Imatinib mesylate (STI 571; Gleevec; Novartis Pharmaceuticals, Basel, 
Switzerland) is an orally available tyrosine kinase inhibitor that targets a 
constitutively activated BCR-ABL tyrosine kinase with additional inhibitory 
effects on platelet derived growth factor (PDGF) receptors alpha and beta, and 
KIT. It has revolutionized the treatment of adult and pediatric patients with 
Philadelphia chromosome positive chronic myelogenous leukemia (CML) and is also 
FDA-approved for KIT-positive advanced gastrointestinal tumor (GIST) and 
dermatofibrosarcoma protuberans. A wide spectrum of dermatologic toxicities has 
been associated with this agent, among which a maculopapular rash is the most 
common event. In addition, a variety of pigmentary abnormalities of the skin and 
mucosal surfaces have been reported. Hypopigmentation is a well-recognized 
adverse effect. In contrast, paradoxical hyperpigmentation has only rarely been 
documented. In this case report we describe imatinib-induced cutaneous 
hyperpigmentation and graying of hair occurring in the same patient with 
dermatofibrosarcoma protuberans treated with imatinib.