Playle Rebecca, Dimitropoulou Polyxeni, Kelson Mark, Quinn Lori, Busse Monica
Centre for Trials Research Cardiff University Cardiff United Kingdom.
School of Mathematics/The Alan Turing Institute University of Exeter Exeter United Kingdom.
Mov Disord Clin Pract. 2019 Jul 18;6(7):567-575. doi: 10.1002/mdc3.12809. eCollection 2019 Sep.
Physical activity may be beneficial in Huntington's disease (HD); however, studies to date have been underpowered to detect change. We combined data from five randomized controlled feasibility trials using individual patient data meta-analyses.
METHODS/DESIGN: All trial interventions comprised a combination of supervised and self-directed physical activity, with varied emphasis on aerobic, strength, endurance, flexibility, and task training. Duration ranged from 8 to 16 weeks. The primary outcome was the modified Unified Huntington's Disease Rating Motor Score. Secondary outcomes included the Symbol Digit Modality Test, Berg Balance Scale, 30-second Chair stand, Timed Up and Go, Gait Speed, Physical Performance Test, Six-Minute Walk, International Physical Activity Questionnaire, Hospital Anxiety and Depression Scale, EuroQol Health Utility Index, and Short-Form 36 Health Related Quality of Life Scale. The primary analysis utilized a two-stage approach. A one-stage approach was explored as a sensitivity analysis using a cross-classified (by study site) linear mixed-effects model.
One hundred twenty-one participants provided complete data. Risk of bias was moderate; however, primary outcomes were blind assessed. Primary pooled effect estimates adjusted for baseline modified motor score (95% confidence interval) were 0.2 (-2.1 to 2.6) favoring control. There was considerable heterogeneity between the studies.
There was no evidence of an exercise effect on the modified motor score in these relatively short-duration interventions. Longer-duration trials incorporating supervised components meeting frequency, intensity, time, and type principles are required. Lack of common outcomes limited the analysis and highlight the importance of a core outcome set for evaluating exercise in HD.
体育活动可能对亨廷顿舞蹈症（HD）有益；然而，迄今为止的研究在检测变化方面的能力不足。我们使用个体患者数据荟萃分析合并了五项随机对照可行性试验的数据。
方法/设计：所有试验干预措施均包括监督性和自主体育活动的组合，对有氧运动、力量训练、耐力训练、灵活性训练和任务训练的侧重点各不相同。持续时间为8至16周。主要结局是改良统一亨廷顿舞蹈症评定运动评分。次要结局包括符号数字模态测试、伯格平衡量表、30秒坐立试验、计时起立行走测试、步速、身体性能测试、六分钟步行试验、国际体力活动问卷、医院焦虑抑郁量表、欧洲生活质量健康效用指数和36项简短健康调查量表。主要分析采用两阶段方法。作为敏感性分析，使用交叉分类（按研究地点）线性混合效应模型探索了单阶段方法。
121名参与者提供了完整数据。偏倚风险为中等；然而，主要结局进行了盲法评估。根据基线改良运动评分调整后的主要合并效应估计值（95%置信区间）为0.2（-2.1至2.6），有利于对照组。各研究之间存在相当大的异质性。
在这些持续时间相对较短的干预措施中，没有证据表明运动对改良运动评分有影响。需要进行持续时间更长的试验，纳入符合频率、强度、时间和类型原则的监督性组成部分。缺乏共同结局限制了分析，并凸显了核心结局集对于评估HD运动的重要性。
