SummaryThis rare encephalopathy that generally affects children is preceded by a respiratory infection and fever associated with convulsions and may progress to coma. Outcome is catastrophic in most cases. This case report describes a Brazilian child of African descent with fever, cephalea and bilateral amaurosis, who evolved to coma with pyramidal signs and associated convulsions. MRI showed diffuse, symmetrical lesions in the thalamotegmental region and brainstem. Following administration of methylprednisolone, the clinical condition of the patient improved and the brain lesions regressed, leaving the child with no current neurological defi cits. This was a case of acute postinfectious encephalopathy, involving various brain structures. Outcome was favourable with no sequelae following therapy. This case was atypical due to the bilateral visual involvement and extensive encephalic lesions in a child of African descent with no neurological sequelae following therapy. No other similar cases have been reported in the literature.