Poststreptococcal acute disseminated encephalomyelitis with basal ganglia involvement and auto-reactive antibasal ganglia antibodies

被引:116
作者
Dale, RC
Church, AJ
Cardoso, F
Goddard, E
Cox, TC
Chong, WK
Williams, A
Klein, NJ
Neville, BG
Thompson, EJ
Giovannoni, G
机构
[1] Inst Neurol, Dept Neurochem, Neuroimmunol Unit, London WC1N 3BG, England
[2] Great Ormond St Hosp Natl Hlth Serv Trust, Dept Neurol, London, England
[3] Great Ormond St Hosp Natl Hlth Serv Trust, Dept Microbiol, London, England
[4] Great Ormond St Hosp Natl Hlth Serv Trust, Dept Radiol, London, England
[5] Great Ormond St Hosp Natl Hlth Serv Trust, Dept Immunobiol, London, England
[6] Univ London, Inst Child Hlth, London WC1N 1EH, England
[7] Univ Fed Minas Gerais, Dept Psychiat & Neurol, Belo Horizonte, MG, Brazil
关键词
D O I
10.1002/ana.1250
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Antibasal ganglia antibodies (ABGA) are associated with Sydenham's chorea and pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections. We present 10 patients with acute disseminated encephalomyelitis (ADEM) associated with Group A beta hemolytic streptococcal infection. The clinical phenotype was novel, with 50% having a dystonic extrapyramidal movement disorder, and 70% a behavioral syndrome. None of the patients had rheumatic fever or Sydenham's chorea. Enzyme-linked immunosorbent assay, Western immunoblotting, and immunohistochemistry were used to detect ABGA. Neurological (n = 40) and streptococcal (n = 40) controls were used for comparison. Enzyme-linked immunosorbent assay results showed significantly elevated ABGA in the patients with poststreptococcal ADEM. Western immunoblotting demonstrated ABGA reactivity to three dominant protein bands of 60, 67, or 80 kDa; a finding not reproduced in controls. Fluorescent immunohistochemistry demonstrated specific binding to large striatal neurones, which was not seen in controls. Streptococcal serology was also significantly elevated in the poststreptococcal ADEM group compared with neurological controls. Magnetic resonance imaging studies showed hyperintense basal ganglia in 80% of patients with poststreptococcal ADEM, compared to 18% of patients with non-streptococcal ADEM. These findings support a new subgroup of postinfectious autoimmune inflammatory disorders associated with Group A beta hemolytic streptococcus, abnormal basal ganglia imaging, and elevated ABGA.
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页码:588 / 595
页数:8
相关论文
共 21 条
[11]   ANTIBODIES REACTING WITH CYTOPLASM OF SUBTHALAMIC AND CAUDATE NUCLEI NEURONS IN CHOREA AND ACUTE RHEUMATIC-FEVER [J].
HUSBY, G ;
VANDERIJN, I ;
ZABRISKIE, JB ;
ABDIN, ZH ;
WILLIAMS, RC .
JOURNAL OF EXPERIMENTAL MEDICINE, 1976, 144 (04) :1094-1110
[12]   ACUTE DISSEMINATED ENCEPHALOMYELITIS - MRI FINDINGS AND THE DISTINCTION FROM MULTIPLE-SCLEROSIS [J].
KESSELRING, J ;
MILLER, DH ;
ROBB, SA ;
KENDALL, BE ;
MOSELEY, IF ;
KINGSLEY, D ;
DUBOULAY, EPGH ;
MCDONALD, WI .
BRAIN, 1990, 113 :291-302
[13]  
KIESSLING LS, 1993, PEDIATRICS, V92, P39
[14]   The psychiatric symptoms of rheumatic fever [J].
Mercadante, MT ;
Busatto, GF ;
Lombroso, PJ ;
Prado, L ;
Rossário-Campos, MC ;
do Valle, R ;
Marques-Dias, MJ ;
Kiss, MH ;
Leckman, JF ;
Miguel, EC .
AMERICAN JOURNAL OF PSYCHIATRY, 2000, 157 (12) :2036-2038
[15]  
MILLER HG, 1956, Q J MED, V25, P427
[16]   ACUTE ENCEPHALOMYELITIS - EXTENDING THE NEUROLOGICAL MANIFESTATIONS OF ACUTE RHEUMATIC-FEVER [J].
MUNN, R ;
FARRELL, K ;
CIMOLAI, N .
NEUROPEDIATRICS, 1992, 23 (04) :196-198
[17]   Therapeutic plasma exchange and intravenous immunoglobulin for obsessive-compulsive disorder and tic disorders in childhood [J].
Perlmutter, SJ ;
Leitman, SF ;
Garvey, MA ;
Hamburger, S ;
Feldman, E ;
Leonard, HL ;
Swedo, SE .
LANCET, 1999, 354 (9185) :1153-1158
[18]   DEMYELINATING ENCEPHALOPATHY IN LYME DISEASE [J].
REIK, L ;
SMITH, L ;
KHAN, A ;
NELSON, W .
NEUROLOGY, 1985, 35 (02) :267-269
[19]  
Segawa M, 2000, BRAIN DEV-JPN, V22, pS1
[20]   Antibodies against human putamen in children with Tourette syndrome [J].
Singer, HS ;
Giuliano, JD ;
Hansen, BH ;
Hallett, JJ ;
Laurino, JP ;
Benson, M ;
Kiessling, LS .
NEUROLOGY, 1998, 50 (06) :1618-1624