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Psychometric Properties of MFM32 in Myotonic Dystrophy Type 1
Hanne Ludt Fossmo1, Mari Ellefsen-Martinsen2, Kristin Ørstavik3
1Department of Public Health Sciences and Interdisciplinary Health Sciences, Institute of Health and Society, Faculty of Medicine, University of Oslo, Oslo; Section for Rare Neuromuscular Disorders and EMAN, Department of Neurology, Oslo University Hospital, Oslo; Frambu Centre for Rare Disorders, Siggerud; Vikersund Rehabilitation Centre, Vikersund.
Objective:
To examine the psychometric properties of the Motor Function Measure 32 (MFM32) in adults with myotonic dystrophy type 1 (DM1). MFM32 is an instrument for evaluating function in neuromuscular diseases that have not been adequately assessed in DM1.
Design:
Cross-sectional study using video recording of the performance for evaluation of intra- and inter-rater reliability.
Setting:
Data collection was performed at 5 different sites (3 neurologic departments, 1 rehabilitation center, 1 center for rare disorders) and in several of the participants' home.
Participants:
Convenience sample of 86 adults (N=86) with DM1.
Interventions:
Not applicable.
Main Outcome Measures:
The MFM32. The objective was to assess internal consistency, intra and inter-rater reliability, measurement error, convergent validity, and floor and ceiling effects of MFM32 in adults with DM1. We analyzed the total percentage score and the 3 domain scores: D1 (standing and transfers), D2 (axial, proximal motor function), and D3 (distal motor function).
Results:
Internal consistency measured by Cronbach's alpha for the total percentage score was 0.94. For the 3 domains it was 0.95 (D1), 0.70 (D2), and 0.66 (D3). The intra-rater reliability coefficients were excellent (ICC2.1=0.96-0.99), and the inter-rater reliability coefficients were good to excellent (ICC2.1=0.86-0.99). SEM of the total percentage score was 1.59 for the intra-rater reliability and 1.66 for the inter-rater reliability. Minimal detectable change was 4.42 for the intra-rater reliability and 4.6 for the inter-rater reliability. Convergent validity was medium to high. There was a tendency for a ceiling effect on the total percentage score, as well as on some of the domain percentage scores.
Conclusions:
The MFM32 has a high internal consistency, intra- and inter- rater reliability, and convergent validity in adults with DM1. Calculated SEM and minimal detectable change were low. There is a ceiling effect of MFM32, especially in the distal function domain and among the participants with high motor function.
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