SUMMARY: Cytomegalovirus (CMV) retinitis is typically diagnosed in patient with AIDS and those who underwent allogeneic hematopoietic cell transplant. However, it may develop in patients with acute lymphoblastic leukemia (ALL) who have not undergone hematopoietic cell transplantation. To increase awareness of CMV retinitis in this group, we describe 3 patients ages 3, 9, and 12, with ALL who developed CMV retinitis. The diagnosis of CMV retinitis was made on the basis of ophthalmological findings suggesting typical retinal lesions. In 2 cases, CMV DNAemia was present, while in 1 patient CMV DNA was detected only in vitreous fluid using the PCR technique. All cases were treated with intravenous ganciclovir for 2 or 3 weeks as induction therapy, followed by oral valganciclovir prophylaxis. Initially, active retinitis lesions resolved in all cases; however, in 1 patient CMV retinitis relapsed 3 times during follow-up. In this case, by using foscarnet therapy, satisfactory responses were achieved and the progression of CMV retinitis lesions stopped and eventually regressed.Cytomegalovirus (CMV) may cause asymptomatic infection or self-limiting illness in most infected, immunocompetent individuals. In patients with reduced cellular immunity, such as patients infected with human immunodeficiency virus, solid organ transplant recipients, hematopoietic cell transplant (HCT) recipients, and those receiving immune suppression therapy, it may lead to end-organ diseases (1), one of which is CMV retinitis. The majority of CMV retinitis cases are related to the hematogenous spread of the virus to the retina, usually after systemic reactivation of a latent infection, and are usually diagnosed by an experienced ophthalmologist on the basis of typical retinal changes. To the best of our knowledge, CMV retinitis in patients with acute lymphoblastic leukemia (ALL) without autologous or allogeneic HCT, has rarely been reported. This report, in the light of the published literature, describes 3 pediatric patients with ALL who developed CMV retinitis while receiving chemotherapy.The 1st case is a 12-year-old boy with ALL who was admitted to the hospital with blurred vision in the right eye (RE) (2). The patient was in the maintenance phase of chemotherapy on the ALL-BFM-2003 protocol and was receiving oral methotrexate and mercaptopurine. His white blood cell (WBC) count was 3,200/mL and his neutrophil count was 1,600/mL. The ophthalmological examination revealed a best corrected visual acuity (BCVA) of 20/20 in both the RE and the left eye (LE). The slit lamp examination of the anterior chamber of the LE was normal, whereas a 3+ anterior chamber cellular reaction was observed in the RE. The retinal examination revealed active retinitis lesions (creamcolored lesions associated with hemorrhages) and perivascular cuffing in the retinal periphery in the RE (Fig. 1A). The LE retina was normal. Laboratory studies revealed positive CMV IgM, but negative CMV IgG,