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Cytomegalovirus (CMV) disease is rare in children who receive anticancer chemotherapy and have no history of stem cell transplantation (SCT). We report a case of CMV retinitis that developed during maintenance chemotherapy for acute leukemia. A 7-year-old boy developed decreased visual acuity and persistent pancytopenia during maintenance chemotherapy. Laboratory investigations initially showed significant CMV antigenemia (51 positive cells/200,000 leukocytes); however, antiviral therapy wasdoi:10.14776/piv.2020.27.e19 fatcat:5gziir65mjfobpiyfg4dj7n4jm