Burden of respiratory syncytial virus in spinal muscular atrophy: a retrospective nationwide cross-sectional and birth cohort study.

IF 3.6 2区 医学 Q2 GENETICS & HEREDITY
Hao-Wei Chung, Chen-Hua Wang, Chia-Hung Yu, Chiao-Yun Huang, Fu-Wen Liang
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引用次数: 0

Abstract

Background: Spinal muscular atrophy (SMA) is a severe neuromuscular disease associated with a high risk of respiratory complications, yet real-world data on respiratory syncytial virus (RSV) infection in this population remain limited. This study aimed to investigate the severity, hospitalization costs, and seasonal patterns of RSV infections in patients with SMA using real-world data in Taiwan.

Methods: This retrospective study analyzed data from Taiwan's National Health Insurance Research Database from 2009 to 2021. Patients with SMA were identified through at least two outpatient clinic visits and one hospital admission. The analysis included a cross-sectional study and a birth cohort analysis for sensitivity testing. RSV-associated hospitalizations were identified using diagnostic codes for RSV. The main outcomes included ICU admissions, ventilator use, hospitalization costs, timing of RSV onset, and seasonal infection trends.

Results: This study documented 914 hospitalizations in patients with SMA over 13 years, including 14 RSV infections (1.5%). All RSV-related hospitalizations were documented before age 18. In a birth cohort analysis comparing 110 patients with SMA to 2,103,997 individuals from the general population, patients with SMA demonstrated significantly higher RSV hospitalization rates (9.8% vs. 1.6%), ICU admissions (54.5% vs. 4.7%; p < 0.0001), and ventilator use (54.5% vs. 1.7%; p < 0.0001), with a later mean onset (2.54 ± 2.05 years vs. 1.05 ± 1.05 years; p = 0.0073) and substantially higher hospitalization costs (median 967 [560-6,462] USD vs. 683 [487-1002] USD; p = 0.0480). Infections peaked in fall and winter (72.7%) for patients with SMA. ICU admission rates for RSV increased with age and decreased with later SMA onset.

Conclusions: Patients with SMA are at heightened risk for severe RSV infections even beyond infancy, underscoring the need for age-specific and seasonal prevention strategies, particularly for those over two years old.

脊髓性肌萎缩的呼吸道合胞病毒负担:一项回顾性全国横断面和出生队列研究。
背景:脊髓性肌萎缩症(SMA)是一种与呼吸道并发症高风险相关的严重神经肌肉疾病,但该人群中呼吸道合胞病毒(RSV)感染的真实数据仍然有限。本研究旨在探讨台湾地区SMA患者RSV感染的严重程度、住院费用和季节特征。方法:回顾性分析2009年至2021年台湾全民健康保险研究数据库的数据。SMA患者是通过至少两次门诊就诊和一次住院来确定的。分析包括横断面研究和出生队列分析的敏感性测试。使用RSV诊断代码确定RSV相关的住院情况。主要结局包括ICU入院、呼吸机使用、住院费用、RSV发病时间和季节性感染趋势。结果:该研究记录了13年间914例SMA患者的住院治疗,包括14例RSV感染(1.5%)。所有与rsv相关的住院记录均在18岁之前。在一项出生队列分析中,将110名SMA患者与普通人群中的2,103,997名患者进行了比较,SMA患者的RSV住院率(9.8%比1.6%)和ICU入院率(54.5%比4.7%)显著高于其他患者。结论:SMA患者甚至在婴儿期之后发生严重RSV感染的风险也更高,这强调了针对年龄和季节性预防策略的必要性,特别是对于2岁以上的患者。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Orphanet Journal of Rare Diseases
Orphanet Journal of Rare Diseases 医学-医学:研究与实验
CiteScore
6.30
自引率
8.10%
发文量
418
审稿时长
4-8 weeks
期刊介绍: Orphanet Journal of Rare Diseases is an open access, peer-reviewed journal that encompasses all aspects of rare diseases and orphan drugs. The journal publishes high-quality reviews on specific rare diseases. In addition, the journal may consider articles on clinical trial outcome reports, either positive or negative, and articles on public health issues in the field of rare diseases and orphan drugs. The journal does not accept case reports.
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