We describe a case of severe cholestatic hepatitis following administration of ticlopidine. A 57-year-old man without known liver disease developed jaundice approximately 3 weeks after initiation of ticlopidine for secondary prevention of stroke. Hyperbilirubinemia and abnormal liver function test values resolved 5 months after withdrawal of ticlopidine. The diagnosis of ticlopidine-induced cholestasis was made after thorough investigations had excluded other causes of jaundice. He was not retreated with ticlopidine. This case may serve to illustrate the possibility of ticlopidine hepatotoxicity, which has rarely been reported. Furthermore, to the best of our knowledge, ticlopidine-induced cholestatic hepatitis accompanied by autoantibody has not been previously reported. This case suggests that regular assessment of liver function should be performed in the initial 3 months of ticlopidine treatment due to the potential risk of adverse effects. In patients with abnormal biochemical test results, autoantibodies should be assessed.