TY - JOUR
T1 - 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
AU - Hino-Fukuyo, Naomi
AU - Haginoya, Kazuhiro
AU - Nakashima, Ichiro
AU - Sato, Douglas Kazutoshi
AU - Takahashi, Toshiyuki
AU - Misu, Tatsuro
AU - Fujihara, Kazuo
AU - Hirose, Mieko
AU - Kakisaka, Yosuke
AU - Uematsu, Mitsugu
AU - Kobayashi, Tomoko
AU - Kure, Shigeo
PY - 2015/10/1
Y1 - 2015/10/1
N2 - Background: 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. Objective: 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. Methods: 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. Results: 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. Conclusions: 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.
AB - Background: 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. Objective: 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. Methods: 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. Results: 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. Conclusions: 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.
KW - Acute disseminated encephalomyelitis
KW - Multiple sclerosis
KW - Myelin-oligodendrocyte glycoprotein
KW - Neuromyelitis optica
KW - Optic neuritis
UR - http://www.scopus.com/inward/record.url?scp=84938114162&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84938114162&partnerID=8YFLogxK
U2 - 10.1016/j.braindev.2015.02.006
DO - 10.1016/j.braindev.2015.02.006
M3 - Article
C2 - 25748628
AN - SCOPUS:84938114162
VL - 37
SP - 849
EP - 852
JO - Brain and Development
JF - Brain and Development
SN - 0387-7604
IS - 9
ER -