{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:08:03Z","timestamp":1787018883920,"version":"build-2736575974"},"reference-count":41,"publisher":"SAGE Publications","issue":"5","license":[{"start":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T00:00:00Z","timestamp":1680307200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100014387","name":"ACADIA Pharmaceuticals","doi-asserted-by":"publisher","award":["N\/A"],"award-info":[{"award-number":["N\/A"]}],"id":[{"id":"10.13039\/100014387","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Child Neurol"],"published-print":{"date-parts":[[2023,4]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Rett syndrome is a rare neurodevelopmental disorder primarily affecting females. This syndrome is associated with many comorbidities and impairments related to motor function, breathing, sleep, expressive language, and repetitive hand movements. The Rett Syndrome Behaviour Questionnaire (RSBQ) is one measure used to assess changes in Rett syndrome\u2013related manifestations or core symptoms. Little is known about how caregivers think about meaningful changes in the items that make up the RSBQ scale.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This qualitative study explored how caregivers of individuals with Rett syndrome viewed changes in the symptoms covered in the RSBQ. We conducted semistructured interviews with 40 caregivers and employed thematic analysis, identifying themes using an iterative process.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Two factors characterized caregivers\u2019 thoughts about meaningful changes in Rett syndrome manifestations. First, general features of these symptoms rendered them bothersome: the extent of bother compared to other symptoms, if or how they prevented desirable behaviors and their temporal qualities. Second, caregivers evaluated the meaning of improvements by considering the decrease in bother and the potential benefits of change. Improvements had social and psychological consequences for individuals with Rett syndrome and caregivers. In addition, implications for health, fine and gross motor skills, and communication were also substantial.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/08830738231172066","type":"journal-article","created":{"date-parts":[[2023,5,10]],"date-time":"2023-05-10T13:07:13Z","timestamp":1683724033000},"page":"270-282","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":13,"title":["Meaningful Improvements in Rett Syndrome: A Qualitative Study of Caregivers"],"prefix":"10.1177","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3208-8129","authenticated-orcid":false,"given":"Sarah A.","family":"McGraw","sequence":"first","affiliation":[{"name":"MNW Consulting Group, Portland, OR, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8241-9574","authenticated-orcid":false,"given":"Constance","family":"Smith-Hicks","sequence":"additional","affiliation":[{"name":"Kennedy Krieger Institute, Baltimore, MD, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Nutter","sequence":"additional","affiliation":[{"name":"MNW Consulting Group, Portland, OR, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey C.","family":"Henne","sequence":"additional","affiliation":[{"name":"The Henne Group, San Francisco, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Victor","family":"Abler","sequence":"additional","affiliation":[{"name":"Medical Affairs, Acadia Pharmaceuticals, Inc, San Diego, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2023,5,9]]},"reference":[{"key":"bibr1-08830738231172066","doi-asserted-by":"publisher","DOI":"10.1002\/ana.22124"},{"key":"bibr2-08830738231172066","doi-asserted-by":"publisher","DOI":"10.1038\/13810"},{"key":"bibr3-08830738231172066","doi-asserted-by":"publisher","DOI":"10.1016\/j.jmb.2019.10.004"},{"key":"bibr4-08830738231172066","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1985.tb10993.x"},{"key":"bibr5-08830738231172066","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2005.10.037"},{"issue":"1","key":"bibr6-08830738231172066","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1002\/ajmg.b.32707","volume":"180","author":"Neul JL","year":"2019","journal-title":"Am J Med Genet B Neuropsychiatr 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D, Youakim JM, Ventola P. Mapping the top concerns of caregivers of individuals with Rett Syndrome with items on the Rett Syndrome Behaviour Questionnaire to identify clinically meaningful outcomes. Paper presented at: National Organization for Rare Disorders (NORD) Rare Diseases and Orphan Products Breakthrough Summit; Washington, DC, October 16-18, 2022."}],"container-title":["Journal of Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/08830738231172066","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/08830738231172066","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/08830738231172066","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T20:40:50Z","timestamp":1777495250000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/08830738231172066"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,4]]},"references-count":41,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2023,4]]}},"alternative-id":["10.1177\/08830738231172066"],"URL":"https:\/\/doi.org\/10.1177\/08830738231172066","relation":{},"ISSN":["0883-0738","1708-8283"],"issn-type":[{"value":"0883-0738","type":"print"},{"value":"1708-8283","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,4]]}}}