PMID- 32671231 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20240330 IS - 2398-502X (Print) IS - 2398-502X (Electronic) IS - 2398-502X (Linking) VI - 5 DP - 2020 TI - Measuring the effects of exercise in neuromuscular disorders: a systematic review and meta-analyses. PG - 84 LID - 10.12688/wellcomeopenres.15825.1 [doi] LID - 84 AB - Background: The benefit and safety of exercise training for patients with neuromuscular disorders (NMDs) has long been a contentious topic. This is, in part, due to recognised challenges associated with rare diseases including small and heterogenous patient populations. We performed a systematic review and meta-analyses to evaluate the effectiveness and safety of interventional exercise and establish minimal clinically important differences (MCID) in outcomes to facilitate clinical interpretation. Methods: We searched six databases from inception to Mar 2018. Aerobic, strength, and combined (aerobic and strength) intervention were eligible. Meta-analyses compared outcomes at baseline with those after at least six weeks (before-after exercise within individuals). A further meta-analysis compared outcomes before-after exercise between groups (exercise training versus usual care). Disease heterogeneity was explored using a random effect model. This study was registered (PROSPERO, CRD42018102183). An interactive database was developed to facilitate full interrogations of data. Results: We identified 130 articles describing 1,805 participants with 35 different forms of NMD. Of these studies, 76 were suitable for meta-analyses. Within group and between group meta-analyses detected an increase in peak aerobic capacity (p=0.04), and peak power (p=0.01). Six-minute walk test (p=0.04), sit-to-stand (STS) (repetitions) (p=0.03), STS (seconds) (p=0.04), rise from supine (p=0.008), SF-36 (p=0.0003), fatigue severity (p=<0.0001), citrate synthase (p=0.0002), central nuclei (p=0.04), type 1 (p=0.002) and type II muscle fibre area (p=0.003), were only able to detect change within group meta-analyses. Substantial I (2) statistic heterogeneity was revealed for STS (seconds) ( I(2)=58.5%; p=0.04) and citrate synthase ( I(2)=70.90%; p=0.002), otherwise heterogeneity for all outcomes was low. No study-related serious adverse events were reported nor significant increases in creatine kinase. Conclusions: Exercise training in patients with NMDs appears to cause no harm across a range of outcomes. With the emergence of new therapeutic strategies, defining MCID is vital in informing future clinical trial design. CI - Copyright: (c) 2020 Stefanetti RJ et al. FAU - Stefanetti, Renae J AU - Stefanetti RJ AUID- ORCID: 0000-0001-6402-6435 AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Blain, Alasdair AU - Blain A AUID- ORCID: 0000-0002-3712-5283 AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Jimenez-Moreno, Cecilia AU - Jimenez-Moreno C AUID- ORCID: 0000-0002-0488-9631 AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Errington, Linda AU - Errington L AD - Faculty of Medical Sciences, Newcastle University, Newcastle upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Ng, Yi Shiau AU - Ng YS AUID- ORCID: 0000-0002-7591-2034 AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - McFarland, Robert AU - McFarland R AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Turnbull, Doug M AU - Turnbull DM AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. FAU - Newman, Jane AU - Newman J AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. AD - NIHR Newcastle Biomedical Research Centre, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, Tyne and Wear, NE4 5PL, UK. FAU - Gorman, Grainne S AU - Gorman GS AD - Wellcome Centre for Mitochondrial Research, Newcastle University, Newcastle Upon Tyne, Tyne and Wear, NE2 4HH, UK. LA - eng GR - WT_/Wellcome Trust/United Kingdom PT - Systematic Review DEP - 20200504 PL - England TA - Wellcome Open Res JT - Wellcome open research JID - 101696457 PMC - PMC7331112 OTO - NOTNLM OT - Neuromuscular OT - aerobic OT - exercise training OT - functional exercise OT - minimal important clinical difference OT - muscle biopsies OT - outcome measures OT - strength COIS- No competing interests were disclosed. EDAT- 2020/07/17 06:00 MHDA- 2020/07/17 06:01 PMCR- 2020/05/04 CRDT- 2020/07/17 06:00 PHST- 2020/04/23 00:00 [accepted] PHST- 2020/07/17 06:00 [entrez] PHST- 2020/07/17 06:00 [pubmed] PHST- 2020/07/17 06:01 [medline] PHST- 2020/05/04 00:00 [pmc-release] AID - 10.12688/wellcomeopenres.15825.1 [doi] PST - epublish SO - Wellcome Open Res. 2020 May 4;5:84. doi: 10.12688/wellcomeopenres.15825.1. eCollection 2020.