儿科多发性硬化及其家族性复发：一项基于人群的研究（1999-2017 年）。

Pediatric multiple sclerosis and its familial recurrence: A population based study (1999-2017).

机构信息

Multiple Sclerosis Research Center, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran.

出版信息

Mult Scler Relat Disord. 2019 Nov;36:101377. doi: 10.1016/j.msard.2019.101377. Epub 2019 Aug 27.

DOI:10.1016/j.msard.2019.101377

PMID:31473489

Abstract

BACKGROUND

The Pediatric onset multiple sclerosis (POMS) prevalence is increasing worldwide accounting for around 3 to 10% of MS cases. The risk of POMS is supposed to reflect a complex interaction between environmental and genetic risk factors that may occur during the childhood, adolescent, or post-pubertal years.

OBJECTIVE

The present study aimed at estimating the prevalence of POMS and assessing the epidemiology of familial recurrence of POMS in Tehran.

METHOD

A retrospective population based cross-sectional study was designed from 1999 to 2017. The baseline characteristic information was collected from MS patient's ≤18 years old (y/o). Pearson's chi-square test and logistic regression were used to analyze the relationship among variables and estimate the odds ratio (OR) via SPSS software, version 23.

RESULTS

A total of 1937 POMS patients (77.80% female and 22.20% male patients) participated in the study. The point prevalence of POMS was 16.20 per 100,000 populations in 2017. Mean age at disease onset was 15.96 ± 2.28 y/o. The female to male ratio was 2.02:1 in pre-pubertal cases (3-12 y/o), but it increased to 3.69:1 in 13-18 y/o age groups (P value = 0.001, OR = 1.82; 95% CI = 1.27-2.26). There were 288 (14.9%) cases with positive familial history of MS. The strongest association between MS risk and positive familial history was observed in second degree relatives who presented MS (P value = 0.046, OR = 1.74; 95%CI = 1.01-3.01). A significant association was observed among maternal second degree relatives with POMS (P value = 0.018, OR = 2.27; 95%CI = 1.15-4.47).

CONCLUSION

In comparison to other large studies, the prevalence of POMS was high in the data collected from Tehran. POMS risk is higher among females and the sex ratio increases after puberty. We found a significant association between POMS risk and familial history in maternal second degree relatives. Further studies of POMS epidemiology might yield greater understanding of the natural history of this disease.

摘要

背景

儿科发病多发性硬化症（POMS）的患病率在全球范围内呈上升趋势，占多发性硬化症病例的 3%至 10%。POMS 的风险被认为反映了环境和遗传风险因素之间的复杂相互作用，这些因素可能发生在儿童期、青春期或青春期后。

目的

本研究旨在估计 POMS 的患病率，并评估德黑兰 POMS 家族复发的流行病学。

方法

从 1999 年到 2017 年，进行了一项回顾性基于人群的横断面研究。从≤18 岁的 MS 患者中收集基线特征信息。采用 Pearson's chi-square 检验和 logistic 回归分析变量之间的关系，并使用 SPSS 软件（版本 23）估计比值比（OR）。

结果

共有 1937 名 POMS 患者（77.80%为女性，22.20%为男性）参与了研究。2017 年 POMS 的时点患病率为每 10 万人中 16.20 人。发病时的平均年龄为 15.96±2.28 岁。在青春期前（3-12 岁）病例中，女性与男性的比例为 2.02:1，但在 13-18 岁年龄组中增加到 3.69:1（P 值=0.001，OR=1.82；95%CI=1.27-2.26）。有 288 例（14.9%）有 MS 阳性家族史。与 MS 风险与阳性家族史之间的最强关联见于患有 MS 的二级亲属（P 值=0.046，OR=1.74；95%CI=1.01-3.01）。在与 POMS 相关的母亲二级亲属中观察到显著的相关性（P 值=0.018，OR=2.27；95%CI=1.15-4.47）。