Chronic myeloid leukemia is a myeloproliferative disorder. Imatinib mesylate (Gleevec®) is a first line pharmacologic treatment for all phases of chronic myeloid leukemia and for advanced gastrointestinal stromal tumors (GISTs). Imatinib mesylate is generally well tolerated. Well-known ocular side effects for Imatinib are periorbital edema, epiphora, conjunctival hemorrhage, blepharoconjunctivitis, visus alterations and ocular dryness and also optic disc edema is the rare ocular event associated with Imatinib use. Here we reported a case of 58-year-old Caucasian male was diagnosed with Chronic Myeloid Leukemia (CML) and he is treating with Imatinib for 7 years. Patient was presented with moderate periorbital edema along with optic disc edema and these ocular events can be self reducible in our case after suspension of Imatinib for 1 week without any treatment. Prompt consultation with an ophthalmologist can lead to early detection, proper diagnosis and appropriate therapeutic measures. Keywords: Chronic myeloid leukemia, Imatinib mesylate, optic disc edema, periorbital edema.