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ABSTRACT: 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.
SUBMITTER: Cano SJ
PROVIDER: S-EPMC4376296 | biostudies-literature | 2014 Mar
REPOSITORIES: biostudies-literature
Cano Stefan J SJ Mayhew Anna A Glanzman Allan M AM Krosschell Kristin J KJ Swoboda Kathryn J KJ Main Marion M Steffensen Birgit F BF Bérard Carole C Girardot Françoise F Payan Christine A M CA Mercuri Eugenio E Mazzone Elena E Elsheikh Bakri B Florence Julaine J Hynan Linda S LS Iannaccone Susan T ST Nelson Leslie L LL Pandya Shree S Rose Michael M Scott Charles C Sadjadi Reza R Yore Mackensie A MA Joyce Cynthia C Kissel John T JT
Muscle & nerve 20130726 3
<h4>Introduction</h4>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).<h4>Methods</h4>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.<h4>Results</h4>Each scale had good reliab ...[more]