Tourette's syndrome: a cross sectional study to examine the PANDAS hypothesis
Tourette's syndrome: a cross sectional study to examine the PANDAS hypothesis
复制标题
DOI：
10.1136/jnnp.74.5.602
复制
发表时间：
2003-05-01
影响因子：
11
通讯作者：
Robertson, MM
中科院分区：
文献类型：
--
作者：
Church, AJ;Dale, RC;Robertson, MM
背景：A组β溶血性链球菌感染后的典型神经系统疾病是西德纳姆舞蹈病。最近描述了A组链球菌感染后发生的抽动障碍，并将其称为PANDAS（与链球菌感染相关的儿科自身免疫性神经精神障碍）。有人建议，A组链球菌感染后诱导的抗体反应与基底节神经元在Sydenham舞蹈病和PANDAS。抗基底神经节抗体（ABGA）是目前在大多数情况下，急性Sydenham的舞蹈病，但很少在controls.Objective：探讨的假设，Tourette综合征可能与A组链球菌感染和ABGA。神经系统疾病的儿童（n = 50）和近期无并发症的链球菌感染（n = 40），成人神经系统疾病（n = 50），和健康的成年人（n = 50）作为对照研究。近期A组链球菌感染定义使用抗链球菌溶血素O滴度（ASOT）。结果：64%的Tourette综合征患儿ASOT升高，与15%的儿童神经系统疾病对照组比较，差异有显著性（p < 0.0001）; 68%的成人Tourette综合征患儿ASOT升高，与12%的成人神经系统疾病对照组和8%的成人健康对照组比较，差异有显著性（p < 0.05）。Western免疫印迹显示，与对照组的2-4%相比，20%的抽动秽语综合征儿童和27%的成人中有阳性结合（p < 0.05）。最常见的基底神经节结合是60 kDa的抗原，类似于Sydenham舞蹈病中提出的抗原。间接免疫荧光显示自身抗体结合基底节神经元。在ABGA阳性的Tourette综合征患者中，91%（21/23）的近期A组链球菌感染的血清学证据被检出，而ABGA阴性的患者中，57%（44/77）的近期A组链球菌感染的血清学证据被检出（p < 0.01）。结果支持A组链球菌感染和基底节自身免疫在抽动秽语综合征患者亚组中的作用，提示西德纳姆舞蹈病和某些抽动秽语综合征患者之间的致病相似性。
Background: The classical neurological disorder after group A beta haemolytic streptococcal infection is Sydenham's chorea. Recently a tic disorder occurring after group A streptococcal infection has been described and termed PANDAS ( paediatric autoimmune neuropsychiatric disorders associated with streptococcal infection). It is proposed that antibodies induced after group A streptococcal infection react with basal ganglia neurones in Sydenham's chorea and PANDAS. Anti-basal ganglia antibodies (ABGA) are present in most cases of acute Sydenham's chorea, but rarely in controls.Objective: To investigate the hypothesis that Tourette's syndrome may be associated with group A streptococcal infection and ABGA.Methods: 100 patients with Tourette's syndrome (DSM-IV-TR) were enrolled in a cross sectional study. Children with neurological disease (n = 50) and recent uncomplicated streptococcal infection ( n = 40), adults with neurological disease ( n = 50), and healthy adults ( n = 50) were studied as controls. Recent group A streptococcal infection was defined using antistreptolysin O titre (ASOT). ABGA were detected using western immunoblotting and indirect immunofluorescence.Results: ASOT was raised in 64% of children with Tourette's syndrome compared with 15% of paediatric neurological disease controls (p < 0.0001), and in 68% of adults with Tourette's syndrome compared with 12% of adult neurological controls and 8% of adult healthy controls (p < 0.05). Western immunoblotting showed positive binding in 20% of children and 27% of adults with Tourette's syndrome, compared with 2-4% of control groups ( p < 0.05). The most common basal ganglia binding was to a 60 kDa antigen, similar to the proposed antigen in Sydenham's chorea. Indirect immunofluorescence revealed autoantibody binding to basal ganglia neurones. Serological evidence of recent group A streptococcal infection, assessed by a raised ASOT, was detected in 91% (21/23) of Tourette's syndrome patients with positive ABGA compared with 57% (44/77) with negative ABGA (p < 0.01).Conclusions: The results support a role of group A streptococcal infection and basal ganglia autoimmunity in a subgroup of patients with Tourette's syndrome and suggest a pathogenic similarity between Sydenham's chorea and some patients with Tourette's syndrome.