To report the unusual case of retinal leukemic infiltration in a child with adult-type chronic myelocytic leukemia. Interventional case report. Complete ophthalmologic examination, including visual acuity measurement and funduscopy. The diagnosis was established by blood cell counts and confirmed by a positive Philadelphia-chromosome translocation (t [9,22]). Treatment consisted of hydroxyurea and stem-cell transplantation. After therapy, complete regression of the retinal lesion was observed with full recovery of visual acuity. This case underlines the importance of interdisciplinary cooperation between ophthalmologists and pediatric oncologists in the diagnosis and treatment of this disease.