{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,2]],"date-time":"2026-08-02T02:11:00Z","timestamp":1785636660719,"version":"3.56.0"},"reference-count":51,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2021,5,2]],"date-time":"2021-05-02T00:00:00Z","timestamp":1619913600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100004243","name":"Prinses Beatrix Spierfonds","doi-asserted-by":"publisher","award":["W.OR12\u201022"],"award-info":[{"award-number":["W.OR12\u201022"]}],"id":[{"id":"10.13039\/501100004243","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2021,7]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background and objectives<\/jats:title><jats:p>Facioscapulohumeral muscular dystrophy (FHSD) is a debilitating inherited muscle disease for which various therapeutic strategies are being investigated. Thus far, little attention has been given in FSHD to the development of scientifically sound outcome measures fulfilling regulatory authority requirements. The aim of this study was to design a patient\u2010reported Rasch\u2010built interval scale on activity and participation for FSHD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A pre\u2010phase FSHD\u2010Rasch\u2010built overall disability scale (pre\u2010FSHD\u2010RODS; consisting of 159 activity\/participation items), based on the World Health Organization international classification of disease\u2010related functional consequences was completed by 762 FSHD patients (Netherlands: <jats:italic>n<\/jats:italic>\u00a0=\u00a0171; UK: <jats:italic>n<\/jats:italic>\u00a0=\u00a0287; United States: <jats:italic>n<\/jats:italic>\u00a0=\u00a0221; France: <jats:italic>n<\/jats:italic>\u00a0=\u00a052; Australia: <jats:italic>n<\/jats:italic>\u00a0=\u00a032). A proportion of the patient cohort completed it twice (<jats:italic>n<\/jats:italic>\u00a0=\u00a0230; interval 2\u20134\u00a0weeks; reliability studies). The pre\u2010FSHD\u2010RODS was subjected to Rasch analyses to create a model fulfilling its requirements. Validity studies were performed through correlation with the motor function measure.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The pre\u2010FSHD\u2010RODS did not meet the Rasch model expectations. Based on determinants such as misfit statistics and misfit residuals, differential item functioning, and local dependency, we systematically removed items until a final 38\u2010inquiry (originating from 32 items; six items split) FSHD\u2010RODS was constructed achieving Rasch model expectations. Adequate test\u2010retest reliability and (cross\u2010cultural and external) validity scores were obtained.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The FSHD\u2010RODS is a disease\u2010specific interval measure suitable for detecting activity and participation restrictions in patients with FSHD with good item\/person reliability and validity scores. The use of this scale is recommended in the near future, to determine the functional deterioration slope in FSHD per year as a preparation for the upcoming clinical intervention trials in FSHD.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.14863","type":"journal-article","created":{"date-parts":[[2021,4,10]],"date-time":"2021-04-10T19:10:59Z","timestamp":1618081859000},"page":"2339-2348","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["The facioscapulohumeral muscular dystrophy Rasch\u2010built overall disability scale (FSHD\u2010RODS)"],"prefix":"10.1111","volume":"28","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8487-9478","authenticated-orcid":false,"given":"Karlien","family":"Mul","sequence":"first","affiliation":[{"name":"Department of Neurology Donders Institute for Brain, Cognition and Behaviour Radboud University Medical Center Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2413-8218","authenticated-orcid":false,"given":"Tatiana","family":"Hamadeh","sequence":"additional","affiliation":[{"name":"Department of Neurology Cura\u00e7ao Medical Center Willemstad Cura\u00e7ao"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corinne G. 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