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

Jin Imai1,2, Hitoshi Ichikawa2,3, Motoki Kaneko1,2

  • 1Department of Gastroenterology, Tokai University School of Medicine, Kanagawa, Japan.

Case Reports in Gastroenterology
|March 30, 2022
PubMed

Insights

This rare case highlights a 19-year-old man with IgG4-related sclerosing cholangitis (IgG4-SC) and colitis. Treatment with corticosteroids improved liver enzymes and IgG4 levels, suggesting a link between these conditions.

Area of Science:

  • Gastroenterology
  • Immunology
  • Hepatology

Background:

  • Immunoglobulin G4-related sclerosing cholangitis (IgG4-SC) is a fibroinflammatory condition.
  • Colitis can present with diverse gastrointestinal symptoms.

Observation:

  • A 19-year-old male presented with diarrhea, hematochezia, and liver dysfunction.
  • Biliary imaging showed diffuse narrowing of the common and intrahepatic bile ducts.
  • Colonoscopy revealed multiple aphthoid colitis.

Findings:

  • Biopsy of the common bile duct confirmed IgG4-related sclerosing cholangitis (IgG4-SC).
  • Corticosteroid therapy led to decreased hepatobiliary enzymes, IgG, and IgG4 levels.
  • The patient's presentation included features of both IgG4-SC and colitis.

Implications:

  • This case suggests a potential association between IgG4-SC and colitis.
  • Further research is needed to understand the pathogenesis of IgG4-SC presenting with colitis.
  • Corticosteroids may be effective in managing this rare combined condition.

Keywords:
Aphtoid colitisIgG4-related sclerosing cholangitisPrimary sclerosing cholangitis

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