{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T14:56:32Z","timestamp":1790520992159,"version":"4.1.0"},"reference-count":44,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2025,6,1]],"date-time":"2025-06-01T00:00:00Z","timestamp":1748736000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2025,6,1]],"date-time":"2025-06-01T00:00:00Z","timestamp":1748736000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2025,3,26]],"date-time":"2025-03-26T00:00:00Z","timestamp":1742947200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100009712","name":"Dravet Syndrome Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009712","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","epilepsybehavior.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Epilepsy &amp; Behavior"],"published-print":{"date-parts":[[2025,6]]},"DOI":"10.1016\/j.yebeh.2025.110381","type":"journal-article","created":{"date-parts":[[2025,4,9]],"date-time":"2025-04-09T12:12:53Z","timestamp":1744200773000},"page":"110381","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":15,"special_numbering":"C","title":["Qualitative evaluation of meaningful change in Dravet syndrome as measured by the Vineland-3: Caregiver and clinician perspectives"],"prefix":"10.1016","volume":"167","author":[{"given":"Carolyn","family":"Condon","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kimberly A.","family":"Parkerson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ann","family":"Dandurand","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fei","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chloe","family":"Howse","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-4191-3305","authenticated-orcid":false,"given":"Megan","family":"Mayhew","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sophie","family":"Whyman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8062-0626","authenticated-orcid":false,"given":"Pamela","family":"Ventola","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary","family":"Anne Meskis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2081-8988","authenticated-orcid":false,"given":"Joseph","family":"Sullivan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelly G.","family":"Knupp","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adam","family":"Gater","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aoife","family":"Mahon-Smith","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.yebeh.2025.110381_b0005","unstructured":"Orpha.net. \u201cDravet syndrome.\u201d https:\/\/www.orpha.net\/en\/disease\/detail\/33069?name=Dravet%20syndrome&mode=name (accessed 2024, 2024)."},{"key":"10.1016\/j.yebeh.2025.110381_b0010","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1111\/epi.12927","article-title":"The incidence of SCN1A-related Dravet syndrome in Denmark is 1: 22 ,000: a population-based study from 2004 to 2009","volume":"56","author":"Bayat","year":"2015","journal-title":"Epilepsia"},{"key":"10.1016\/j.yebeh.2025.110381_b0015","doi-asserted-by":"crossref","first-page":"2303","DOI":"10.1093\/brain\/awz195","article-title":"Incidence and phenotypes of childhood-onset genetic epilepsies: a prospective population based national cohort","volume":"142","author":"Symonds","year":"2019","journal-title":"Brain"},{"key":"10.1016\/j.yebeh.2025.110381_b0020","doi-asserted-by":"crossref","first-page":"2329","DOI":"10.1093\/brain\/aws151","article-title":"Prognostic, clinical and demographic features in SCN1A mutation-positive Dravet syndrome","volume":"135","author":"Brunklaus","year":"2012","journal-title":"Brain"},{"key":"10.1016\/j.yebeh.2025.110381_b0025","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1111\/j.1528-1157.1990.tb05494.x","article-title":"Epidemiology of severe myoclonic epilepsy of infancy","volume":"31","author":"Hurst","year":"1990","journal-title":"Epilepsia"},{"key":"10.1016\/j.yebeh.2025.110381_b0030","doi-asserted-by":"crossref","first-page":"299","DOI":"10.1016\/S0387-7604(12)80147-1","article-title":"Early diagnosis of severe myoclonic epilepsy in infancy","volume":"14","author":"Yakoub","year":"1992","journal-title":"Brain Devel"},{"issue":"136","key":"10.1016\/j.yebeh.2025.110381_b0035","doi-asserted-by":"crossref","first-page":"e1310","DOI":"10.1542\/peds.2015-1807","article-title":"Incidence of Dravet Syndrome in a US Population","volume":"5","author":"Wu","year":"2015","journal-title":"Pediatrics"},{"key":"10.1016\/j.yebeh.2025.110381_b0040","doi-asserted-by":"crossref","first-page":"102","DOI":"10.1016\/j.seizure.2020.12.021","article-title":"Sleep in Dravet syndrome: a parent-driven survey","volume":"85","author":"Van Nuland","year":"2021","journal-title":"Seizure"},{"issue":"10","key":"10.1016\/j.yebeh.2025.110381_b0045","doi-asserted-by":"crossref","first-page":"1343","DOI":"10.1111\/dmcn.15561","article-title":"Functional mobility in children and young adults with Dravet syndrome","volume":"65","author":"Verheyen","year":"2023","journal-title":"Dev Med Child Neurol"},{"key":"10.1016\/j.yebeh.2025.110381_b0050","doi-asserted-by":"crossref","first-page":"81","DOI":"10.1016\/j.yebeh.2017.06.031","article-title":"Dravet syndrome: characteristics, comorbidities, and caregiver concerns","volume":"74","author":"Villas","year":"2017","journal-title":"Epilepsy Behav"},{"key":"10.1016\/j.yebeh.2025.110381_b0055","doi-asserted-by":"crossref","DOI":"10.1016\/j.yebeh.2022.108978","article-title":"Challenging behavior in children and adolescents with Dravet syndrome: Exploring the lived experiences of parents","volume":"138","author":"Postma","year":"2023","journal-title":"Epilepsy Behav"},{"issue":"6","key":"10.1016\/j.yebeh.2025.110381_b0060","article-title":"Dravet syndrome: an overview","volume":"11","author":"Anwar","year":"2019","journal-title":"Cureus"},{"key":"10.1016\/j.yebeh.2025.110381_b0065","article-title":"Natural history of rare diseases using natural language processing of narrative unstructured electronic health records: the example of Dravet syndrome","author":"Lo Barco","year":"2023","journal-title":"Epilepsia"},{"issue":"10","key":"10.1016\/j.yebeh.2025.110381_b0070","doi-asserted-by":"crossref","first-page":"1229","DOI":"10.1111\/dmcn.14186","article-title":"Zogenix and Adelphi Values study group, \u201cPerception of impact of Dravet syndrome on children and caregivers in multiple countries: looking beyond seizures","volume":"61","author":"Nabbout","year":"2019","journal-title":"Dev Med Child Neurol"},{"issue":"1","key":"10.1016\/j.yebeh.2025.110381_b0075","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1111\/dmcn.13591","article-title":"Quality of life and comorbidities associated with Dravet syndrome severity: a multinational cohort survey","volume":"60","author":"Lagae","year":"2018","journal-title":"Dev Med Child Neurol"},{"key":"10.1016\/j.yebeh.2025.110381_b0080","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/1750-1172-8-176","article-title":"Encephalopathy in children with Dravet syndrome is not a pure consequence of epilepsy","volume":"8","author":"Nabbout","year":"2013","journal-title":"Orphanet J Rare Dis"},{"issue":"4","key":"10.1016\/j.yebeh.2025.110381_b0085","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1002\/mgg3.217","article-title":"Pitfalls in genetic testing: the story of missed SCN1A mutations","volume":"4","author":"Dj\u00e9mi\u00e9","year":"2016","journal-title":"Mol Genet Genomic Med"},{"key":"10.1016\/j.yebeh.2025.110381_b0090","doi-asserted-by":"crossref","DOI":"10.1016\/j.yebeh.2023.109604","article-title":"Adaptive functioning and neurodevelopment in patients with Dravet syndrome: 12-month interim analysis of the BUTTERFLY observational study","volume":"151","author":"Sullivan","year":"2024","journal-title":"Epilepsy Behav"},{"key":"10.1016\/j.yebeh.2025.110381_b0095","series-title":"Vineland Adaptive Behavior Scales, Third Edition (Vineland-3)","author":"Sparrow","year":"2016"},{"issue":"5","key":"10.1016\/j.yebeh.2025.110381_b0100","doi-asserted-by":"crossref","first-page":"342","DOI":"10.1177\/15357597241280687","article-title":"ENVISIONing a critical period to preserve development: communication delays in SCN1A+ dravet syndrome","volume":"24","author":"Fine","year":"2024","journal-title":"Epilepsy Currents"},{"issue":"5","key":"10.1016\/j.yebeh.2025.110381_b0105","doi-asserted-by":"crossref","first-page":"613","DOI":"10.1001\/jamaneurol.2020.0073","article-title":"GWPCARE2 Study Group., \u201cDose-Ranging Effect of Adjunctive Oral Cannabidiol vs Placebo on Convulsive Seizure Frequency in Dravet Syndrome: A Randomized Clinical Trial","volume":"77","author":"Miller","year":"2020","journal-title":"JAMA Neurol"},{"key":"10.1016\/j.yebeh.2025.110381_b0110","doi-asserted-by":"crossref","unstructured":"Chladek M, Burbridge C, Gibbons E, Willgoss T, Smith J, and Clinch S, Qualitative exploration in exit interviews of changes observed in clinical trials for individuals with autism spectrum disorder without intellectual disability, Patient Related Outcome Measures, pp. 313-335, 2023.","DOI":"10.2147\/PROM.S385682"},{"key":"10.1016\/j.yebeh.2025.110381_b0115","doi-asserted-by":"crossref","unstructured":"Clinch S. et al., Quantitative and Qualitative Exploration of Meaningful Change on the Vineland Adaptive Behavior Scales (Vineland\u2122-II) in Children and Adolescents with Autism Without Intellectual Disability Following Participation in a Clinical Trial, Patient Related Outcome Measures, pp. 337-354, 2023.","DOI":"10.2147\/PROM.S385542"},{"issue":"7","key":"10.1016\/j.yebeh.2025.110381_b0120","doi-asserted-by":"crossref","first-page":"838","DOI":"10.1016\/j.jval.2017.05.015","article-title":"Patient-reported outcome and observer-reported outcome assessment in rare disease clinical trials: an ISPOR COA emerging good practices task force report","volume":"20","author":"Benjamin","year":"2017","journal-title":"Value Health"},{"key":"10.1016\/j.yebeh.2025.110381_b0125","unstructured":"US Food and Drug Administration. \u201cPatient-Focused Drug Development: Incorporating Clinical Outcome Assessments into Endpoints for Regulatory Decision-Making.\u201d https:\/\/www.fda.gov\/regulatory-information\/search-fda-guidance-documents\/patient-focused-drug-development-incorporating-clinical-outcome-assessments-endpoints-regulatory (accessed."},{"key":"10.1016\/j.yebeh.2025.110381_b0130","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/s41687-019-0100-y","article-title":"An overview of using qualitative techniques to explore and define estimates of clinically important change on clinical outcome assessments","volume":"3","author":"Staunton","year":"2019","journal-title":"J Patient-reported Outcomes"},{"key":"10.1016\/j.yebeh.2025.110381_b0135","unstructured":"US Food and Drug Administration. \u201cPatient-Focused Drug Development: Workshop to Discuss Methodologic and Other Challenges Related to Patient Experience Data.\u201d https:\/\/www.fda.gov\/drugs\/news-events-human-drugs\/patient-focused-drug-development-workshop-discuss-methodologic-and-other-challenges-related-patient#event-materials (accessed December 16, 2024."},{"key":"10.1016\/j.yebeh.2025.110381_b0140","doi-asserted-by":"crossref","first-page":"1159","DOI":"10.1007\/s11136-018-1829-0","article-title":"COSMIN methodology for evaluating the content validity of patient-reported outcome measures: a Delphi study","volume":"27","author":"Terwee","year":"2018","journal-title":"Qual Life Res"},{"key":"10.1016\/j.yebeh.2025.110381_b0145","series-title":"Cognitive interviewing: A tool for improving questionnaire design","author":"Willis","year":"2004"},{"issue":"4","key":"10.1016\/j.yebeh.2025.110381_b0150","doi-asserted-by":"crossref","first-page":"636","DOI":"10.1093\/poq\/nfr035","article-title":"Sample size for cognitive interview pretesting","volume":"75","author":"Blair","year":"2011","journal-title":"Public Opin Q"},{"issue":"7","key":"10.1016\/j.yebeh.2025.110381_b0155","doi-asserted-by":"crossref","first-page":"839","DOI":"10.1016\/j.jval.2017.11.014","article-title":"Informing a priori sample size estimation in qualitative concept elicitation interview studies for clinical outcome assessment instrument development","volume":"21","author":"Turner-Bowker","year":"2018","journal-title":"Value Health"},{"key":"10.1016\/j.yebeh.2025.110381_b0160","unstructured":"Dravet Syndrome Foundation. \u201cExternally-Led Patient-Focused Drug Development Meeting on Dravet Syndrome.\u201d https:\/\/dravetfoundation.org\/dsf-funded-research\/externally-led-pfdd-meeting\/ (accessed."},{"key":"10.1016\/j.yebeh.2025.110381_b0165","unstructured":"S. Friese. \u201cATLAS.ti 22 User Manual.\u201d https:\/\/atlasti.com\/manuals-docs\/ (accessed 29 September 2023."},{"issue":"2","key":"10.1016\/j.yebeh.2025.110381_b0170","doi-asserted-by":"crossref","first-page":"77","DOI":"10.1191\/1478088706qp063oa","article-title":"Using thematic analysis in psychology","volume":"3","author":"Braun","year":"2006","journal-title":"Qual Res Psychol"},{"key":"10.1016\/j.yebeh.2025.110381_b0175","unstructured":"Joffe HY. Content and thematic analysis 2003."},{"issue":"5","key":"10.1016\/j.yebeh.2025.110381_b0180","doi-asserted-by":"crossref","first-page":"405","DOI":"10.1038\/gim.2015.30","article-title":"Standards and guidelines for the interpretation of sequence variants: a joint consensus recommendation of the American College of Medical Genetics and Genomics and the Association for Molecular Pathology","volume":"17","author":"Richards","year":"2015","journal-title":"Genet Med"},{"key":"10.1016\/j.yebeh.2025.110381_b0185","doi-asserted-by":"crossref","first-page":"135","DOI":"10.1016\/j.yebeh.2017.06.012","article-title":"Life impact of caregiving for severe childhood epilepsy: results of expert panels and caregiver focus groups","volume":"74","author":"Jensen","year":"2017","journal-title":"Epilepsy Behav"},{"issue":"3","key":"10.1016\/j.yebeh.2025.110381_b0190","doi-asserted-by":"crossref","first-page":"531","DOI":"10.1093\/brain\/awg053","article-title":"Mutations of sodium channel \u03b1 subunit type 1 (SCN1A) in intractable childhood epilepsies with frequent generalized tonic\u2013clonic seizures","volume":"126","author":"Fujiwara","year":"2003","journal-title":"Brain"},{"key":"10.1016\/j.yebeh.2025.110381_b0195","doi-asserted-by":"crossref","first-page":"138","DOI":"10.1007\/s40474-019-00172-y","article-title":"The link between problem behavior and communication impairment in persons with developmental disabilities","volume":"6","author":"Durand","year":"2019","journal-title":"Curr Dev Disord Rep"},{"key":"10.1016\/j.yebeh.2025.110381_b0200","unstructured":"US Food and Drug Administration, \u201cPatient-focused drug development: selecting, developing, or modifying fit-for purpose clinical outcome assessments (draft guidance),\u201d 2022."},{"key":"10.1016\/j.yebeh.2025.110381_b0205","unstructured":"Gater A, Marshall C, Grant L, Wells J. The Use of Qualitative (Exit) Interviews in Clinical Trials: Value, Implementation and Key Considerations. In: VALUE IN HEALTH, 2017, vol. 20, no. 5: ELSEVIER SCIENCE INC 360 PARK AVE SOUTH, NEW YORK, NY 10010-1710 USA, pp. A343-A344."},{"key":"10.1016\/j.yebeh.2025.110381_b0210","unstructured":"Williamson N, Bonner N, and W. J, \u201cQualitative in-trial or exit interview methods to elicit supplementary patient experience data during clinical studies,\u201d in ISOQOL, 2022."},{"key":"10.1016\/j.yebeh.2025.110381_b0215","unstructured":"US Food and Drug Administration. \u201cClinical Outcome Assessment (COA): Frequently Asked Questions.\u201d https:\/\/www.fda.gov\/about-fda\/clinical-outcome-assessment-coa-frequently-asked-questions#:\u223c:text=A%20clinical%20outcome%20assessment%20is,%2Dreported%20outcome%20(ClinRO)%20measures (accessed."},{"key":"10.1016\/j.yebeh.2025.110381_b0220","unstructured":"US Food and Drug Administration, \u201cGuidance for Industry Patient-Reported Outcome Measures: Use in Medical Product Development to Support Labeling Claims,\u201d 2009. [Online]. Available: https:\/\/www.fda.gov\/media\/77832\/download."}],"container-title":["Epilepsy &amp; Behavior"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1525505025001209?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1525505025001209?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,11,7]],"date-time":"2025-11-07T20:44:20Z","timestamp":1762548260000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1525505025001209"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6]]},"references-count":44,"alternative-id":["S1525505025001209"],"URL":"https:\/\/doi.org\/10.1016\/j.yebeh.2025.110381","relation":{},"ISSN":["1525-5050"],"issn-type":[{"value":"1525-5050","type":"print"}],"subject":[],"published":{"date-parts":[[2025,6]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Qualitative evaluation of meaningful change in Dravet syndrome as measured by the Vineland-3: Caregiver and clinician perspectives","name":"articletitle","label":"Article Title"},{"value":"Epilepsy & Behavior","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.yebeh.2025.110381","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2025 The Authors. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"110381"}}