弗里德赖希共济失调评定量表的心理测量学特性。
Psychometric properties of the Friedreich Ataxia Rating Scale.
作者信息
Rummey Christian, Corben Louise A, Delatycki Martin B, Subramony S H, Bushara Khalaf, Gomez Christopher M, Hoyle Joseph Chad, Yoon Grace, Ravina Bernard, Mathews Katherine D, Wilmot George, Zesiewicz Theresa, Perlman Susan, Farmer Jennifer M, Lynch David R
机构信息
Clinical Data Science GmbH (C.R.), Basel, Switzerland; Bruce Lefroy Centre for Genetic Health Research (L.A.C., M.B.D.), Murdoch Children's Research Institute, Parkville, Victoria, Australia; Department of Paediatrics (L.A.C., M.B.D.), University of Melbourne, Parkville, Victoria, Australia; Department of Neurology (S.H.S.), McKnight Brain Institute, Room, Gainesville, FL; University of Minnesota (K.B.); University of Chicago (C.M.G.); Ohio State University (J.C.H.); Divisions of Neurology and Clinical and Metabolic Genetics (G.Y.), Department of Paediatrics, the Hospital for Sick Children, University of Toronto, Ontario, Canada Hospital; University of Rochester (B.R.); University of Iowa (K.D.M.); Emory University (G.W.); University of South Florida (T.Z.); Friedreich's Ataxia Research Alliance (S.P.), Downingtown, PA; and Division of Neurology (D.R.L.), Children's Hospital of Philadelphia.
出版信息
Neurol Genet. 2019 Oct 29;5(6):371. doi: 10.1212/NXG.0000000000000371. eCollection 2019 Dec.
OBJECTIVE
To investigate the psychometric properties of the Friedreich Ataxia Rating Scale neurologic examination (FARSn) and its subscores, as well as the influence of the modifications resulting in the now widely used modified FARS (mFARS) examination.
METHODS
Based on cross-sectional FARS data from the FA-Clinical Outcome Measures cohort, we conducted correlation-based psychometric analyses to investigate the interplay of items and subscores within the FARSn/mFARS constructs.
RESULTS
The results provide support for both the FARSn and the mFARS constructs, as well as individually for their upper limb and lower limb coordination components. The omission of the peripheral nervous system subscore (D) and 2 items of the bulbar subscore (A) in the mFARS strengthens the overall construct compared with the complete FARS.
CONCLUSIONS
A correlation-based psychometric analysis of the neurologic FARSn score justifies the overall validity of the scale. In addition, omission of items of limited functional significance as created in the mFARS improves the features of the measures. Such information is crucial to the ongoing application of the mFARS in natural history studies and clinical trials. Additional analyses of longitudinal changes will be necessary to fully ascertain its utility, especially in nonambulant patients.
目的
研究弗里德赖希共济失调评定量表神经学检查（FARSn）及其子评分的心理测量特性，以及导致目前广泛使用的改良FARS（mFARS）检查的修改所产生的影响。
方法
基于FA临床结局测量队列的横断面FARS数据，我们进行了基于相关性的心理测量分析，以研究FARSn/mFARS结构中各项目与子评分之间的相互作用。
结果
结果为FARSn和mFARS结构提供了支持，也分别为其上、下肢协调成分提供了支持。与完整的FARS相比，mFARS中省略了周围神经系统子评分（D）和延髓子评分（A）的2个项目，增强了整体结构。
结论
对神经学FARSn评分进行基于相关性的心理测量分析证明了该量表的整体有效性。此外，mFARS中省略功能意义有限的项目改善了测量指标的特性。这些信息对于mFARS在自然史研究和临床试验中的持续应用至关重要。有必要对纵向变化进行额外分析，以充分确定其效用，尤其是在非行走患者中。
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