Wearable-Derived Patterns of Performance Fatigability During Gait in Spinal Muscular Atrophy.

IF 3.1 3区 医学 Q2 CLINICAL NEUROLOGY
Muscle & Nerve Pub Date : 2025-09-01 Epub Date: 2025-06-25 DOI:10.1002/mus.28461
Jacqueline Montes, Cara H Kanner, Ton Duong, Rafael Rodriguez-Torres, David Uher, Sally Dunaway Young, Rabia Farooquee, Abby Druffner, Amy Pasternak, Maria Fragala-Pinkham, Damiano Zanotto
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引用次数: 0

Abstract

Introduction/aims: Despite disease-modifying therapies, fatigability persists in spinal muscular atrophy (SMA). Performance fatigability (PF) during the six-minute walk test (6MWT) is mostly unchanged in treated SMA. This cross-sectional study characterized PF using instrumented insoles.

Methods: Ambulatory individuals with SMA (n = 14) and controls (HC) (n = 10) were included. Spatiotemporal and kinetic parameters were collected with custom-engineered instrumented insoles during the 6MWT. Linear mixed models analyzed parameter trends, with trend slope representing PF. Changes in mean velocity (Vavg) and stride-by-stride parameters were compared between minute 1 and 6 and between groups.

Results: Decreases in Vavg were greatest for severe SMA (p < 0.001). Changes were found in stride length (SL) (p = 0.048) and stride velocity (SV) (p = 0.030) for severe SMA, and in stance phase (%St) (p = 0.012) and percent terminal double support (%DS) (p = 0.02) for mild SMA. SMA subgroups showed downward trends from minute 1 to 6 in SL, SV, and anterior-posterior center of pressure (AP-COP) (p < 0.001), and increases in Absolute COP-Cyclogram Asymmetry Index (|ASI|) (p < 0.05). Trends differed between severe SMA and other groups for SL, SV, %St, and %DS (p < 0.001), and for AP-COP and |ASI| (p < 0.05). Trends for SL (p < 0.001), SV and AP-COP (p < 0.01) differed between HC and mild SMA.

Discussion: PF in SMA manifests as changes in gait parameters. Instrumented insoles revealed fatigue-related changes not captured with the conventional method of comparing the first and last minutes of the 6MWT. Spatiotemporal and kinetic parameters contribute to understanding of impairments and inform therapeutic development.

Trial registration: ClinicalTrials.gov: NCT04193085.

脊髓性肌萎缩症患者步态中疲劳表现的可穿戴模式。
简介/目的:尽管有疾病改善疗法,但脊髓性肌萎缩症(SMA)患者仍然存在疲劳。6分钟步行试验(6MWT)期间的疲劳性能(PF)在治疗SMA中基本没有变化。本横断面研究采用内固定鞋垫表征PF。方法:纳入流动SMA患者(n = 14)和对照组(HC) (n = 10)。在6MWT期间,使用定制设计的仪器鞋垫收集时空和动力学参数。线性混合模型分析参数趋势,趋势斜率表示PF,比较1分钟和6分钟以及组间平均速度(Vavg)和跨步参数的变化。结果:严重SMA患者Vavg下降最大(p讨论:SMA患者的PF表现为步态参数的改变。仪器的鞋垫揭示了疲劳相关的变化,而不是用比较6MWT的第一和最后几分钟的传统方法捕获的。时空和动力学参数有助于理解损伤,并为治疗发展提供信息。试验注册:ClinicalTrials.gov: NCT04193085。
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
Muscle & Nerve
Muscle & Nerve 医学-临床神经学
CiteScore
6.40
自引率
5.90%
发文量
287
审稿时长
3-6 weeks
期刊介绍: Muscle & Nerve is an international and interdisciplinary publication of original contributions, in both health and disease, concerning studies of the muscle, the neuromuscular junction, the peripheral motor, sensory and autonomic neurons, and the central nervous system where the behavior of the peripheral nervous system is clarified. Appearing monthly, Muscle & Nerve publishes clinical studies and clinically relevant research reports in the fields of anatomy, biochemistry, cell biology, electrophysiology and electrodiagnosis, epidemiology, genetics, immunology, pathology, pharmacology, physiology, toxicology, and virology. The Journal welcomes articles and reports on basic clinical electrophysiology and electrodiagnosis. We expedite some papers dealing with timely topics to keep up with the fast-moving pace of science, based on the referees'' recommendation.
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