{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T02:32:17Z","timestamp":1790562737307,"version":"4.1.0"},"reference-count":18,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2024,5,14]],"date-time":"2024-05-14T00:00:00Z","timestamp":1715644800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,5,14]],"date-time":"2024-05-14T00:00:00Z","timestamp":1715644800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100009726","name":"Foundation for Angelman Syndrome Therapeutics","doi-asserted-by":"publisher","award":["FT2018-005"],"award-info":[{"award-number":["FT2018-005"]}],"id":[{"id":"10.13039\/100009726","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Patient Rep Outcomes"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Aims<\/jats:title>\n                <jats:p>Caregivers rate improved communication ability as one of the most desired outcomes for successful interventions for individuals with Angelman syndrome (AS). When measuring communication ability in clinical trials, the reliability of such measures is critical for detecting significant changes over time. This study examined the reliability of the Observed-Reported Communication Ability (ORCA) measure completed by caregivers of individuals with AS.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Methods<\/jats:title>\n                <jats:p>The ORCA measure was completed by 249 caregivers with 170 caregivers completing the ORCA measure again after 5\u201312 days. Generalizability theory was used to examine the following sources of measurement error in ORCA scores: concepts, subdomains, assessment points, and the interactions among those facets and the object of measurement: communication ability. Three generalizability studies were conducted to understand the reliability of the ORCA measure for different measurement designs. Decision studies were carried out to demonstrate the optimization of measurement procedures of the ORCA measure.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Results<\/jats:title>\n                <jats:p>G and Phi coefficients of the original measurement design exceeded the 0.80 threshold considered sufficiently reliable to make relative and absolute decisions about the communication ability of individuals with AS based on their caregivers\u2019 observed scores. The optimization procedures indicated that increasing the number of communication concepts and\/or assessment points leads to more reliable estimates of communication.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>The ORCA measure was able to reliably distinguish different levels of communication ability among individuals with AS. Multiple assessment points and or more concepts would provide more precise estimates of an individual\u2019s communication ability but at the cost of survey fatigue.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s41687-024-00725-9","type":"journal-article","created":{"date-parts":[[2024,5,14]],"date-time":"2024-05-14T11:02:19Z","timestamp":1715684539000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Understanding reliability of the observer-reported communication ability measure within Angelman syndrome through the lens of generalizability theory"],"prefix":"10.1186","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1936-080X","authenticated-orcid":false,"given":"Dandan","family":"Chen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christina K.","family":"Zigler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Li","family":"Lin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicole","family":"Lucas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Molly","family":"McFatrich","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer","family":"Panagoulias","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Allyson","family":"Berent","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bryce B.","family":"Reeve","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,5,14]]},"reference":[{"issue":"4","key":"725_CR1","doi-asserted-by":"publisher","first-page":"654","DOI":"10.1007\/s10578-020-01051-z","volume":"52","author":"T Willgoss","year":"2021","unstructured":"Willgoss T et al (2021) Measuring what matters to individuals with Angelman Syndrome and their families: development of a patient-centered Disease Concept Model. Child Psychiatry Hum Dev 52(4):654\u2013668","journal-title":"Child Psychiatry Hum Dev"},{"issue":"3","key":"725_CR2","doi-asserted-by":"publisher","first-page":"641","DOI":"10.1007\/s13311-015-0361-y","volume":"12","author":"SS Margolis","year":"2015","unstructured":"Margolis SS et al (2015) Angelman Syndrome. Neurotherapeutics 12(3):641\u2013650","journal-title":"Neurotherapeutics"},{"key":"725_CR3","doi-asserted-by":"crossref","unstructured":"Zigler CK, et al (2023) Validation of the Observer-Reported Communication Ability (ORCA) measure for individuals with Angelman syndrome. Am J Intellect Dev Disabil 128(3):204\u2013218","DOI":"10.1352\/1944-7558-128.3.204"},{"key":"725_CR4","doi-asserted-by":"crossref","unstructured":"Zigler CK, et al (2023) Exploring communication ability in individuals with Angelman syndrome: findings from qualitative interviews with caregivers. 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The current study was ruled exempt from IRB review by the Duke University Health System. Informed consent was obtained from all individual participants included in the study.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"The authors affirm that human research participants provided informed consent for publication sensitive nature of the research.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"Dr. Zigler, Dr. Reeve, Ms. Lucas, Ms. McFatrich, and Ms. Lin have developed the technology that is being used in the study. If the technology is commercially successful in the future, the developers and Duke University may benefit financially draft approval.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing Interests"}}],"article-number":"50"}}