A case report of cytomegalovirus retinitis in a patient who underwent orthotopic liver transplantation, and suffered from a prior episode of systemic cytomegalovirus disease, is described. Although the diagnosis was obtained only when clinical symptoms prompted ophthalmoscopic evaluation, a successful outcome was attained after ganciclovir treatment. The role of clinical and virologic monitoring of organ transplant recipients, and that of primary and secondary chemoprophylaxis against cytomegalovirus infection are discussed, according to personal observations and to an updated literature review.