A 51 year old patient who worked in Africa for eight years, presented twelv
e years later a progressive ataxia associated with headaches. Neuroimaging
studies done after a partial complex seizure demonstrated multiple supra an
d sub-tentorial cortical ring enhancing lesions. Histoplasma capsulatum his
toplasmosis was found on histological examination of brain biopsy and confi
rmed by isolation of the fungus. Medical treatment with intravenous amphote
ricin B followed by oral itraconazole (400 mg per day) improved both clinic
al and radiological status. This observation of cerebral histoplasmosis is
rather unusual for a seronegative HIV patient in a non endemic area.