A 38-year-old man with human immunodeficiency virus was referred for evaluation of retinal lesions in both eyes. Optical coherence tomography was performed after dilating the pupils. Biomicroscopy of the retina showed an atypical, solitary, yellowish-white lesion in the macula of both eyes with no inflammation of the vitreous. Optical coherence tomography of the lesions showed an area of extremely low reflectivity with well-defined but irregular borders in the outer retina. The surrounding retina showed normal reflectivity and was of normal thickness. Optical coherence tomography showed selective necrosis of the outer layers due to progressive outer retinal necrosis. Optical coherence tomography may serve as a useful tool for the early diagnosis of progressive outer retinal necrosis.