{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T03:38:24Z","timestamp":1788406704057,"version":"build-2803163510"},"reference-count":48,"publisher":"Elsevier BV","issue":"7","license":[{"start":{"date-parts":[[2026,7,1]],"date-time":"2026-07-01T00:00:00Z","timestamp":1782864000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,7,1]],"date-time":"2026-07-01T00:00:00Z","timestamp":1782864000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","jhandsurg.org","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["The Journal of Hand Surgery"],"published-print":{"date-parts":[[2026,7]]},"DOI":"10.1016\/j.jhsa.2026.02.015","type":"journal-article","created":{"date-parts":[[2026,4,2]],"date-time":"2026-04-02T11:31:22Z","timestamp":1775129482000},"page":"782.e1-782.e7","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":1,"title":["Health-Related Quality of Life Outcomes in Pediatric Patients With Apert Syndrome"],"prefix":"10.1016","volume":"51","author":[{"given":"Hannah C.","family":"Brown","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura C.","family":"Nuzzi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"III","given":"Joseph","family":"Upton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian I.","family":"Labow","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amir H.","family":"Taghinia","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib1","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1016\/S0094-1298(20)30826-9","article-title":"Apert syndrome. Classification and pathologic anatomy of limb anomalies","volume":"18","author":"Upton","year":"1991","journal-title":"Clin Plast Surg"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib2","doi-asserted-by":"crossref","first-page":"101","DOI":"10.1186\/s12955-024-02312-4","article-title":"The use of patient-reported outcome measures to improve patient-related outcomes\u2014a systematic review","volume":"22","author":"Bonsel","year":"2024","journal-title":"Health Qual Life Outcomes"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib3","doi-asserted-by":"crossref","first-page":"66","DOI":"10.3171\/jns.1996.85.1.0066","article-title":"Prognosis for mental function in Apert\u2019s syndrome","volume":"85","author":"Renier","year":"1996","journal-title":"J Neurosurg"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib4","doi-asserted-by":"crossref","first-page":"164","DOI":"10.1136\/jmg.25.3.164","article-title":"Intellectual development in Apert\u2019s syndrome: a long term follow up of 29 patients","volume":"25","author":"Patton","year":"1988","journal-title":"J Med Genet"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib5","doi-asserted-by":"crossref","first-page":"963","DOI":"10.1590\/S0004-282X2005000600011","article-title":"Apert syndrome: factors involved in the cognitive development","volume":"63","author":"Yacubian-Fernandes","year":"2005","journal-title":"Arq Neuropsiquiatr"},{"issue":"6","key":"10.1016\/j.jhsa.2026.02.015_bib6","doi-asserted-by":"crossref","first-page":"879","DOI":"10.1007\/s00381-012-1681-4","article-title":"Health-related problems and quality of life in patients with syndromic and complex craniosynostosis","volume":"28","author":"de Jong","year":"2012","journal-title":"Childs Nerv Syst"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib7","doi-asserted-by":"crossref","first-page":"450","DOI":"10.1097\/01.sap.0000149387.95212.df","article-title":"Neuropsychological diversity in Apert syndrome: a comparison of cognitive profiles","volume":"54","author":"Da Costa","year":"2005","journal-title":"Ann Plast Surg"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib8","doi-asserted-by":"crossref","first-page":"325","DOI":"10.1080\/13682820210138816","article-title":"Speech and language skills and cognitive functioning in children with Apert syndrome: a pilot study","volume":"37","author":"Shipster","year":"2002","journal-title":"Int J Lang Commun Disord"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib9","first-page":"317","article-title":"Cognitive functioning of young children with Apert\u2019s syndrome","volume":"8","author":"Sarimski","year":"1997","journal-title":"Genet Couns"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib10","doi-asserted-by":"crossref","first-page":"510","DOI":"10.1016\/0007-1226(86)90122-0","article-title":"A psychiatric profile before and after reconstructive surgery in children with Apert\u2019s syndrome","volume":"39","author":"Lefebvre","year":"1986","journal-title":"Br J Plast Surg"},{"key":"10.1016\/j.jhsa.2026.02.015_bib11","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1186\/s40510-015-0078-9","article-title":"Psychological impact of visible differences in patients with congenital craniofacial anomalies","volume":"16","author":"Singh","year":"2015","journal-title":"Prog Orthod"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib12","doi-asserted-by":"crossref","first-page":"461","DOI":"10.1177\/10556656211069817","article-title":"Psychological adjustment in Apert syndrome: parent and young person perspectives","volume":"60","author":"Netherton","year":"2023","journal-title":"Cleft Palate Craniofac J"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib13","doi-asserted-by":"crossref","first-page":"1601","DOI":"10.1097\/PRS.0b013e31820a64b6","article-title":"Treatment of Apert syndrome: a long-term follow-up study","volume":"127","author":"Allam","year":"2011","journal-title":"Plast Reconstr Surg"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib15","doi-asserted-by":"crossref","first-page":"200e","DOI":"10.1097\/PRS.0000000000001444","article-title":"Applying the patient-reported outcomes measurement information system to assess upper extremity function among children with congenital hand differences","volume":"136","author":"Waljee","year":"2015","journal-title":"Plast Reconstr Surg"},{"issue":"5","key":"10.1016\/j.jhsa.2026.02.015_bib16","doi-asserted-by":"crossref","first-page":"1215","DOI":"10.1097\/PRS.0000000000006743","article-title":"Patient-reported outcomes after syndactyly reconstruction","volume":"145","author":"Park","year":"2020","journal-title":"Plast Reconstr Surg"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib17","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1016\/j.jhsa.2017.10.006","article-title":"Functional impact of congenital hand differences: early results from the congenital upper limb differences (CoULD) registry","volume":"43","author":"Bae","year":"2018","journal-title":"J Hand Surg Am"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib18","doi-asserted-by":"crossref","first-page":"194","DOI":"10.1177\/1558944718793195","article-title":"Performance of pediatric PROMIS CATs in children with upper extremity fractures","volume":"15","author":"Gerull","year":"2020","journal-title":"Hand (N Y)"},{"issue":"10","key":"10.1016\/j.jhsa.2026.02.015_bib19","doi-asserted-by":"crossref","first-page":"927.e1","DOI":"10.1016\/j.jhsa.2021.01.023","article-title":"Establishing the minimal clinically important difference for the PROMIS upper extremity computer adaptive test version 2.0 in a nonshoulder hand and upper extremity population","volume":"46","author":"Kazmers","year":"2021","journal-title":"J Hand Surg Am"},{"issue":"7","key":"10.1016\/j.jhsa.2026.02.015_bib20","doi-asserted-by":"crossref","first-page":"357","DOI":"10.1097\/BPO.0000000000001527","article-title":"Patient-reported outcomes in arthrogryposis","volume":"40","author":"Wall","year":"2020","journal-title":"J Pediatr Orthop"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib21","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1016\/j.jhsa.2020.08.017","article-title":"Social deprivation and congenital upper extremity differences-an assessment using PROMIS","volume":"46","author":"Wall","year":"2021","journal-title":"J Hand Surg Am"},{"issue":"5","key":"10.1016\/j.jhsa.2026.02.015_bib22","doi-asserted-by":"crossref","first-page":"e466","DOI":"10.1097\/BPO.0000000000002106","article-title":"Long-term outcomes after treatment for type B ulnar polydactyly","volume":"42","author":"Goebel","year":"2022","journal-title":"J Pediatr Orthop"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib23","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1016\/j.jhsa.2022.10.018","article-title":"Effect of race and geography on patient- and parent-reported quality of life for children with congenital upper limb differences","volume":"48","author":"Daley","year":"2023","journal-title":"J Hand Surg Am"},{"issue":"7","key":"10.1016\/j.jhsa.2026.02.015_bib24","doi-asserted-by":"crossref","first-page":"681","DOI":"10.1016\/j.jhsa.2024.02.011","article-title":"The impact on families of children with congenital upper extremity differences","volume":"49","author":"Ballatori","year":"2024","journal-title":"J Hand Surg Am"},{"issue":"8","key":"10.1016\/j.jhsa.2026.02.015_bib25","doi-asserted-by":"crossref","first-page":"772","DOI":"10.1016\/j.jhsa.2024.01.019","article-title":"Patient and caregiver impressions of the impact of Madelung deformity: a CoULD registry analysis","volume":"49","author":"Goldfarb","year":"2024","journal-title":"J Hand Surg Am"},{"issue":"5","key":"10.1016\/j.jhsa.2026.02.015_bib26","article-title":"Hand function in Apert syndrome","volume":"7","author":"Raposo-Amaral","year":"2019","journal-title":"Plast Reconstr Surg Glob Open"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib27","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1097\/PRS.0000000000005479","article-title":"Long-term functional upper-extremity outcomes in adults with Apert syndrome","volume":"143","author":"Taghinia","year":"2019","journal-title":"Plast Reconstr Surg"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib28","doi-asserted-by":"crossref","first-page":"33","DOI":"10.1016\/j.jhsa.2019.08.003","article-title":"Convergent validity of PODCI and PROMIS domains in congenital upper limb anomalies","volume":"45","author":"Wall","year":"2020","journal-title":"J Hand Surg Am"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib29","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1016\/j.jbi.2008.08.010","article-title":"Research Electronic Data Capture (REDCap)\u2014a metadata-driven methodology and workflow process for providing translational research informatics support","volume":"42","author":"Harris","year":"2009","journal-title":"J Biomed Inform"},{"issue":"7","key":"10.1016\/j.jhsa.2026.02.015_bib36","doi-asserted-by":"crossref","first-page":"1223","DOI":"10.1007\/s11136-011-0025-2","article-title":"PROMIS\u00ae Parent Proxy Report Scales: an item response theory analysis of the parent proxy report item banks","volume":"21","author":"Varni","year":"2012","journal-title":"Qual Life Res"},{"key":"10.1016\/j.jhsa.2026.02.015_bib37","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1186\/1477-7525-7-3","article-title":"Cognitive interviewing methodology in the development of a pediatric item bank: a patient reported outcomes measurement information system (PROMIS) study","volume":"7","author":"Irwin","year":"2009","journal-title":"Health Qual Life Outcomes"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib38","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1186\/1477-7525-10-22","article-title":"Development of six PROMIS pediatrics proxy-report item banks","volume":"10","author":"Irwin","year":"2012","journal-title":"Health Qual Life Outcomes"},{"issue":"9","key":"10.1016\/j.jhsa.2026.02.015_bib40","doi-asserted-by":"crossref","first-page":"830","DOI":"10.1016\/j.jhsa.2020.05.011","article-title":"Upper extremity function, peer relationships, and pain interference: evaluating the biopsychosocial model in a pediatric hand surgery population using PROMIS","volume":"45","author":"Kazmers","year":"2020","journal-title":"J Hand Surg Am"},{"issue":"7","key":"10.1016\/j.jhsa.2026.02.015_bib41","doi-asserted-by":"crossref","first-page":"838","DOI":"10.1097\/BPO.0b013e3181455886a","article-title":"A clinicoradiologic study of the shoulder in Apert syndrome","volume":"27","author":"Murnaghan","year":"2007","journal-title":"J Pediatr Orthop"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib42","doi-asserted-by":"crossref","first-page":"126","DOI":"10.1097\/01.bth.0000185378.87246.61","article-title":"Algorithm for treatment of Apert hand","volume":"9","author":"Guero","year":"2005","journal-title":"Tech Hand Up Extrem Surg"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib43","first-page":"13","article-title":"Syndactyly in Apert syndrome. Utility of a prognostic classification","volume":"18","author":"Journeau","year":"1999","journal-title":"Ann Chir Main Memb Super"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib44","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1097\/00001665-199805000-00002","article-title":"The elbow in syndromic craniosynostosis","volume":"9","author":"Anderson","year":"1998","journal-title":"J Craniofac Surg"},{"issue":"3","key":"10.1016\/j.jhsa.2026.02.015_bib45","first-page":"197","article-title":"The Apert foot: anatomical changes, classification, thoughts on surgical treatment","volume":"54","author":"Piza-Katzer","year":"2022","journal-title":"Handchir Mikrochir Plast Chir"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib46","doi-asserted-by":"crossref","first-page":"1104","DOI":"10.1097\/SCS.0b013e318258814a","article-title":"Apert syndrome: quality of life and challenges of a management protocol in Brazil","volume":"23","author":"Raposo-Amaral","year":"2012","journal-title":"J Craniofac Surg"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib47","doi-asserted-by":"crossref","first-page":"182e","DOI":"10.1097\/01.prs.0000437260.31693.75","article-title":"Patient-reported quality of life in highest-functioning Apert and Crouzon syndromes: a comparative study","volume":"133","author":"Raposo-Amaral","year":"2014","journal-title":"Plast Reconstr Surg"},{"issue":"6","key":"10.1016\/j.jhsa.2026.02.015_bib48","doi-asserted-by":"crossref","first-page":"1510","DOI":"10.1097\/SCS.0000000000002886","article-title":"The quality of life in adult patients with syndromic craniosynostosis from their perspective","volume":"27","author":"Lloyd","year":"2016","journal-title":"J Craniofac Surg"},{"issue":"12","key":"10.1016\/j.jhsa.2026.02.015_bib49","doi-asserted-by":"crossref","first-page":"1972","DOI":"10.1016\/j.bjps.2010.01.036","article-title":"Health-related quality of life in children and adolescents with syndromic craniosynostosis","volume":"63","author":"Bannink","year":"2010","journal-title":"J Plast Reconstr Aesthet Surg"},{"key":"10.1016\/j.jhsa.2026.02.015_bib50","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1186\/s41687-024-00774-0","article-title":"Agreement between child self-report and parent-proxy report for functioning in pediatric chronic pain","volume":"8","author":"Hanania","year":"2024","journal-title":"J Patient Rep Outcomes"},{"issue":"2","key":"10.1016\/j.jhsa.2026.02.015_bib51","doi-asserted-by":"crossref","first-page":"177","DOI":"10.1002\/art.21840","article-title":"Level of agreement between children, parents, and physicians in rating pain intensity in juvenile idiopathic arthritis","volume":"55","author":"Garcia-Munitis","year":"2006","journal-title":"Arthritis Rheum"},{"issue":"9","key":"10.1016\/j.jhsa.2026.02.015_bib52","doi-asserted-by":"crossref","first-page":"488","DOI":"10.5435\/JAAOS-D-24-01046","article-title":"Do patient-answered versus caregiver-answered patient-reported outcomes differ in pediatric upper extremity fracture care?","volume":"33","author":"Smelley","year":"2025","journal-title":"J Am Acad Orthop Surg"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib53","doi-asserted-by":"crossref","first-page":"168","DOI":"10.1016\/j.pmn.2005.08.004","article-title":"Validity of parent ratings as proxy measures of pain in children with cognitive impairment","volume":"6","author":"Voepel-Lewis","year":"2005","journal-title":"Pain Manag Nurs"},{"issue":"1","key":"10.1016\/j.jhsa.2026.02.015_bib54","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1155\/2014\/736053","article-title":"Patient versus parental perceptions about pain and disability in children and adolescents with a variety of chronic pain conditions","volume":"19","author":"Vetter","year":"2014","journal-title":"Pain Res Manag"},{"issue":"9","key":"10.1016\/j.jhsa.2026.02.015_bib55","doi-asserted-by":"crossref","first-page":"e949","DOI":"10.1097\/BPO.0000000000002223","article-title":"Patient-reported outcomes for children with unilateral congenital below elbow deficiency","volume":"42","author":"Shoghi","year":"2022","journal-title":"J Pediatr Orthop"},{"issue":"4","key":"10.1016\/j.jhsa.2026.02.015_bib56","doi-asserted-by":"crossref","DOI":"10.1002\/alz.70169","article-title":"Factors associated with non-participation in the Healthy Cognitive Ageing Project","volume":"21","author":"Assaad","year":"2025","journal-title":"Alzheimers Dement"}],"container-title":["The Journal of Hand Surgery"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0363502326001632?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0363502326001632?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,8,20]],"date-time":"2026-08-20T20:57:39Z","timestamp":1787259459000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0363502326001632"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7]]},"references-count":48,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2026,7]]}},"alternative-id":["S0363502326001632"],"URL":"https:\/\/doi.org\/10.1016\/j.jhsa.2026.02.015","relation":{},"ISSN":["0363-5023"],"issn-type":[{"value":"0363-5023","type":"print"}],"subject":[],"published":{"date-parts":[[2026,7]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Health-Related Quality of Life Outcomes in Pediatric Patients With Apert Syndrome","name":"articletitle","label":"Article Title"},{"value":"The Journal of Hand Surgery","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jhsa.2026.02.015","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2026 by the American Society for Surgery of the Hand. All rights are reserved, including those for text and data mining, AI training, and similar technologies.","name":"copyright","label":"Copyright"}]}}