Student Research Committee, University of Social Welfare and Rehabilitation Sciences, Koodakyar Alley, Daneshjoo Blvd., Evin St, Tehran, Iran.
Department of Genetics and Molecular Biology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran.
BMC Neurol. 2021 Jun 28;21(1):246. doi: 10.1186/s12883-021-02267-9.
Considering that many recent studies have reported the prevalence of familial multiple sclerosis (FMS), we performed an updated meta-analysis of the worldwide prevalence of FMS by the addition of recent publications.
A search in PubMed, Scopus, the ISI Web of Science, and Google Scholar was undertaken up to 20 December 2020. The inclusion criteria were based on the CoCoPop approach (condition, context, and population). Meta-analysis of the qualified studies was conducted by comprehensive meta-analysis ver. 2 software.
The pooled prevalence of MS in relatives of 16,179 FMS cases was estimated to be 11.8% (95% CI: 10.7-13) based on a random-effects model. The pooled mean age of disease onset in adult probands was calculated to be 28.7 years (95% CI: 27.2 ± 30.2). Regarding 13 studies that reported the data of FMS in pediatrics (n = 877) and adults (n = 6636), the FMS prevalence in pediatrics and adults was 15.5% (95% CI: 13.8-17.4) and 10.8% (95% CI: 8.1-14.2), respectively. The prevalence of FMS in affected males (n = 5243) and females (n = 11,503) was calculated to be 13.7% (95% CI: 10.1-18.2) and 15.4% (95% CI: 10.3-22.4), respectively. The odds ratio of male/female in FMS cases was not statistically significant (OR = 0.9; 95% CI: 0.6-1.2, P = 0.55). Subgroup analysis demonstrated a significant difference in the prevalence of FMS between the geographical areas (P = 0.007). The meta-regression model indicated that the prevalence of FMS is lower with higher latitude and higher MS prevalence (P < 0.001). In contrast, meta-regression based on prevalence day was not statistically significant (P = 0.29).
The prevalence of FMS is higher in the pediatric group than that of adults, distinct between geographical areas, and diminishes with the increment of MS prevalence and latitude. Also, the symptoms initiate relatively at younger ages in the FMS cases. Interestingly, our analysis unveiled that FMS is not more prevalent in men than women and the risk of MS development in relatives is not higher when the affected proband is male.
鉴于最近许多研究报告了家族性多发性硬化症(FMS)的流行情况,我们通过增加最近的出版物,对全球范围内 FMS 的流行情况进行了更新的荟萃分析。
截至 2020 年 12 月 20 日,在 PubMed、Scopus、ISI Web of Science 和 Google Scholar 中进行了检索。纳入标准基于 CoCoPop 方法(条件、背景和人群)。使用 Comprehensive Meta-Analysis ver.2 软件对合格研究进行荟萃分析。
基于随机效应模型,16179 例 FMS 病例的亲属中 MS 的汇总患病率估计为 11.8%(95%CI:10.7-13)。成年先证者疾病发病的平均年龄计算为 28.7 岁(95%CI:27.2±30.2)。对于 13 项报告儿科(n=877)和成人(n=6636)FMS 数据的研究,儿科和成人 FMS 的患病率分别为 15.5%(95%CI:13.8-17.4)和 10.8%(95%CI:8.1-14.2)。在受累男性(n=5243)和女性(n=11503)中,FMS 的患病率分别为 13.7%(95%CI:10.1-18.2)和 15.4%(95%CI:10.3-22.4)。FMS 病例中男性/女性的优势比无统计学意义(OR=0.9;95%CI:0.6-1.2,P=0.55)。亚组分析表明,FMS 的流行率在地理区域之间存在显著差异(P=0.007)。荟萃回归模型表明,FMS 的患病率随纬度升高和 MS 患病率升高而降低(P<0.001)。相反,基于患病率日的荟萃回归无统计学意义(P=0.29)。
FMS 在儿科组中的患病率高于成人组,在地理区域之间存在差异,并且随着 MS 患病率和纬度的增加而降低。此外,FMS 病例的症状起始年龄相对较小。有趣的是,我们的分析表明,FMS 在男性中的患病率并不高于女性,受累先证者为男性时,亲属患 MS 的风险也不会更高。