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迟发性多发性硬化症的临床特征:系统评价和荟萃分析。

Clinical Features of Late-Onset Multiple Sclerosis: a Systematic Review and Meta-analysis.

机构信息

Student Research Committee, Tabriz University of Medical Sciences, Tabriz, Iran; Neurosciences Research Center (NSRC), Tabriz University of Medical Sciences, Tabriz, Iran; Research Center for Evidence Based Medicine, Iranian EBM Centre, a Joanna Briggs Institute Affiliated Center, Tabriz University of Medical Sciences, Tabriz, Iran.

Sina MS Research Center, Sina hospital, and department of neurology, Tehran University of Medical Sciences, Tehran, Iran.

出版信息

Mult Scler Relat Disord. 2021 May;50:102816. doi: 10.1016/j.msard.2021.102816. Epub 2021 Feb 4.

Abstract

BACKGROUND

Multiple sclerosis (MS) commonly affects young adults at the ages 20 to 40 years old, but it can onset at each age. Late-onset multiple sclerosis (LOMS) is defined as symptoms initiating after the age of 50. Misdiagnosis and a remarkable gap in diagnosis of LOMS is a challenge of the elderly population so in this article we described the proportion of LOMS and the clinical features and phenotype of the disease in this age group.

METHODS

After registration of the study protocol, an electronic search was performed in 3 databases and for full coverage of the published studies, we also checked the references of each related article. Two independent researchers screened the records in title/abstract and full-text stages and extracted the data using a data extraction table. The risk of bias was assessed using Joanna Briggs Institute checklist and meta-analysis was conducted by CMA 2. Only the studies with 50 years old cut-off and using McDonald or Poser diagnostic criteria were included in the meta-analysis.

RESULTS

After removing duplicated studies, out of 733 results of electronic and hand searching, 31 studies met our inclusion criteria for the systematic review, and 11 of them were included in the quantitative synthesis. With different cut-offs and diagnostic methods 1.1% to 21.3% proportion of LOMS, has been reported in the studies. Meta-analysis reached a 5.01% (95% CI: 3.78% to 6.57%), proportion of LOMS in total MS cases. The female cases were more than males (range between 57.7% to 70.2%) and 64.46% (95% CI: 61.94% to 66.91%) proportion of females has been found in this study. 65.00% (95% CI: 44.71% to 81.02%) proportion of spinal cord involvements and 49.80% (95% CI: 39.28% to 60.24%) proportion of relapsing-remitting multiple sclerosis (RRMS) was also observed in LOMS cases. In 4 of included studies, the progressive form was the predominant phenotype. The most prevalent first disease presentation of LOMS was motor dysfunction (ranges between 100% to 35.4%) followed by sensory problems (ranges between 94% to 5%), visual symptoms (ranges between 22.9% to 5%), and brainstem dysfunction (ranges between 25% to 12.3%). The proportion of positive oligoclonal band (OCB), was varied from 46% to 98% in different studies and positive immunoglobulin G (IgG) index also was seen in 45.04% and 66% of the patients. 2.2% - 12.5% of the LOMS cases had a positive family history.

CONCLUSION

In about 5% of cases, MS can be diagnosed at ages above 50 years old. There is an increasing concern of a more progressive form of MS in LOMS cases. Unlike the adult-onset MS, the first presentation of LOMS is usually motor dysfunction. Understanding the proportion and clinical features of LOMS will help clinicians with the diagnosis of MS in this age group and prevention of wrong management plans and complications in these patients. Although the proportion of females is still more than males in LOMS cases; but there is a trend to increase in male cases with aging.

摘要

背景

多发性硬化症(MS)通常在 20 至 40 岁的年轻人中发病,但也可能在任何年龄发病。迟发性多发性硬化症(LOMS)是指症状在 50 岁后出现。老年人的误诊和对 LOMS 的诊断差距是一个挑战,因此本文描述了 LOMS 的比例以及该年龄组的疾病的临床表现和表型。

方法

在注册研究方案后,我们在 3 个数据库中进行了电子搜索,并为了全面覆盖已发表的研究,我们还检查了每篇相关文章的参考文献。两名独立的研究人员在标题/摘要和全文阶段筛选记录,并使用数据提取表提取数据。使用 Joanna Briggs 研究所检查表评估偏倚风险,并使用 CMA 2 进行荟萃分析。仅纳入 50 岁截止且使用 McDonald 或 Poser 诊断标准的研究进行荟萃分析。

结果

在排除重复研究后,电子和手工搜索的 733 项结果中,有 31 项研究符合我们的系统评价纳入标准,其中 11 项研究纳入了定量综合分析。使用不同的截止值和诊断方法,LOMS 的比例为 1.1%至 21.3%,这在研究中有所报道。荟萃分析得出 LOMS 占总 MS 病例的 5.01%(95%CI:3.78%至 6.57%)。女性病例多于男性(范围在 57.7%至 70.2%),在这项研究中发现女性的比例为 64.46%(95%CI:61.94%至 66.91%)。LOMS 病例中脊髓受累的比例为 65.00%(95%CI:44.71%至 81.02%),复发缓解型多发性硬化症(RRMS)的比例为 49.80%(95%CI:39.28%至 60.24%)。在纳入的 4 项研究中,进行性表型是主要表型。LOMS 首次发病的最常见表现是运动功能障碍(范围在 100%至 35.4%之间),其次是感觉问题(范围在 94%至 5%之间)、视觉症状(范围在 22.9%至 5%之间)和脑干功能障碍(范围在 25%至 12.3%之间)。不同研究中寡克隆带(OCB)阳性的比例从 46%到 98%不等,阳性免疫球蛋白 G(IgG)指数也在 45.04%和 66%的患者中出现。2.2%至 12.5%的 LOMS 病例有阳性家族史。

结论

约 5%的 MS 病例可在 50 岁以上确诊。LOMS 病例中更常见进展性 MS。与成人发病的 MS 不同,LOMS 的首发表现通常是运动功能障碍。了解 LOMS 的比例和临床表现将有助于临床医生在这一年龄组中诊断 MS,并防止对这些患者的错误管理计划和并发症。尽管 LOMS 病例中女性的比例仍然高于男性,但男性病例的比例随着年龄的增长呈上升趋势。

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