A fatal case of meningoencephalitis caused by Scedosporium inflatum (Scedosporium prolificans) in a 5-year-old boy with acute myeloblastic leukemia who was given intrathecal treatment is reported. Itraconazole treatment was ineffective. The fungus was identified on brain sections at autopsy and was not observed in any other organ. As no other portal of entry was detected, meningoencephalitis may have originated via direct introduction of the fungus at therapeutic lumbar puncture.