2',3'-Dideoxyinosine (ddI), a purine analog with antiretroviral activity currently used to treat patients with AIDS (acquired immunodeficiency syndrome), is being used to treat both adults and children in clinical protocols at the National Institutes of Health. The purpose of this study is to follow prospectively patients treated with ddI for the development of ocular complications secondary to drug toxicity. Ninety- five children with symptomatic (CDC class P-2) HIV (human immune deficiency virus) infection were enrolled in a phase I/II study to assess the safety and antiretroviral activity of ddI. Five children developed peripheral atrophy of the retinal pigment epithelium during ddI therapy. The tow children with the most severe retinal atrophy were enrolled in the study at the highest dose level studied (540 mg/m2/day). Electro- oculograms were abnormal in one of three patients with retinal toxicity who could be tested. A group of 75 adults treated with ddI are being followed with periodic fundus examinations and electro-oculograms. During the past year similar retinal lesions were found in one adult patient treated with ddI.