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Imatinib mesylate is a newly developed tyrosine kinase inhibitor approved as a unique agent for the therapy of oncological conditions like chronic myeloid leukemia and dermatofibrosarcoma protuberans. It is a specific and potent inhibitor of the BCR-ABL gene which is a protein kinase. The cutaneous side effects encountered include periorbital edema, maculopapular exanthems, cutaneous hypopigmentation, urticaria and sweet syndrome. We report a case of a 27-year-old male with generalized cutaneous hypopigmentation secondary to oral imatinib mesylate intake as a treatment for chronic myeloid leukemia.