{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,5]],"date-time":"2026-05-05T03:53:38Z","timestamp":1777953218573,"version":"3.51.4"},"reference-count":24,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2026,6]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction\/Aims<\/jats:title>\n                    <jats:p>Facioscapulohumeral muscular dystrophy (FSHD) is a muscle disease that leads, among other manifestations, to facial weakness. This weakness can severely impact communication and quality of life, yet it remains under\u2010researched with limited objective clinical measures. Current manual scoring methods are subjective and exhibit suboptimal inter\u2010observer agreement. This study aimed to develop and validate a deep learning\u2010based facial weakness score (DLFWS) as an objective clinical outcome measure for facial weakness in FSHD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>One hundred and twenty\u2010two genetically confirmed FSHD patients and 56 controls were recruited. Sixty\u2010four patients had a 5\u2010year follow\u2010up visit. Video recordings of participants performing seven facial exercises, each repeated three times, were analyzed using a convolutional neural network. The deep learning\u2010based facial weakness score (DLFWS) was trained by comparing start and end frames from these exercises with the manual facial weakness scores (MFWS) assigned by three experienced observers. Pearson correlation coefficients and intraclass correlation coefficients (ICC) were used to evaluate the DLFWS's performance and reliability.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The DLFWS showed a strong correlation with the MFWS across exercises, with a mean Pearson correlation of 0.79 and a maximum of 0.85 for individual exercises. The DLFWS demonstrated excellent test\u2013retest reliability, with an ICC of 0.90. Over a 5\u2010year follow\u2010up period, no significant progression of facial weakness was detected.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>The DLFWS provides a reliable and objective assessment of facial weakness in FSHD patients. This automated tool holds potential for widespread clinical and research applications, enabling standardized assessment of facial weakness in FSHD.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mus.70228","type":"journal-article","created":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T01:59:17Z","timestamp":1775008757000},"page":"1096-1102","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Development and Validation of a Deep Learning\u2010Based Facial Weakness Score for Objective Assessment in Facioscapulohumeral Muscular Dystrophy"],"prefix":"10.1002","volume":"73","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9628-5754","authenticated-orcid":false,"given":"T. G. J.","family":"Loonen","sequence":"first","affiliation":[{"name":"Radboudumc 3D\u2010Lab, Department of Oral and Maxillofacial Surgery Radboud University Nijmegen Medical Centre  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"T. C.","family":"ten Harkel","sequence":"additional","affiliation":[{"name":"Radboudumc 3D\u2010Lab, Department of Oral and Maxillofacial Surgery Radboud University Nijmegen Medical Centre  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0412-0670","authenticated-orcid":false,"given":"S. C. C.","family":"Vincenten","sequence":"additional","affiliation":[{"name":"Department of Neurology Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C. G. C.","family":"Horlings","sequence":"additional","affiliation":[{"name":"Department of Neurology Medical University Innsbruck  Innsbruck Austria"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C. H. G.","family":"Beurskens","sequence":"additional","affiliation":[{"name":"Department of Orthopedics, Section of Physical Therapy Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9277-2643","authenticated-orcid":false,"given":"S.","family":"Knuijt","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"G. W. A. M.","family":"Padberg","sequence":"additional","affiliation":[{"name":"Department of Neurology Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"B. G. M.","family":"van Engelen","sequence":"additional","affiliation":[{"name":"Department of Neurology Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"T. J. J.","family":"Maal","sequence":"additional","affiliation":[{"name":"Radboudumc 3D\u2010Lab, Department of Oral and Maxillofacial Surgery Radboud University Nijmegen Medical Centre  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8487-9478","authenticated-orcid":false,"given":"K.","family":"Mul","sequence":"additional","affiliation":[{"name":"Department of Neurology Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center  Nijmegen the Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2026,3,31]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1136\/practneurol-2015-001353"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000007013"},{"key":"e_1_2_10_4_1","volume-title":"Facioscapulohumeral Disease","author":"Padberg G. W.","year":"1982"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1590\/0004-282x-anp-2021-0034"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.23949"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1080\/09638288.2021.1973122"},{"issue":"724","key":"e_1_2_10_8_1","first-page":"339","article-title":"Discrimination of Genetic Entities in Muscular Dystrophy","volume":"11","author":"Chung C. S.","year":"1959","journal-title":"American Journal of Human Genetics"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1080\/09638288.2016.1212109"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.3109\/09638288.2016.1172673"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1093\/ptj\/76.12.1288"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1080\/09638288.2022.2066208"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1177\/026921559500900206"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.28254"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-020-10281-z"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13446"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.21671"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/1531-8249(199906)45:6<751::AID-ANA9>3.0.CO;2-M"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1109\/CVPR.2014.241"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.5201\/ipol.2013.26"},{"key":"e_1_2_10_21_1","first-page":"1","volume-title":"3rd International Conference on Learning Representations, ICLR 2015 \u2010 Conference Track Proceedings","author":"Simonyan K.","year":"2015"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.amjoto.2023.103810"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-642-35289-8"},{"key":"e_1_2_10_24_1","doi-asserted-by":"crossref","unstructured":"L. N.Smith \u201cCyclical learning rates for training neural networks \u201d(2017) in Proceedings \u2013 2017 IEEE Winter Conference on Applications of Computer Vision WACV 2017 464\u2013472.","DOI":"10.1109\/WACV.2017.58"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.3233\/JND-221624"}],"container-title":["Muscle &amp; Nerve"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/mus.70228","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/mus.70228","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/mus.70228","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T14:35:35Z","timestamp":1777905335000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/mus.70228"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3,31]]},"references-count":24,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2026,6]]}},"alternative-id":["10.1002\/mus.70228"],"URL":"https:\/\/doi.org\/10.1002\/mus.70228","archive":["Portico"],"relation":{},"ISSN":["0148-639X","1097-4598"],"issn-type":[{"value":"0148-639X","type":"print"},{"value":"1097-4598","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3,31]]},"assertion":[{"value":"2025-07-01","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-03-23","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-03-31","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}