Objective To observe the clinical characteristics and treatment of cytomegalovirus retinitis (CMVR) in leukemia patients. Methods This is a retrospective analysis. Seven leukemia patients (13 eyes) with CMVR were studied. All patients underwent examinations of visual acuity, slit lamp microscope, ophthalmoscope, color fundus photography, peripheral blood CD4+T cell count and serum/aqueous CMV-DNA test. All patients were treated with ganciclovir or zoledronic acid combined with intravitreal injection of ganciclovir. The follow-up period was 3-14 months. Results Six patients were treated with hematopoietic stem cell transplantation and 1 patient was with chronic leukemia. All patients were CMV-DNA positive for serum, and 18.5% (2/7) for aqueous humor. CMVR in leukemia patients showed mild anterior segment inflammation, ocular fundus with irregular yellowish-white retinal necrosis and radial hemorrhage (7 eyes). Some (2 eyes) also shoed gray and white granular retinal infiltrates. Intravenous ganciclovir/zoledronic acid combined with intravitreal injection of high concentration ganciclovir was an effective treatment, while systemic corticosteroids were effective in reducing vitreous opacity. Conclusions CMVR is characterized by progressive necrotic retinitis with hemorrhage and vasculitis. Intravenous ganciclovir/zoledronic acid combined with intravitreal injection of ganciclovir is effective in the treatment of CMVR with leukemia.