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Chronic myeloid leukemia (CML) rarely presents under 15 years of age, and its incidence is as low as 2% of all leukemia. Ophthalmic manifestations often present either initially with the diagnosis or as a sign of relapse after remission. The recommended treatment is systemic chemotherapy, intrathecal chemotherapy, ocular irradiation, and/or intravitreal methotrexate. Herein, we report a 13-year-old child with a history of CML and central nervous system relapse under remission currently. He presented to us with bilateral ophthalmic relapse. He was treated with ocular irradiation and intrathecal chemotherapy. On follow-up, the ophthalmic infiltration showed resolution, which was also seen on multimodal imaging.