Imatinib (Gleevec, Novartis Pharmaceuticals Corp, East Hanover, NJ) is widely used in the treatment of chronic myelogenous leukemia and gastrointestinal stromal tumors. To our knowledge, only one case report of histologically proven Imatinib-induced hepatotoxicity has been reported. We describe another case of hepatotoxicity in a 22-year-old woman including the histopathologic changes and the clinical course after the discontinuation of Imatinib.