Abstract
Increasing evidence suggests that high-intensity training (HIT) is a time-efficient exercise strategy to improve fitness. HIT has never been explored in neuromuscular diseases, likely because it may seem counterintuitive. A single session of high-intensity exercise has been studied without signs of muscle damage in facioscapulohumeral muscular dystrophy type 1 (FSHD1). We aimed to determine whether HIT is safe and effective in FSHD1 in a randomized, controlled parallel study. Untrained adults with genetically verified FSHD1 (n = 13) able to perform cycle-ergometer exercise were randomized to 8 weeks of supervised HIT (n = 6) (3 × 10-min cycle-ergometer-HIT/week) or 8 weeks of usual care (n = 7). Following this, all participants performed 8 weeks of unsupervised HIT (3 × 10-min cycle-ergometer-HIT/week). Primary outcome was fitness, maximal oxygen uptake/min/kg body weight. Furthermore, workload, 6-min walk distance, 5-time sit-to-stand time, muscle strength, and daily activity levels were measured. Pain, fatigue, and plasma-CK were monitored. Twelve patients completed the randomized part of the study. Plasma-CK levels and pain scores were unaffected by HIT. Supervised HIT improved fitness (3.3 ml O2/min/kg, CI 1.2–5.5, P < 0.01, n = 6, NNT = 1.4). Unsupervised HIT also improved fitness (2.0 ml O2/min/kg, CI 0.1–3.9, P = 0.04, n = 4). There was no training effect on other outcomes. Patients preferred HIT over strength and moderate-intensity aerobic training. It may seem counterintuitive to perform HIT in muscular dystrophies, but this RCT shows that regular HIT is safe, efficacious, and well liked by moderately affected patients with FSHD1, which suggests that HIT is a feasible method for rehabilitating patients with FSHD1.
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Acknowledgements
We thank Søren P. Andersen MSc, Human Physiology University of Copenhagen, for his help with designing the training program. Dr. Andersen contributed to the design of study, analysis, acquisition and interpretation of data, and drafting the manuscript. Dr. Heje contributed to the design of study, analysis, acquisition of data, and critical revision of the manuscript. Buch BSc contributed to the analysis, acquisition of data, and critical revision of the manuscript. Dr. Vissing contributed to the design of study, interpretation of data, and critical revision of the manuscript. We thank Aase and Einar Danielsens Foundation, Augustinus Foundation, and AP Moeller Foundation for financial support. Dr. Andersen reports no disclosure. Dr. Heje reports no disclosure. Buch BSc reports no disclosure. Dr. Vissing has received research and travel support and speaker honoraria from Genzyme/Sanofi and Ultragenyx Pharmaceuticals, and has acted as consultant on advisory boards for Genzyme/Sanofi, Sarepta, Lundbeck, Ultragenyx Pharmaceuticals, NOVO Nordisk and Alexion Pharmaceuticals within the last 3 years.
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The authors declare that they have no competing interests.
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The study was conducted in accordance with the Helsinki declaration of 1964, approved by the Ethics Committee of Copenhagen (H-4-2014-035), and all participants gave informed written consent to participate prior inclusion in the study.
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Statistical analyses were conducted by Grete Andersen, MD with advice from the Section of Biostatistics, Department of Public Health, University of Copenhagen.
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Andersen, G., Heje, K., Buch, A.E. et al. High-intensity interval training in facioscapulohumeral muscular dystrophy type 1: a randomized clinical trial. J Neurol 264, 1099–1106 (2017). https://doi.org/10.1007/s00415-017-8497-9
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DOI: https://doi.org/10.1007/s00415-017-8497-9