Sudden neurologic death after intrathecal methotrexate

Methotrexate leukoencephalopathy is a chronic syndrome of ataxia and confusion which may progress to seizures, coma, and death. We report a fatal case of this syndrome in a patient who displayed no evidence of the typical prodrome of neurologic symptoms or signs. This patient suffered brain death after receiving 11 doses of intrathecal methotrexate for leukemic meningitis. Since leukoencephalopathy was not clinically suspected, this case underscores the need for a test that would reliably monitor central nervous system toxicity due to intrathecal therapy.