PurposeTo report a case of bilateral cytomegalovirus (CMV) retinitis in an otherwise healthy infant.MethodsA four-month-old, healthy, male infant was evaluated for visual inattention.ResultsThis full-term infant with a normal birth weight and an uneventful gestational period was referred with symptoms of visual inattention, fever, and agitation one week prior to admission. Ocular involvements were detected in the form of bilateral pan uveitis with diffuse bilateral retinitis and vasculitis with hemorrhage in the peripheral retina and posterior pole. CMV DNA was detected in the patient's ocular sample and cerebrospinal fluid by polymerase chain reaction (PCR). He was treated with intravitreal and systemic ganciclovir. Unfortunately, the infant died because of CMV encephalitis. Therefore, bilateral CMV retinitis (CMVR), which was probably transmitted from the mother, was diagnosed in this immunocompetent infant.ConclusionsThe present case highlights the possibility of CMVR in immunocompetent infant associated with systemic CMV infection, even during the postnatal period. Therefore, a high index of clinical suspicion and prompt treatment may be life-saving in similar cases.