Immune checkpoint inhibitor cholangitis (IMC) due to its rarity poses difficulties in diagnosis and treatment. IMC includes a range of biliary tract injuries with different clinical and pathological characteristics, from small-duct to large-duct involvement. A 32-year-old man presented hospital with presyncope, nausea, and vomiting. Upon computed tomography, he was found to have multiple cryptogenic liver lesions. He had a history of lung adenocarcinoma on maintenance Keytruda. He had multiple admissions related to Keytruda complications which included pancreatitis requiring high-dose steroids, esophagitis, and gastritis (last esophagogastroduodenoscopy showing Severe hemorrhagic gastritis, gastric stenosis). A magnetic resonance cholangiopancreatography was obtained for cholestatic elevation of transaminases and showed intrahepatic and extrahepatic biliary dilatation with periductal enhancement. A liver biopsy was inconclusive. However, the findings could be associated with obstructive changes. The likely differentials were primary versus secondary sclerosing cholangitis. In the setting of prolonged use of pembrolizumab for 1.5 years and taking into consideration the timeline of symptoms, secondary sclerosing cholangitis was diagnosed. He was treated with steroids and Keytruda was discontinued with improvement in symptoms. Immune checkpoint inhibitors (ICI) can affect any organ system, including the liver, causing cholangitis, although this is less common than immune-mediated hepatitis. Steroids alone or with immunosuppression show similar results.
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