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Dopa-responsive acute disseminated encephalomyelitis with marked atrophy of the striate body

A 16-year-old boy was admitted to our hospital, with a 4-day history of hyperthermia, vomiting, confusion, nuchal rigidity and dysbasia. Cerebrospinal fluid (CSF) examination showed: white cell count, 673/μL (69% polymorphonuclear cells); protein, 305 mg/dL; glucose ratio of CSF to serum, 0.35; and negative results for culture, antiviral antibodies, Epstein-Barr virus DNA and oligoclonal IgG bands (OB). Antibiotics and acyclovir were given intravenously under a tentative diagnosis of infectious meningitis. However, unconsciousness was prolonged. CSF myelin basic protein (MBP) and IgG index were abnormally elevated to 257 pg/mL (normal, <102 pg/mL) and 1.52, respectively. Cerebral white matter showed multiple high-intensity lesions on fluid-attenuated inversion recovery (FLAIR) imaging, and bilateral striate bodies showed signal hyperintensity with swelling on T2-weighted imaging (figure 1A). We finally diagnosed acute disseminated encephalomyelitis (ADEM). Two courses of high-dose intravenous methylprednisolone, intravenous immunoglobulin and oral prednisolone therapy improved CSF findings within 2 weeks. However, extrapyramidal symptoms, including mask-like facies, rigidity of the left extremities, neck and trunk, and myoclonus, developed in addition to the pre-existing prolonged bedridden and mute state at 3 weeks after admission, and lasted for another 2 months. Atrophy …