Purpose: To describe the clinical course, treatment, and subsequent care of a full-term newborn with atypical, unilateral, recurrent cytomegalovirus chorioretinitis. Case report: The perinatal period of a full-term newborn girl was complicated by the development of petechiae on the skin. The initial laboratory findings were notable for thrombocytopenia, hyperbilirubinemia, and polycythemia. An ultrasound of the brain revealed cystic changes in the area of the lateral ventricles. MRI confirmed pseudocystic periventricular changes with periventricular calcifications. PCR testing confirmed CMV positivity, and the condition was diagnosed as congenital CMV infection requiring systemic treatment with ganciclovir. Indirect ophthalmoscopy revealed nonspecific lucency in the central area of the right eye, with several small hemorrhages on the retinal periphery. Although systemic therapy was initiated promptly, the original lesion reactivated multiple times on its periphery over the following months. The lesions were gradually healed, with pigmented scarring limiting the visual function of the affected eye. Conclusion: In contrast with the focal changes in the peripheral retina observed in adults, congenital CMV infection affecting children is characterized by macular involvement. The degree of visual impairment is directly proportional to the extent of reparative scarring affecting the central retina. From the perspective of prognosis and prevention of potential late retinal complications, screening and regular ophthalmological examinations are essential.