Clinical features and long-term outcome of a group of Japanese children with inflammatory central nervous system disorders and seropositivity to myelin-oligodendrocyte glycoprotein antibodies

• DOI: 10.1016/j.braindev.2015.02.006
• Publication Date: 2015-03-03T11:07:47Z
• Authors: Naomi Hino-Fukuyo; Kazuhiro Haginoya; Ichiro Nakashima; Douglas Kazutoshi...; Toshiyuki Takahashi; Tatsuro Misu; Kazuo Fujihara; Mieko Hirose; Yosuke Kakisaka; Mitsugu Uematsu; Tomoko Kobayashi; Shigeo Kure
• Keywords: Myelin oligodendrocyte glycoprotein; Neuromyelitis Optica; Acute disseminated encephalomyelitis; Optic neuritis; Demyelinating Disorder

Myelin-oligodendrocyte glycoprotein and aquaporin-4 have been extensively analyzed as targets for humoral immune reactions in central nervous system (CNS) demyelinating diseases, and the results indicated a possible role of these antibodies in the pathogenesis of various demyelinating diseases.To investigate the antibody titer levels against myelin-oligodendrocyte glycoprotein and aquaporin-4 in pediatric patients with inflammatory CNS disorders, and to evaluate clinical significance to study anti-myelin-oligodendrocyte glycoprotein antibodies.Sera at onset from patients with acute disseminated encephalomyelitis (ADEM) in 7, optic neuritis (ON) in 5, pediatric MS in 4 and neuromyelitis optica in one were tested for myelin-oligodendrocyte glycoprotein and aquaporin-4 antibodies using cell-based assays with live transfected cells. The duration of the observation periods ranged from 1 to 21 years (median, 10 years). We also described clinical course of patients with positive anti-myelin-oligodendrocyte glycoprotein antibodies.Among 17 patients diagnosed with inflammatory CNS demyelinating diseases nine (52%) were positive to anti-myelin-oligodendrocyte glycoprotein antibodies. Of note, all cases with positive anti-myelin-oligodendrocyte glycoprotein antibodies showed seronegativity against anti-aquaporin-4 antibodies and had a favorable prognosis.This preliminary report showed that anti-myelin-oligodendrocyte glycoprotein antibodies testing at onset could be a useful tool predicting clinical outcome of children with ADEM, ON, and MS.