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Previous systematic reviews have focussed on single diseases, whereas this study systematically reviewed the collective evidence for respiratory muscle training in children and adults with any neuromuscular disease.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Seven databases were searched for randomised controlled trials. Three reviewers independently reviewed eligibility, extracted characteristics, results, determined risk of bias and combined results using narrative synthesis and meta-analysis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>37 studies (40 publications from 1986\u20132021, n=951 participants) were included. Respiratory muscle training improved forced vital capacity (standardised mean difference (SMD) 0.40 (95% confidence interval 0.12\u20130.69)), maximal inspiratory (SMD 0.53 (0.21\u20130.85)) and maximal expiratory pressure (SMD 0.70 (0.35\u20131.04)) compared to control (usual care, sham or alternative treatment). No impact on cough, dyspnoea, voice, physical capacity or quality of life was detected. There was high degree of variability between studies.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>Study heterogeneity (children and adults, different diseases, interventions, dosage and comparators) suggests that the results should be interpreted with caution. Including all neuromuscular diseases increased the evidence pool and tested the intervention overall.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Respiratory muscle training improves lung volumes and respiratory muscle strength in neuromuscular disease, but confidence is tempered by limitations in the underlying research.<\/jats:p><\/jats:sec>","DOI":"10.1183\/16000617.0065-2022","type":"journal-article","created":{"date-parts":[[2022,11,30]],"date-time":"2022-11-30T08:55:45Z","timestamp":1669798545000},"page":"220065","update-policy":"https:\/\/doi.org\/10.1183\/ers-crossmark-policy","source":"Crossref","is-referenced-by-count":52,"title":["Respiratory muscle training in neuromuscular disease: a systematic review and meta-analysis"],"prefix":"10.1183","volume":"31","author":[{"given":"Kathryn","family":"Watson","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thorlene","family":"Egerton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1847-4266","authenticated-orcid":false,"given":"Nicole","family":"Sheers","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Retica","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebekah","family":"McGaw","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Talia","family":"Clohessy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5834-4047","authenticated-orcid":false,"given":"Penny","family":"Webster","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2543-8722","authenticated-orcid":false,"given":"David J.","family":"Berlowitz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"81","published-online":{"date-parts":[[2022,11,29]]},"reference":[{"key":"2024102404065755000_31.166.220065.1","doi-asserted-by":"publisher","DOI":"10.1378\/chest.118.5.1390"},{"key":"2024102404065755000_31.166.220065.2","doi-asserted-by":"publisher","DOI":"10.1177\/0269215513512215"},{"key":"2024102404065755000_31.166.220065.3","doi-asserted-by":"crossref","first-page":"1522","DOI":"10.1097\/00005768-199911000-00005","article-title":"Respiratory muscle training in neuromuscular disease: long-term effects on strength and load perception","volume":"31","author":"Gozal","year":"1999","journal-title":"Med Sci Sports Exerc"},{"key":"2024102404065755000_31.166.220065.4","doi-asserted-by":"crossref","first-page":"2","DOI":"10.1097\/01823246-201324040-00022","article-title":"Does inspiratory muscle training improve lung function, inspiratory muscle strength or inspiratory muscle endurance in people with Duchenne muscular dystrophy?","volume":"24","author":"Anderson","year":"2013","journal-title":"Cardiopulm Phys Ther J"},{"key":"2024102404065755000_31.166.220065.5","first-page":"CD008507","article-title":"Respiratory muscle training for cervical spinal cord injury","volume":"7","author":"Berlowitz","year":"2013","journal-title":"Cochrane Database Syst Rev"},{"key":"2024102404065755000_31.166.220065.6","first-page":"CD003792","article-title":"Inspiratory muscle training for asthma","volume":"9","author":"Silva","year":"2013","journal-title":"Cochrane Database Syst Rev"},{"key":"2024102404065755000_31.166.220065.7","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1016\/j.nmd.2017.03.009","article-title":"Inspiratory muscle training for children and adolescents with neuromuscular diseases: a systematic review","volume":"27","author":"Human","year":"2017","journal-title":"Neuromuscul Disord"},{"key":"2024102404065755000_31.166.220065.8","doi-asserted-by":"publisher","DOI":"10.1378\/chest.12-1442"},{"key":"2024102404065755000_31.166.220065.9","doi-asserted-by":"crossref","first-page":"98","DOI":"10.1016\/j.rmed.2018.01.012","article-title":"Airway clearance techniques in neuromuscular disorders: a state of the art review","volume":"136","author":"Chatwin","year":"2018","journal-title":"Respir Med"},{"key":"2024102404065755000_31.166.220065.10","doi-asserted-by":"publisher","DOI":"10.1097\/00002060-199106000-00004"},{"key":"2024102404065755000_31.166.220065.11","doi-asserted-by":"publisher","DOI":"10.2174\/1874306400903010123"},{"key":"2024102404065755000_31.166.220065.12","unstructured":"Higgins JPY , Green S , eds. 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