Muscle & nerve · 2013
Rasch analysis of clinical outcome measures in spinal muscular atrophy.
СтатьяПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод пока не подготовлен
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Cano Stefan J, Mayhew Anna, Glanzman Allan M, Krosschell Kristin J, Swoboda Kathryn J, Main Marion, Steffensen Birgit F, Bérard Carole, Girardot Françoise, Payan Christine A M, Mercuri Eugenio, Mazzone Elena, Elsheikh Bakri, Florence Julaine, Hynan Linda S, Iannaccone Susan T, Nelson Leslie L, Pandya Shree, Rose Michael, Scott Charles, Sadjadi Reza, Yore Mackensie A, Joyce Cynthia, Kissel John T, International Coordinating Committee for SMA Clinical Trials Rasch Task Force
Аннотация
INTRODUCTION: Trial design for SMA depends on meaningful rating scales to assess outcomes. In this study Rasch methodology was applied to 9 motor scales in spinal muscular atrophy (SMA).
METHODS: Data from all 3 SMA types were provided by research groups for 9 commonly used scales. Rasch methodology assessed the ordering of response option thresholds, tests of fit, spread of item locations, residual correlations, and person separation index.
RESULTS: Each scale had good reliability. However, several issues impacting scale validity were identified, including the extent that items defined clinically meaningful constructs and how well each scale measured performance across the SMA spectrum.
CONCLUSIONS: The sensitivity and potential utility of each SMA scale as outcome measures for trials could be improved by establishing clear definitions of what is measured, reconsidering items that misfit and items whose response categories have reversed thresholds, and adding new items at the extremes of scale ranges.
METHODS: Data from all 3 SMA types were provided by research groups for 9 commonly used scales. Rasch methodology assessed the ordering of response option thresholds, tests of fit, spread of item locations, residual correlations, and person separation index.
RESULTS: Each scale had good reliability. However, several issues impacting scale validity were identified, including the extent that items defined clinically meaningful constructs and how well each scale measured performance across the SMA spectrum.
CONCLUSIONS: The sensitivity and potential utility of each SMA scale as outcome measures for trials could be improved by establishing clear definitions of what is measured, reconsidering items that misfit and items whose response categories have reversed thresholds, and adding new items at the extremes of scale ranges.