Why the study?
The availability of disease-modifying therapies for spinal muscular atrophy highlighted a growing need for normative 6-minute walk test reference data to compare patients with healthy controls.
There is significant variability in % predicted values for the 6MWT in SMA patients depending on the reference equation used, highlighting limitations in current normative data.
Existing 6MWT norms produce highly variable % predicted values in SMA (P<0.001); leaves open need for standardized, disease-specific references.
INTRODUCTION: The 6-minute walk test (6MWT) is a well-established clinical assessment of functional endurance, validated as a measure of walking ability in spinal muscular atrophy (SMA). The current availability of disease-modifying therapies for SMA indicates a growing need for normative reference data to compare SMA patients with healthy controls. METHODS: The literature was searched in two scientific databases. Studies were evaluated and selected based on adherence to American Thoracic Society guidelines for administering the 6MWT. Reference equations from the selected studies were applied to 6MWT data collected from SMA patients to calculate and compare % predicted values. RESULTS: Three pediatric and six adult studies were selected for comparison. The % predicted values using the pediatric and adult equations ranged from 47.7 ± 18.2% to 67.6 ± 26.2% and 43.0 ± 17.9% to 59.5 ± 26.2%, respectively, and were significantly different (P < 0.001). DISCUSSION: Results suggest significant variability between % predicted values derived from published reference equations in children and adults, despite adherence to 6MWT standardization.
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Goodwin et al. (2019) studied this question.
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