{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T16:01:26Z","timestamp":1791820886897,"version":"4.3.4"},"reference-count":26,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2019,10,30]],"date-time":"2019-10-30T00:00:00Z","timestamp":1572393600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100000156","name":"Fonds de Recherche du Qu\u00e9bec - Sant\u00e9","doi-asserted-by":"publisher","award":["31011"],"award-info":[{"award-number":["31011"]}],"id":[{"id":"10.13039\/501100000156","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2019,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>There is currently little evidence regarding oculopharyngeal muscular dystrophy (OPMD) disease burden reported by patients. In this study we aim to elicit direct patient input regarding OPMD disease burden.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We conducted semistructured interviews with 25 participants with genetically confirmed OPMD and a wide range of disease duration (15\u2009\u00b1\u20098\u2009years). Using the Framework Technique, themes and categories were then extracted.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Analyses revealed 7 themes (physical impact, mental impact, social impact, perception of progression, treatment perceptions, coping strategies, and access to disease information), encompassing 27 categories of OPMD disease burden. The most frequent categories were related to dysphagia, coping strategies for dysphagia, and impaired mobility.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>This study demonstrates the importance of considering, when providing clinical care, the broad range of coping strategies patients use to deal with OPMD symptoms, especially dysphagia, to properly assess limitations and monitor real disease progression.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mus.26712","type":"journal-article","created":{"date-parts":[[2019,9,18]],"date-time":"2019-09-18T03:16:17Z","timestamp":1568776577000},"page":"724-731","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Patient\u2010reported disease burden in oculopharyngeal muscular dystrophy"],"prefix":"10.1002","volume":"60","author":[{"given":"Nicolette S.","family":"Kurtz","sequence":"first","affiliation":[{"name":"Department of Neurology University of New Mexico Health Sciences Center  Albuquerque New Mexico"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Cote","sequence":"additional","affiliation":[{"name":"Centre de recherche Charles\u2010Le Moyne\u2013Saguenay\u2010Lac\u2010Saint\u2010Jean sur les innovations en sant\u00e9 Sherbrooke University  Sherbrooke Qu\u00e9bec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chad","family":"Heatwole","sequence":"additional","affiliation":[{"name":"Department of Neurology University of Rochester  Rochester New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cynthia","family":"Gagnon","sequence":"additional","affiliation":[{"name":"Centre de recherche Charles\u2010Le Moyne\u2013Saguenay\u2010Lac\u2010Saint\u2010Jean sur les innovations en sant\u00e9 Sherbrooke University  Sherbrooke Qu\u00e9bec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Youssof","sequence":"additional","affiliation":[{"name":"Department of Neurology University of New Mexico Health Sciences Center  Albuquerque New Mexico"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,10,30]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0298-164"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM196212202672501"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24932"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.23529"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e318260cbe6"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.25957"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2015.12.009"},{"key":"e_1_2_9_9_1","first-page":"35","article-title":"Living with oculopharyngeal muscular dystrophy: a phenomenological study","volume":"30","author":"Krause\u2010Bachand J","year":"2008","journal-title":"Can J Neurosci Nurs."},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1186\/1477-7525-4-79"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1586\/14737175.4.6.985"},{"key":"e_1_2_9_12_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. 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