We report a case study of a patient with cholestatic hepatitis undergoing treatment with oral ticlopidine. The patient was admitted to our hospital for a transient ischemic attack, and after 40 days of ticlopidine therapy developed cholestatic jaundice, fever, itching, and arthralgias. Results from an ultrasound, computed tomography scan, and endoscopic retrograde cholangiopancreatography eliminated the presence of biliary obstruction; results from a liver biopsy showed a histopathologic picture consistent with drug-induced cholestatic hepatitis. One year after discontinuation of ticlopidine, the patient was still asymptomatic, but laboratory data showed relevant abnormalities, indicating persistence of cholestasis and hepatic injury. This is the first reported instance of a patient with cholestatic hepatitis that developed during treatment with ticlopidine and persisted despite interruption of drug administration.