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A 3-year-old child was incidentally found to have chronic myelogenous leukemia (CML) during an admission for a routine ophthalmic examination under anesthesia. The child had received systemic chemotherapy and focal treatment for Groups C and D retinoblastoma in the right and left eye, respectively, when she was 7 months old. CML was treated with dasatinib, and the child attained a major molecular response. The child is now 3 years after treatment of CML, and the retinoblastoma remains inactive. CML following treatment of retinoblastoma is a rare occurrence. Long term and close monitoring of retinoblastoma patients who received systemic chemotherapy using serial blood tests is essential.