Clinicopathologic findings in a case of bilateral intraocular cryptococcosis are reported. Involvement of the retina and the leptomeningeal sheaths of the optic nerves was associated with a disseminated cryptococcal infection that affected the brain and meninges, suggesting that extension to intraocular structures from the central nervous system was by way of the leptomeningeal sheaths of the optic nerves. This is the second histopathologically proven case of cryptococcal neuroretinitis to be recorded, and it is one of 13 proven cases of intraocular cryptococcosis. Leukokoria in the absence of lens changes was an unusual finding. This patient also had systemic lupus erythematosus and had been on prolonged systemic corticosteroid therapy.
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