{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,11]],"date-time":"2026-09-11T12:33:09Z","timestamp":1789129989588,"version":"build-2803163510"},"reference-count":36,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2023,9,11]],"date-time":"2023-09-11T00:00:00Z","timestamp":1694390400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2024,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>In this review we sought to characterize the lived experience of people living with FSHD (pwFSHD) to help clinicians to orient their services to the needs of these individuals.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Five electronic databases were systematically searched for qualitative research studies containing quotations from pwFSHD. ENhancing Transparency in REporting the Synthesis of Qualitative research and Preferred Reporting Items for Systematic Reviews and Meta\u2010Analyses guidelines informed the methodology. Study quality was assessed using the Critical Appraisal Skills Programme Checklist tool, which measures the methodological quality of qualitative research. Data extracted from included studies were analyzed using thematic synthesis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Ninety\u2010nine pwFSHD took part in the six studies included in this review \u2010 from research teams based in two countries. Five descriptive themes emerged: \u201cEngaging with life as symptoms progress\u201d; \u201cThe emotional journey\u201d; \u201cA family burden to bear\u201d; \u201cSocial connection and disconnection\u201d; and \u201cTension between visibility and invisibility.\u201d From these, two analytical themes were derived: \u201cThe emotional challenge of continuing and intensifying adaptation\u201d and \u201cThe relational burden of rare disease.\u201d<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>The lived experience of pwFSHD is characterized by physical, emotional, and social challenges that impact on engagement with life, particularly as symptoms progress. Further research is needed to provide a fuller understanding of the experience of pain in FSHD and of the lived experience of FSHD across cultures.<\/jats:p><\/jats:sec>","DOI":"10.1002\/mus.27964","type":"journal-article","created":{"date-parts":[[2023,9,11]],"date-time":"2023-09-11T08:04:30Z","timestamp":1694419470000},"page":"7-17","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["The lived experience of facioscapulohumeral muscular dystrophy: A systematic review and synthesis of the qualitative literature"],"prefix":"10.1002","volume":"69","author":[{"given":"Roisin","family":"Murray","sequence":"first","affiliation":[{"name":"Psychology Service for Older People Western Health and Social Care Trust  Londonderry UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clare M.","family":"Donnelly","sequence":"additional","affiliation":[{"name":"Psychological Therapies Service Northern Health and Social Care Trust  Antrim UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kent D.","family":"Drescher","sequence":"additional","affiliation":[{"name":"National Center for PTSD VA Palo Alto Healthcare System  Palo Alto California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8456-9154","authenticated-orcid":false,"given":"Christopher D.","family":"Graham","sequence":"additional","affiliation":[{"name":"Psychology Service for Older People Western Health and Social Care Trust  Londonderry UK"},{"name":"School of Psychological Sciences and Health University of Strathclyde  Glasgow UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,9,11]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.20522"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molmed.2011.01.001"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.2522\/ptj.20070104"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000008123"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.23529"},{"key":"e_1_2_12_7_1","first-page":"1916","article-title":"Facioscapulohumeral muscular dystrophy","volume":"22","author":"Statland JM","year":"2016","journal-title":"Continuum"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000001783"},{"key":"e_1_2_12_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-011-6062-5"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1177\/0269215513511340"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24487"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1080\/09638288.2022.2092779"},{"key":"e_1_2_12_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24079"},{"key":"e_1_2_12_14_1","doi-asserted-by":"publisher","DOI":"10.2340\/16501977-0482"},{"key":"e_1_2_12_15_1","first-page":"355","article-title":"Predictors of health\u2010related quality of life in adult ambulatory independence neuromuscular disease patients","volume":"14","author":"Oksuz C","year":"2009","journal-title":"Neurosci J."},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpsychores.2012.09.012"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.1037\/rep0000092"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2021.01.007"},{"key":"e_1_2_12_19_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1600-0404.2003.00185.x"},{"key":"e_1_2_12_20_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0033291722000083"},{"key":"e_1_2_12_21_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000001008"},{"key":"e_1_2_12_22_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2288-12-181"},{"key":"e_1_2_12_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijsu.2010.02.007"},{"issue":"8","key":"e_1_2_12_24_1","article-title":"Checking reference lists to find additional studies for systematic reviews","volume":"2011","author":"Horsley T","year":"2011","journal-title":"Cochrane Database Syst Rev."},{"key":"e_1_2_12_25_1","doi-asserted-by":"publisher","DOI":"10.1177\/1049732312452938"},{"key":"e_1_2_12_26_1","unstructured":"Critical Appraisal Skills Program.CASP qualitative checklist. 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