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《Cure SMA 会员调查》:脊髓性肌萎缩症个体关键人口统计学和临床特征要点。

The Cure SMA Membership Surveys: Highlights of Key Demographic and Clinical Characteristics of Individuals with Spinal Muscular Atrophy.

机构信息

Cure SMA, Elk Grove Village, IL, USA.

Biogen, Cambridge, MA, USA.

出版信息

J Neuromuscul Dis. 2021;8(1):109-123. doi: 10.3233/JND-200563.

Abstract

BACKGROUND

Cure SMA maintains the largest patient-reported database for people affected with spinal muscular atrophy (SMA). In 2017, Cure SMA initiated annual surveys with their membership to collect demographic and disease characteristics, healthcare, and burden of disease information from patients and caregivers.

OBJECTIVE

To summarize results from two large-scale Cure SMA surveys in 2017 and 2018.

METHODS

Cure SMA database members were invited to complete surveys; these were completed by caregivers for living or deceased individuals with SMA and/or affected adults.

RESULTS

In 2017, 726 surveys were completed for 695 individuals with SMA; in 2018, 796 surveys were completed for 760 individuals with SMA. Data from both survey years are available for 313 affected individuals. Age at symptom onset, distribution of SMN2 gene copy number, and representation of each SMA type in the surveys were consistent with that expected in the SMA population. In the 2018 survey, the average age at diagnosis was 5.2 months for SMA type I and the reported mean age at death for this subgroup was 27.8 months. Between survey years, there was consistency in responses for factors that should not change within individuals over time (e.g., reported age at diagnosis).

CONCLUSIONS

Results from the Cure SMA surveys advance the understanding of SMA and facilitate advocacy efforts and healthcare services planning. Longitudinal surveys are important for evaluating the impact of effective treatments on changing phenotypes, and burden of disease and care in individuals with SMA.

摘要

背景

Cure SMA 维护着最大的脊髓性肌萎缩症(SMA)患者报告数据库。2017 年,Cure SMA 开始对其会员进行年度调查,以从患者和护理人员那里收集人口统计学和疾病特征、医疗保健以及疾病负担信息。

目的

总结 2017 年和 2018 年两次 Cure SMA 大型调查的结果。

方法

邀请 Cure SMA 数据库成员完成调查;这些调查由 SMA 患者和/或受影响的成年人的护理人员完成。

结果

2017 年,有 726 名 SMA 患者完成了 726 份调查;2018 年,有 760 名 SMA 患者完成了 796 份调查。来自这两年调查的数据可用于 313 名受影响的个体。发病年龄、SMN2 基因拷贝数分布以及各型 SMA 在调查中的表现与 SMA 人群中的预期一致。在 2018 年的调查中,SMA 型 I 的平均诊断年龄为 5.2 个月,该亚组报告的平均死亡年龄为 27.8 个月。在调查年份之间,对于不应随时间在个体中变化的因素(例如,报告的诊断年龄)的反应保持一致。

结论

Cure SMA 调查的结果增进了对 SMA 的认识,并有助于倡导工作和医疗保健服务计划。纵向调查对于评估有效治疗对改变表型以及 SMA 患者的疾病负担和护理的影响非常重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f1c4/7902958/1b7512fcb190/jnd-8-jnd200563-g001.jpg

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