{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T20:34:12Z","timestamp":1777494852107,"version":"3.51.4"},"reference-count":30,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2022,9,21]],"date-time":"2022-09-21T00:00:00Z","timestamp":1663718400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100006108","name":"National Center for Advancing Translational Sciences","doi-asserted-by":"publisher","award":["UL1 TR001079"],"award-info":[{"award-number":["UL1 TR001079"]}],"id":[{"id":"10.13039\/100006108","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Research Accelerator and Mentorship Program (RAMP)Johns Hopkins Clinical Research Network"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Child Health Care"],"published-print":{"date-parts":[[2024,6]]},"abstract":"<jats:p>Hypermobile Ehlers-Danlos syndrome (hEDS) is a common disorder in children and adolescents that negatively impacts health-related quality of life (HRQOL). It can include chronic pain, fatigue, autonomic dysfunction, and mood problems. The objective of this study was to examine levels of agreement between children and parents in the setting of hEDS and HRQOL. Individuals with hEDS, ages 10-20\u00a0years, and their parents were recruited to complete a series of surveys. Instruments included pediatric quality of life generic and multidimensional fatigue scales, Functional Disability Index, Pain-Frequency-Severity-Duration scale, Brief Illness Perception Questionnaire, and Herth Hope Index. Agreement on each measure was evaluated using statistical calculations. Thirty-six parent-child dyads completed the surveys. There were no significant differences between the means of parent and child scores. There was moderate to strong agreement on all survey scores. However, the proportion of dyads with disagreement was relatively high for each individual score. Eighteen dyads disagreed on at least half of the surveys. Body mass index centile and child perception of cognitive fatigue most strongly predicted disagreement in total HRQOL score. Proxy-reporters for children and adolescents with hEDS may agree with their child on average. However, due to significant frequency of clinically important disagreement, information from both children and their parents should be sought whenever possible.<\/jats:p>","DOI":"10.1177\/13674935221110081","type":"journal-article","created":{"date-parts":[[2022,9,21]],"date-time":"2022-09-21T05:06:09Z","timestamp":1663736769000},"page":"316-328","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Agreement between parent-proxy and child self-report in pediatric hypermobile Ehlers-Danlos syndrome"],"prefix":"10.1177","volume":"28","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6573-7519","authenticated-orcid":false,"given":"You","family":"Wang","sequence":"first","affiliation":[{"name":"Johns Hopkins University Krieger School of Arts and Sciences, Baltimore, MD, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Julia L","family":"Clemens","sequence":"additional","affiliation":[{"name":"University of Wisconsin Hospitals and Clinics, Madison, WI"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael","family":"Muriello","sequence":"additional","affiliation":[{"name":"Medical College of Wisconsin, Milwaukee, WI"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Weiyi","family":"Mu","sequence":"additional","affiliation":[{"name":"McKusick-Nathans Department of Genetic Medicine, Johns Hopkins University, Baltimore, MD"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christy H","family":"Smith","sequence":"additional","affiliation":[{"name":"McKusick-Nathans Department of Genetic Medicine, Johns Hopkins University, Baltimore, MD"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7022-1576","authenticated-orcid":false,"given":"Phuong T","family":"Tran","sequence":"additional","affiliation":[{"name":"Faculty of Pharmacy HUTECH University, Ho Chi Minh City, Vietnam"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peter C","family":"Rowe","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Johns Hopkins School of Medicine, Johns Hopkins University, Baltimore, MD"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Clair","family":"Francomano","sequence":"additional","affiliation":[{"name":"Department of Medical and Molecular Genetics, Indiana University School of Medicine, Indianapolis, IN"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antonie D","family":"Kline","sequence":"additional","affiliation":[{"name":"Harvey Institute for Human Genetics, Greater Baltimore Medical Center, Towson, MD"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1667-5972","authenticated-orcid":false,"given":"Joann","family":"Bodurtha","sequence":"additional","affiliation":[{"name":"McKusick-Nathans Department of Genetic Medicine, Johns Hopkins University, Baltimore, MD"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2022,9,21]]},"reference":[{"key":"bibr1-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-005-3690-1"},{"key":"bibr2-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980428)77:1<31::AID-AJMG8>3.0.CO;2-O"},{"key":"bibr3-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1044\/2019_LSHSS-OCHL-19-0017"},{"key":"bibr4-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1177\/0272989X03256008"},{"key":"bibr5-13674935221110081","doi-asserted-by":"publisher","DOI":"10.23736\/S0026-4946.20.05766-7"},{"key":"bibr6-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1038\/ijo.2009.241"},{"key":"bibr7-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-005-4184-x"},{"key":"bibr8-13674935221110081","doi-asserted-by":"publisher","DOI":"10.2165\/00019053-200523070-00003"},{"key":"bibr9-13674935221110081","doi-asserted-by":"publisher","DOI":"10.1023\/A:1012253723272"},{"key":"bibr10-13674935221110081","unstructured":"Elias A. 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