{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,1]],"date-time":"2026-08-01T05:43:03Z","timestamp":1785562983067,"version":"3.56.0"},"reference-count":36,"publisher":"SAGE Publications","issue":"3","license":[{"start":{"date-parts":[[2023,5,2]],"date-time":"2023-05-02T00:00:00Z","timestamp":1682985600000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JND"],"published-print":{"date-parts":[[2023,5,2]]},"abstract":"<jats:p>Background: Recent pharmaceutical breakthroughs in neuromuscular diseases may considerably change the prognosis and natural history these diseases. The ability to measure clinically relevant outcomes such as motor function is critical for the assessment of therapeutics and the follow up of individuals. The Motor Function Measure (MFM) is a quantitative scale designed to measure motor function in adult and children with neuromuscular disease (NMD). Objective: The objective of this study is to assess the quality and level of evidence of the MFM\u2019s published measurement properties by completing a systematic review of the validation and responsiveness studies of the MFM20 (a 20-item version of MFM adapted for children 2 to 6 years of age) and the MFM32 (the original 32 item version), in all NMDs and in specific diseases. Methods: A search for MFM responsiveness and MFM validation studies was completed in February 2023 in EMBASE, MEDLINE, SCOPUS and Web of Science databases. The PRISMA guidelines and the COSMIN manual for systematic reviews were followed for databases searches, articles screening and selection, study quality and measurement properties evaluation. Results: 49 studies were included in analysis. In studies including individuals with all NMDs, MFM\u2019s internal consistency, reliability, convergent validity, construct validity and responsiveness were rated as sufficient with a high quality of evidence. Structural validity was rated sufficient with a moderate quality of evidence In SMA in particular, MFM\u2019s reliability, internal consistency, convergent validity, discriminant validity and responsiveness are sufficient with a high quality of evidence. More studies would be required to assess specific measurement properties in different diseases. MFM32\u2019s minimal clinically relevant difference has been defined between 2 and 6%. Conclusion: MFM\u2019s structural validity, internal consistency, reliability, construct validity, convergent validity and responsiveness have been verified with moderate to high level of evidence.<\/jats:p>","DOI":"10.3233\/jnd-230001","type":"journal-article","created":{"date-parts":[[2023,4,25]],"date-time":"2023-04-25T12:35:34Z","timestamp":1682426134000},"page":"301-314","source":"Crossref","is-referenced-by-count":13,"title":["Psychometric Characteristics of the Motor Function Measure in Neuromuscular Diseases: A Systematic Review1"],"prefix":"10.1177","volume":"10","author":[{"given":"Shams","family":"Ribault","sequence":"first","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"},{"name":"Pathophysiology and Genetics of Neuron and Muscle, CNRS UMR 5261, INSERM U1315, Universit\u00e9 Lyon1, Facult\u00e9 de M\u00e9decine Lyon Est, Lyon, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pascal","family":"Rippert","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"},{"name":"Hospices Civils de Lyon, P\u00f4le Sant\u00e9 Publique, Service Recherche et Epid\u00e9miologie Clinique, Lyon F-69003"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Minal","family":"Jain","sequence":"additional","affiliation":[{"name":"Rehabilitation Medicine Department, National Institutes of Health, Bethesda, MD, US"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laure","family":"Le Goff","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominique Vincent","family":"Genod","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aur\u00e9lie","family":"Barriere","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Berruyer","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Camille","family":"Garde","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Tinat","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christelle","family":"Pons","sequence":"additional","affiliation":[{"name":"SSR p\u00e9diatrique, fondation Ildys, rue Alain-Colas, 29200 Brest, France; Service de m\u00e9decine physique et de r\u00e9adaptation, CHRU de Brest, Brest, France; Laboratoire de traitement de l\u2019information m\u00e9dicale (LaTIM), inserm U1101, universit\u00e9 Bretagne Occidentale, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carole","family":"Vuillerot","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon, H\u00f4pital Femme M\u00e8re Enfant, Service de m\u00e9decine physique et r\u00e9adaptation p\u00e9diatrique, Bron cedex F-69677"},{"name":"Pathophysiology and Genetics of Neuron and Muscle, CNRS UMR 5261, INSERM U1315, Universit\u00e9 Lyon1, Facult\u00e9 de M\u00e9decine Lyon Est, Lyon, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","reference":[{"issue":"1","key":"10.3233\/JND-230001_ref1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.3233\/JND-190424","article-title":"Advances in treatment of spinal muscular atrophy \u2013new phenotypes, new challenges, new implications for care","volume":"7","author":"Schorling","year":"2020","journal-title":"J Neuromuscul Dis"},{"issue":"4","key":"10.3233\/JND-230001_ref2","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1016\/j.nmd.2014.01.003","article-title":"Hammersmith Functional Motor Scale and Motor Function Measure-20 in non ambulant SMA patients","volume":"24","author":"Mazzone","year":"2014","journal-title":"Neuromuscul Disord NMD"},{"issue":"12","key":"10.3233\/JND-230001_ref3","doi-asserted-by":"crossref","first-page":"1499","DOI":"10.1177\/0883073811420294","article-title":"Validation of the Expanded Hammersmith Functional Motor Scale in spinal muscular atrophy type II and III","volume":"26","author":"Glanzman","year":"2011","journal-title":"J Child Neurol"},{"issue":"11","key":"10.3233\/JND-230001_ref4","doi-asserted-by":"crossref","first-page":"712","DOI":"10.1016\/j.nmd.2010.06.014","article-title":"North Star Ambulatory Assessment, 6-minute walk test and timed items in ambulant boys with Duchenne muscular dystrophy","volume":"20","author":"Mazzone","year":"2010","journal-title":"Neuromuscul Disord NMD"},{"key":"10.3233\/JND-230001_ref5","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1016\/0021-9681(60)90105-3","article-title":"Maintenance of ambulation in childhood muscular dystrophy","volume":"12","author":"Vignos","year":"1960","journal-title":"J Chronic Dis"},{"issue":"3","key":"10.3233\/JND-230001_ref6","doi-asserted-by":"crossref","first-page":"186","DOI":"10.1002\/mus.880040304","article-title":"Clinical trial in Duchenne dystrophy. 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