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Vanishing Bile Duct Syndrome Caused by a Treatment Regimen Including Avacopan for Microscopic Polyangiitis

Mako Hishinuma1, Midori Hasegawa2, Shun Minatoguchi1

  • 1Department of Nephrology, Fujita Health University School of Medicine, Japan.

Internal Medicine (Tokyo, Japan)
|September 10, 2025
PubMed

Insights

Avacopan treatment for microscopic polyangiitis led to vanishing bile duct syndrome and liver injury, despite successful vasculitis remission. The patient ultimately died from progressive liver damage.

Area of Science:

  • Hepatology
  • Immunology
  • Nephrology

Background:

  • Microscopic polyangiitis is a severe autoimmune vasculitis.
  • Avacopan is a novel treatment for ANCA-associated vasculitis.
  • Drug-induced liver injury is a significant clinical concern.

Purpose of the Study:

  • To report a case of vanishing bile duct syndrome associated with avacopan treatment.
  • To investigate the potential hepatotoxicity of avacopan in a patient with microscopic polyangiitis.
  • To highlight the challenges in managing drug-induced liver injury in elderly patients.

Main Methods:

  • Case report of an 81-year-old male patient.
  • Treatment with prednisolone, avacopan, and rituximab for microscopic polyangiitis.
  • Monitoring of liver enzymes, liver biopsy, and autopsy findings.

Main Results:

  • Elevated liver enzymes were observed 42 days after initiating avacopan.
  • Liver biopsy confirmed vanishing bile duct syndrome.
  • Autopsy revealed persistent bile ductopenia, liver cell injury, cholestasis, and fibrosis.

Conclusions:

  • Avacopan may be associated with vanishing bile duct syndrome and severe liver injury.
  • Early recognition and discontinuation of the offending agent are crucial.
  • Further research is needed to understand the mechanisms and risk factors for avacopan-induced liver injury.
Keywords:
avacopanliver injurymicroscopy polyangiitis

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