{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T08:38:25Z","timestamp":1793263105484,"version":"4.5.0"},"reference-count":60,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2026,5,16]],"date-time":"2026-05-16T00:00:00Z","timestamp":1778889600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,5,16]],"date-time":"2026-05-16T00:00:00Z","timestamp":1778889600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"name":"Public Dental Service, Region V\u00e4stra G\u00f6taland, Sweden","id":[{"id":"https:\/\/ror.org\/00a4x6777","id-type":"ROR","asserted-by":"crossref"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Neuromuscular Diseases"],"published-print":{"date-parts":[[2026,11]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Some symptoms of orofacial dysfunction, such as dysphagia and dysarthria, are commonly encountered in congenital or childhood-onset neuromuscular diseases (NMD), while others, such as reduced saliva control and difficulties using facial expressions, are not as well researched. Overall, there is a lack of knowledge regarding the extents to which different orofacial functions are affected in persons with NMD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To identify orofacial dysfunction profiles at group level for congenital or childhood-onset NMD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This database study included 206 individuals in the age range of 3\u201348 years with 21 different congenital or childhood-onset NMD, categorized as anterior horn cell diseases, neuropathies and myopathies. Orofacial dysfunction profiles from the Nordic Orofacial Test - Screening were extracted from a national database of oral health and orofacial functions in rare health conditions.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>In the study population, 70% presented with signs of orofacial dysfunction. Neuropathies had the lowest rate of orofacial dysfunctions (25%), followed by myopathies (72%). Anterior horn cell diseases had the highest rate of orofacial dysfunctions (82%). In all the groups, there were individuals without orofacial dysfunction, except for the subgroups of Spinal Muscular Atrophy type 1 and Duchenne Muscular Dystrophy with age range of 19\u201349 years in which all the participants (100%) demonstrated orofacial dysfunction according to NOT-S.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>This study identifies a considerable proportion of individuals with congenital and childhood-onset NMD exhibiting signs of orofacial dysfunction, indicating the need for further investigation. It also shows to what extents different orofacial functions are affected in subgroups of NMD.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/22143602261452338","type":"journal-article","created":{"date-parts":[[2026,5,16]],"date-time":"2026-05-16T13:28:57Z","timestamp":1778938137000},"page":"1430-1445","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Orofacial dysfunction in persons with congenital or childhood-onset neuromuscular disorders"],"prefix":"10.1177","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8975-8165","authenticated-orcid":false,"given":"Lisa","family":"Bengtsson-Stelzer","sequence":"first","affiliation":[{"name":"The Sahlgrenska Academy"},{"name":"University of Gothenburg"},{"name":"V\u00e4stra G\u00f6taland Public Dental Service"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna-Karin","family":"Kroksmark","sequence":"additional","affiliation":[{"name":"The Sahlgrenska Academy"},{"name":"University of Gothenburg"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christina","family":"Persson","sequence":"additional","affiliation":[{"name":"The Sahlgrenska Academy"},{"name":"University of Gothenburg"},{"name":"Sahlgrenska University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lisa","family":"Tuomi","sequence":"additional","affiliation":[{"name":"The Sahlgrenska Academy"},{"name":"University of Gothenburg"},{"name":"Sahlgrenska University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne-Berit","family":"Ekstr\u00f6m","sequence":"additional","affiliation":[{"name":"The Sahlgrenska Academy"},{"name":"University of Gothenburg"},{"name":"Queen Silvia Children\u2019s Hospital"},{"name":"Sahlgrenska University Hospital"},{"name":"Sahlgrenska University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,5,16]]},"reference":[{"key":"e_1_3_6_2_2","volume-title":"Muscle Disorders in Childhood","author":"Dubowitz V","year":"1995","unstructured":"Dubowitz V. 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