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Abstract: We report on a boy with bilateral optic nerve hypoplasia and mild psychomotor retardation. At 1 year and 9 months of age, he was admitted to hospital with a cluster of febrile convulsions and unconsciousness. Magnetic resonance imaging (MRI) revealed widespread areas of high signal intensity on diffusion-weighted imaging of the deep cerebral white matter and corpus callosum. This imaging disappeared at five days of illness. No atrophy or abnormalities were noted on the 6-month follow-up MRI. Despite full recovery after the acute episode, the patient showed retarded developmental progress. We discuss the differential diagnosis for this case. [Copyright &y& Elsevier]