Unit of Biostatistics, Fondazione IRCCS Casa Sollievo Della Sofferenza, San Giovanni Rotondo, Italy.
Ministère de la Sante et des Solidarités, Cellule Interministérielle Recherche MSS/MESRI, Paris, France.
J Neurol. 2021 Aug;268(8):2973-2984. doi: 10.1007/s00415-021-10464-2. Epub 2021 Feb 25.
A noticeable change of the male-to-female sex ratio (SR) has been observed in Amyotrophic Lateral Sclerosis (ALS) leading to an apparent regression of SR with time (SR close to 1:1).
To provide a global SR estimate and investigate its relation with respect to population age.
A systematic review and meta-analysis was conducted including only population-based studies with a high-quality methodology in European ancestral origin population. Male-to-female SR was estimated by three different measures: SR number, SR crude incidence and SR standardized incidence. Standard and dose-response meta-analyses were performed to assess the pooled SR measures (irrespective of population age) and the evolution of the SR measures with respect to population age, respectively. Potential sources of heterogeneity were investigated via meta-regression.
Overall, 3254 articles were retrieved in the literature search. Thirty-nine studies stratified by time periods were included. The overall pooled male-to-female ratio was 1.28 (95% CI 1.23-1.32) for SR number, 1.33 (95% CI 1.29-1.38) for SR crude incidence and 1.35 (95% CI 1.31-1.40) for SR standardized incidence. The SR number with respect to population age reveals a progressive reduction of SR at increasing age, while the SR crude incidence in relation to age displays a U-shaped curve.
The number and the incidence of ALS cases were consistently higher in males than females. Dose-response meta-analysis showed that SR measures change with respect to population age. Further original research is needed to clarify if our findings are reproducible in other populations.
肌萎缩侧索硬化症（ALS）中男性与女性的性别比例（SR）发生了显著变化，导致 SR 随时间呈明显回归（SR 接近 1:1）。
提供全球 SR 估计值，并研究其与人口年龄的关系。
仅纳入欧洲祖先人群中具有高质量方法学的基于人群的研究，进行系统评价和荟萃分析。通过三种不同的测量方法（SR 数量、SR 粗发病率和 SR 标准化发病率）来估计男性与女性的 SR：SR 数量、SR 粗发病率和 SR 标准化发病率。分别进行标准和剂量反应荟萃分析，以评估汇总的 SR 测量值（无论人口年龄如何）和 SR 测量值随人口年龄的变化。通过荟萃回归分析调查潜在的异质性来源。
文献检索中总共检索到 3254 篇文章。纳入了 39 项按时间划分的研究。总体男性与女性的比值为 1.28（95% CI 1.23-1.32），用于 SR 数量；1.33（95% CI 1.29-1.38）用于 SR 粗发病率；1.35（95% CI 1.31-1.40）用于 SR 标准化发病率。SR 数量与人口年龄的关系表明，随着年龄的增长，SR 逐渐降低，而与年龄相关的 SR 粗发病率呈 U 形曲线。
男性的 ALS 病例数量和发病率始终高于女性。剂量反应荟萃分析表明，SR 测量值随人口年龄而变化。需要进一步的原始研究来澄清我们的发现是否可以在其他人群中重现。
