Key result
The Endurance Shuttle Tests demonstrated moderate to excellent test-retest reliability and good discriminative validity, with 54% to 73% of SMA patients showing increased fatigability and dropping out compared to 0-5% of healthy controls.
Why the study?
The construct validity and test-retest reliability of Endurance Shuttle Tests as outcome measures for fatigability of remaining motor functions across the severity spectrum in SMA needed to be determined.
Are Endurance Shuttle Tests valid and reliable for measuring fatigability in patients with spinal muscular atrophy?
Cross-Sectional (n=101)
Yes
Are Endurance Shuttle Tests valid and reliable for measuring fatigability in patients with spinal muscular atrophy?
Endurance Shuttle Tests are valid and reliable outcome measures for assessing fatigability across the severity spectrum of spinal muscular atrophy.
Supports Endurance Shuttle Tests for SMA fatigability assessment; hypothesis-generating pending longitudinal validation.
BACKGROUND: To determine construct validity and test-retest reliability of Endurance Shuttle Tests as outcome measures for fatigability of remaining motor functions in children and adults with Spinal Muscular Atrophy (SMA) across the severity spectrum. RESULTS: We assessed the Endurance Shuttle - Nine Hole Peg Test (ESNHPT), - Box and Block Test (ESBBT) and - Walk Test (ESWT) in 61 patients with SMA types 2-4, 25 healthy controls (HC) and 15 disease controls (DC). Convergent validity, discriminative validity and test-retest reliability were investigated. Additionally, we compiled the Endurance Shuttle Combined Score (ESTCS) by selecting the most relevant endurance test of each individual. 54, 70 and 73% of patients with SMA demonstrated increased fatigability on the ESNHPT, ESBBT and the ESWT. Endurance response in SMA was characterized by a decrease in muscle strength, an increase in muscle fatigue and an increase in motor adaptions, thereby confirming convergent validity. Patients with SMA showed increased drop-out rates and a shorter endurance time compared to HC and DC demonstrating good discriminative validity. Test-retest reliability was moderate to excellent (ICC's ranging from .78 to .91) with a trend towards better performance on retest. The ESTCS increased sample size and drop-out rate up to 100 and 85%. CONCLUSIONS: Fatigability is an important additional dimension of physical impairments across the severity spectrum in children and adults with SMA. The EST's are reliable and valid to document fatigability of walking, proximal- and distal arm function in SMA and thus are promising outcome measures for use in clinical trials.
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Bartels et al. (2020) conducted a cross-sectional in Spinal Muscular Atrophy (SMA) (n=101). Endurance Shuttle Tests vs. Healthy and disease controls was evaluated on Drop-out rate on the Endurance Shuttle - Nine Hole Peg Test (ESNHPT) (p=<0.001). The Endurance Shuttle Tests demonstrated moderate to excellent test-retest reliability and good discriminative validity, with 54% to 73% of SMA patients showing increased fatigability and dropping out compared to 0-5% of healthy controls.
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