{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,28]],"date-time":"2026-01-28T08:19:02Z","timestamp":1769588342733,"version":"3.49.0"},"reference-count":30,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2016,3,1]],"date-time":"2016-03-01T00:00:00Z","timestamp":1456790400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100005202","name":"Muscular Dystrophy Association","doi-asserted-by":"crossref","award":["260590"],"award-info":[{"award-number":["260590"]}],"id":[{"id":"10.13039\/100005202","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"National Institute for Research Resources"},{"name":"National Center for Advancing Transnational Sciences"},{"name":"Clinical and Translational Sciences Awards program","award":["8UL1TR000041"],"award-info":[{"award-number":["8UL1TR000041"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2016,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>Oculopharyngeal muscular dystrophy (OPMD) causes ptosis, dysphagia, and limb weakness. Health\u2010related quality of life (HRQoL) and its relationship to physical symptoms was investigated.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>The 36\u2010item Short Form (SF\u201036) was completed by 89 participants in the U.S. OPMD Registry. Multiple hierarchical regression was used to determine the relative contributions of dysphagia severity and lower extremity functional impairment to the physical (PCS) and mental (MCS) components of the SF\u201036.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>HRQoL was reduced in OPMD compared with population norms. Lower extremity functional impairment explained a significant proportion of variance in PCS and MCS. Dysphagia symptom severity explained a moderate amount of variance only in MCS. Dysphagia symptom severity had the strongest associations with general health perception and social functioning domains.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Lower extremity functional impairment in OPMD deserves attention due to its large influence on HRQoL. Both generic and dysphagia\u2010specific measures are necessary to assess HRQoL in OPMD. <jats:italic>Muscle Nerve<\/jats:italic> <jats:bold>53<\/jats:bold>: 694\u2013699, 2016<\/jats:p><\/jats:sec>","DOI":"10.1002\/mus.24932","type":"journal-article","created":{"date-parts":[[2015,10,10]],"date-time":"2015-10-10T01:50:39Z","timestamp":1444441839000},"page":"694-699","source":"Crossref","is-referenced-by-count":17,"title":["The relationship between physical symptoms and health\u2010related quality of life in oculopharyngeal muscular dystrophy"],"prefix":"10.1002","volume":"53","author":[{"given":"Sarah","family":"Youssof","sequence":"first","affiliation":[{"name":"Department of Neurology University of New Mexico Health Sciences Center, MSC 10 5620 Albuquerque New Mexico 87131 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2016,3]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM196212202672501"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0298-164"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/0960-8966(93)90119-5"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0033291798006667"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-011-6062-5"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1472-8206.2004.00234.x"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1574-0064(00)80045-1"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000001336"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.23320"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/mt.2013.155"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1044\/1092-4388(2010\/09-0068)"},{"key":"e_1_2_6_13_1","first-page":"269","article-title":"The NIH Office of Rare Diseases Research Patient Registry Standard: a report from the University of New Mexico's Oculopharyngeal Muscular Dystrophy Patient Registry","volume":"2013","author":"Daneshvari S","year":"2013","journal-title":"AMIA Annu Symp Proc"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-199206000-00002"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00455-001-0109-1"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000000041"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2014.11.010"},{"key":"e_1_2_6_18_1","unstructured":"National Institute of Neurological Disorders and Stroke (NINDS).User Manual for the Quality of Life in Neurological Disorders (Neuro\u2010QOL) Measures version 1.0 September 2010. Available athttp:\/\/www.neuroqol.org\/HowDoI\/UserManual\/User%20Manual\/Neuro-QOL_UserManual.pdf\/. 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