PurposeTo report spectral-domain optical coherence tomography (SD-OCT) findings of presumed Cryptococcus neoformans infection limited to the retina.MethodsWe report a 39-year-old male with decreased vision for 3 months. Clinical examination revealed multiple cream-colored retinal lesions in the posterior pole of both eyes. SD-OCT demonstrated multiple areas of discrete, hyperreflective deposits in the inner retina, outer retina, and subretinal space without evidence of choroidal involvement. Fundus autofluorescence demonstrated areas of hyperautofluorescence of lesions with variable areas of hypoautofluorescence.ResultsSubsequent laboratory workup revealed systemic Cryptococcus neoformans infection. After 3 months of systemic antifungal treatment and follow-up, there was clinical improvement in the size of the lesions.ConclusionsThis is the first report of SD-OCT demonstrating presumed localized Cryptococcus infection confined to the retina. Our findings support the assertion that Cryptococcus can cause a focal retinitis involving all layers of the retina without demonstrable evidence of choroidal involvement.