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Journal of Nippon Medical School

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-Case Reports-

A Case of Autoimmune Hepatitis/Primary Biliary Cholangitis Overlap Syndrome during Treatment with Brodalumab for Generalized Pustular Psoriasis

Shizuka Okazaki1, Toshihiko Hoashi2, Hidehisa Saeki2 and Naoko Kanda1

1Department of Dermatology, Nippon Medical School Chiba Hokusoh Hospital, Chiba, Japan
2Department of Dermatology, Nippon Medical School Hospital, Tokyo, Japan


Psoriasis is a chronic inflammatory skin disease characterized by accelerated tumor necrosis factor-α (TNF-α)/interleukin (IL)-23/IL-17 axis, epidermal hyperproliferation, and dysregulated differentiation. Psoriasis is occasionally associated with autoimmune liver diseases such as autoimmune hepatitis (AIH) or primary biliary cholangitis (PBC), caused by autoimmunity against hepatocyte- or cholangiocyte-specific autoantigens, respectively. Overlap syndrome is a condition in which patients have features of both AIH and PBC. It has been reported that AIH, PBC, or the overlap syndrome can be triggered by certain drug therapies. A 65-year-old Japanese man developed increased serum levels of aspartate and alanine aminotransferases, and positive anti-nuclear and anti-mitochondrial M2 antibodies, along with neutropenia, at 4 weeks of treatment with an anti-IL-17 receptor A antibody brodalumab for generalized pustular psoriasis. Histological evaluation of the liver revealed interface hepatitis and non-suppurative destructive cholangitis, which is compatible with the overlap syndrome of AIH and PBC. This is the first case of AIH/PBC overlap syndrome during treatment with brodalumab for generalized pustular psoriasis. The relationship between brodalumab and AIH/PBC overlap syndrome should be further elucidated. The risk of autoimmune liver diseases in patients with psoriasis treated with brodalumab should be carefully considered.

J Nippon Med Sch 2021; 88: 569-573

Keywords
autoimmune hepatitis, primary biliary cholangitis, overlap syndrome, generalized pustular psoriasis, brodalumab

Correspondence to
Naoko Kanda, MD, PhD, Department of Dermatology, Nippon Medical School Chiba Hokusoh Hospital, 1715 Kamagari, Inzai, Chiba 270-1694, Japan
n-kanda@nms.ac.jp

Received, October 9, 2020
Accepted, December 7, 2020