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Peer-reviewedCC BY 4.0Frontiers in Immunology

Case Report: Cholangioscopic and histopathological findings before and after treatment in immune-related cholangitis

  1. 1Department of Gastroenterology, Faculty of Medical Sciences, University of Fukui
  2. 2Graduate School of Health and Human Life Sciences, Fukui Prefectural University

Abstract

Immune-related cholangitis is a rare immune-related adverse event, and its diagnostic criteria and treatment strategies have not yet been established. Here, we report a case of immune-related cholangitis in which treatment response could be objectively visualized through reassessment of cholangioscopic and histopathological findings before and after treatment. A 79-year-old man with recurrent cutaneous squamous cell carcinoma of the scalp developed high fever and general fatigue after six courses of nivolumab, and laboratory tests revealed marked elevation of hepatobiliary enzymes. Contrast-enhanced computed tomography and endoscopic ultrasonography showed bile duct wall thickening, endoscopic retrograde cholangiopancreatography demonstrated shaggy irregularity in the distal bile duct, and peroral cholangioscopy revealed diffuse surface irregularity and granular changes of the biliary mucosa. Furthermore, cholangioscopy-guided bile duct biopsy demonstrated marked lymphocytic infiltration, particularly infiltration of CD8-positive T cells, leading to a diagnosis of immune-related cholangitis. Prednisolone and ursodeoxycholic acid were initiated, with nivolumab discontinued, after which the clinical symptoms and hepatobiliary enzyme levels gradually improved. Furthermore, on Day 28 after treatment initiation, reassessment by cholangioscopy and bile duct biopsy confirmed improvement in the cholangioscopic findings and reduction of lymphocytic infiltration, particularly CD8-positive T-cell infiltration, on bile duct biopsy. These objective findings supported tapering of steroid therapy. To our knowledge, very few cases have described reassessment of cholangioscopic and histopathological findings before and after treatment in immune-related cholangitis. We suggest that such evaluation may contribute to confirming treatment response and guiding therapeutic strategy in immune-related cholangitis.

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BibTeX
@article{akazawa2026case,
  title = {{Case Report: Cholangioscopic and histopathological findings before and after treatment in immune-related cholangitis}},
  author = {Yu Akazawa and Takuto Nosaka and Shouichi Namikawa and Arisa Saito and Yosuke Murata and Tomoko Tanaka and Kazuto Takahashi and Tatsushi Naito and Masahiro Ohtani and Yasunari Nakamoto},
  journal = {Frontiers in Immunology},
  year = {2026},
  month = oct,
  doi = {10.3389/fimmu.2026.1934941},
  url = {https://www.frontiersin.org/articles/10.3389/fimmu.2026.1934941/full},
  note = {CC BY 4.0},
}
RIS
TY  - JOUR
TI  - Case Report: Cholangioscopic and histopathological findings before and after treatment in immune-related cholangitis
AU  - Yu Akazawa
AU  - Takuto Nosaka
AU  - Shouichi Namikawa
AU  - Arisa Saito
AU  - Yosuke Murata
AU  - Tomoko Tanaka
AU  - Kazuto Takahashi
AU  - Tatsushi Naito
AU  - Masahiro Ohtani
AU  - Yasunari Nakamoto
PY  - 2026
DA  - 2026/10/05
JO  - Frontiers in Immunology
DO  - 10.3389/fimmu.2026.1934941
UR  - https://www.frontiersin.org/articles/10.3389/fimmu.2026.1934941/full
AB  - Immune-related cholangitis is a rare immune-related adverse event, and its diagnostic criteria and treatment strategies have not yet been established. Here, we report a case of immune-related cholangitis in which treatment response could be objectively visualized through reassessment of cholangioscopic and histopathological findings before and after treatment. A 79-year-old man with recurrent cutaneous squamous cell carcinoma of the scalp developed high fever and general fatigue after six courses of nivolumab, and laboratory tests revealed marked elevation of hepatobiliary enzymes. Contrast-enhanced computed tomography and endoscopic ultrasonography showed bile duct wall thickening, endoscopic retrograde cholangiopancreatography demonstrated shaggy irregularity in the distal bile duct, and peroral cholangioscopy revealed diffuse surface irregularity and granular changes of the biliary mucosa. Furthermore, cholangioscopy-guided bile duct biopsy demonstrated marked lymphocytic infiltration, particularly infiltration of CD8-positive T cells, leading to a diagnosis of immune-related cholangitis. Prednisolone and ursodeoxycholic acid were initiated, with nivolumab discontinued, after which the clinical symptoms and hepatobiliary enzyme levels gradually improved. Furthermore, on Day 28 after treatment initiation, reassessment by cholangioscopy and bile duct biopsy confirmed improvement in the cholangioscopic findings and reduction of lymphocytic infiltration, particularly CD8-positive T-cell infiltration, on bile duct biopsy. These objective findings supported tapering of steroid therapy. To our knowledge, very few cases have described reassessment of cholangioscopic and histopathological findings before and after treatment in immune-related cholangitis. We suggest that such evaluation may contribute to confirming treatment response and guiding therapeutic strategy in immune-related cholangitis.
SN  - 1664-3224
ER  -