Psychiatric and Neurodevelopmental Genetics Unit, Center for Human Genetics Research, Departments of Neurology and Psychiatry, Massachusetts General Hospital, Boston, Massachusetts, USA; Division of Cognitive and Behavioral Neurology, Department of Neurology, Brigham and Women's Hospital, Boston, Massachusetts, USA.
Mov Disord. 2015 Feb;30(2):221-8. doi: 10.1002/mds.26089. Epub 2014 Dec 8.
The aim of this study was to refine the population prevalence estimate of Tourette Syndrome (TS) in children and to investigate potential sources of heterogeneity in previously published studies. A systematic review was conducted and all qualifying published studies of TS prevalence were examined. Extracted data were subjected to a random-effects meta-analysis weighted by sample size; meta-regressions were performed to examine covariates that have previously been proposed as potential sources of heterogeneity. Twenty-six articles met study inclusion criteria. Studies derived from clinically referred cases had prevalence estimates that were significantly lower than those derived from population-based samples (P = 0.004). Among the 21 population-based prevalence studies, the pooled TS population prevalence estimate was 0.52% (95% confidence interval CI: 0.32-0.85). In univariable meta-regression analysis, study sample size (P = 0.002) and study date (P = 0.03) were significant predictors of TS prevalence. In the final multivariable model including sample size, study date, age, and diagnostic criteria, only sample size (P < 0.001) and diagnostic criteria (omnibus P = 0.003; Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision [DSM-IV-TR]: P = 0.005) were independently associated with variation in TS population prevalence across studies. This study refines the population prevalence estimate of TS in children to be 0.3% to 0.9%. Study sample size, which is likely a proxy for case assessment method, and the use of DSM-IV-TR diagnostic criteria are the major sources of heterogeneity across studies. The true TS population prevalence rate is likely at the higher end of these estimates, given the methodological limitations of most studies. Further studies in large, well-characterized samples will be helpful to determine the burden of disease in the general population.
本研究旨在完善儿童抽动障碍（TS）的人群患病率估计，并探讨先前发表的研究中存在的异质性的潜在来源。进行了系统评价，检查了所有符合条件的 TS 患病率的已发表研究。提取的数据采用样本量加权的随机效应荟萃分析；进行了荟萃回归分析，以检验先前提出的可能作为异质性潜在来源的协变量。有 26 篇文章符合研究纳入标准。来自临床转诊病例的研究的患病率估计明显低于来自基于人群的样本的患病率估计（P = 0.004）。在 21 项基于人群的患病率研究中，TS 人群患病率的汇总估计值为 0.52%（95%置信区间 CI：0.32-0.85）。在单变量荟萃回归分析中，研究样本量（P = 0.002）和研究日期（P = 0.03）是 TS 患病率的显著预测因子。在包括样本量、研究日期、年龄和诊断标准的最终多变量模型中，只有样本量（P < 0.001）和诊断标准（整体 P = 0.003；《精神障碍诊断与统计手册》第四版，文本修订版 [DSM-IV-TR]：P = 0.005）与研究间 TS 人群患病率的变化独立相关。本研究将儿童 TS 的人群患病率估计值从 0.3%细化到 0.9%。研究样本量可能是病例评估方法的代表，以及使用 DSM-IV-TR 诊断标准是研究间异质性的主要来源。鉴于大多数研究的方法学限制，TS 的真实人群患病率率可能更接近这些估计值的上限。在大型、特征明确的样本中进行进一步研究将有助于确定普通人群的疾病负担。
