our hospital because of diarrhea and blood and liver dysfunction. Magnetic and endoscopic retrograde cholangiography revealed diffuse narrowing of the common biliary tract and intrahepatic biliary tract. A biopsy specimen from the narrowed area of the common biliary tract revealed IgG4-related sclerosing cholangitis (IgG4-SC). Colonoscopy showed multiple aphthoid colitis. He was treated with corticosteroids, and the levels of hepatobiliary enzymes, IgG, and IgG4 gradually decreased. This is an extremely rare case of colitis in which the patient had a phenotype of sclerosing cholangitis similar to IgG4-SC, which was finally diagnosed as primary sclerosing cholangitis.

Published in Case Reports in Gastroenterology

ISSN: 1662-0631 (Online)
Publisher: Karger Publishers
Country of publisher: Switzerland
LCC subjects: Medicine: Internal medicine: Specialties of internal medicine: Diseases of the digestive system. Gastroenterology
Website: http://www.karger.com/crg

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