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Type 2 autoimmune pancreatitis (AIP) can present with unusual biliary issues, mimicking other serious conditions. This case highlights successful treatment of AIP-associated cholangiopathy with azathioprine when steroids failed.

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Area of Science:

  • Gastroenterology
  • Immunology

Background:

  • Autoimmune pancreatitis (AIP) is a rare chronic inflammatory condition with two distinct subtypes.
  • Type 1 AIP is associated with IgG4-related disease and can involve multiple organs, including the hepatobiliary system.
  • Type 2 AIP is typically confined to the pancreas.

Observation:

  • A patient presented with histopathologically confirmed Type 2 AIP.
  • Despite initial steroid response, the patient developed persistent cholestasis due to a left hepatic duct stricture.
  • Recurrence of pancreatitis and biliary stricture progression occurred upon steroid tapering, raising concerns for cholangiocarcinoma.

Findings:

  • The patient's cholangiopathy, a rare association with Type 2 AIP, did not resolve with steroids.
  • Investigations ruled out malignancy and primary sclerosing cholangitis.
  • Azathioprine treatment led to the resolution of both pancreatic and biliary manifestations.

Implications:

  • This case expands the known clinical spectrum of Type 2 AIP to include cholangiopathy.
  • It suggests azathioprine as a potential therapeutic option for steroid-refractory AIP-associated cholangiopathy.
  • Further research is warranted to understand the pathogenesis and optimal management of this AIP subtype and its biliary complications.