{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,21]],"date-time":"2025-09-21T17:18:28Z","timestamp":1758475108369,"version":"3.41.2"},"reference-count":44,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2022,12,22]],"date-time":"2022-12-22T00:00:00Z","timestamp":1671667200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,5,27]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>The importance of multidisciplinary long-term follow-up for adults born with esophageal atresia (EA) is increasingly recognized. Hence, a valid, condition-specific instrument to measure health-related quality of life (HRQoL) becomes imperative. This study aimed to develop and validate such an instrument for adults with EA. The Specific Quality of life in Esophageal atresia Adults (SQEA) questionnaire was developed through focus group-based item generation, pilot testing, item reduction and a multicenter, nationwide field test to evaluate the feasibility, reliability (internal and retest) and validity (structural, construct, criterion and convergent), in compliance with the consensus-based standards for the selection of health measurement instruments guidelines. After pilot testing (n\u2009=\u200942), items were reduced from 144 to 36 questions. After field testing (n\u2009=\u2009447), three items were discarded based on item-response theory results. The final SQEA questionnaire (33 items) forms a unidimensional scale generating an unweighted total score. Feasibility, internal reliability (Cronbach\u2019s alpha 0.94) and test\u2013retest agreement (intra-class coefficient 0.92) were good. Construct validity was discriminative for esophageal replacement (P\u2009&amp;lt;\u20090.001), dysphagia (P\u2009&amp;lt;\u20090.001) and airway obstruction (P\u2009=\u20090.029). Criterion validity showed a good correlation with dysphagia (area under the receiver operating characteristic 0.736). SQEA scores correlated well with other validated disease-specific HRQoL scales such as the GIQLI and SGRQ, but poorly with the more generic RAND-36. Overall, this first condition-specific instrument for EA adults showed satisfactory feasibility, reliability and validity. Additionally, it shows discriminative ability to detect disease burden. Therefore, the SQEA questionnaire is both a valid instrument to assess the HRQoL in EA adults and an interesting signaling tool, enabling clinicians to recognize more severely affected patients.<\/jats:p>","DOI":"10.1093\/dote\/doac088","type":"journal-article","created":{"date-parts":[[2022,12,23]],"date-time":"2022-12-23T07:36:42Z","timestamp":1671781002000},"source":"Crossref","is-referenced-by-count":7,"title":["Development and validation of a condition-specific quality of life instrument for adults with esophageal atresia: the SQEA questionnaire"],"prefix":"10.1093","volume":"36","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9921-7776","authenticated-orcid":false,"given":"Chantal A","family":"ten Kate","sequence":"first","affiliation":[{"name":"Erasmus University Medical Centre\u2014Sophia Children\u2019s Hospital Department of Pediatric Surgery and Intensive Care Children, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nadine M","family":"Teunissen","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre\u2014Sophia Children\u2019s Hospital Department of Pediatric Surgery and Intensive Care Children, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Joost","family":"van Rosmalen","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre Department of Biostatistics, , Rotterdam, The Netherlands"},{"name":"Erasmus University Medical Centre Department of Epidemiology, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lieke S","family":"Kamphuis","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre Department of Pulmonology, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michiel P","family":"van Wijk","sequence":"additional","affiliation":[{"name":"Amsterdam University Medical Centers\u2014Emma Children\u2019s Hospital Department of Pediatric Gastroenterology and Nutrition, , Amsterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maja","family":"Joosten","sequence":"additional","affiliation":[{"name":"Radboud University Medical Center\u2014Amalia Children\u2019s Hospital Department of Pediatric Surgery, , Nijmegen, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"E Sofie","family":"van Tuyll van Serooskerken","sequence":"additional","affiliation":[{"name":"Wilhelmina Children\u2019s Hospital\u2014University Medical Center Utrecht Department of Pediatric Surgery, , Utrecht, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ren\u00e9","family":"Wijnen","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre\u2014Sophia Children\u2019s Hospital Department of Pediatric Surgery and Intensive Care Children, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hanneke","family":"IJsselstijn","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre\u2014Sophia Children\u2019s Hospital Department of Pediatric Surgery and Intensive Care Children, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andr\u00e9 B","family":"Rietman","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre\u2014Sophia Children\u2019s Hospital Department of Pediatric Surgery and Intensive Care Children, , Rotterdam, The Netherlands"},{"name":"Erasmus University Medical Centre\u2014Sophia Children's Hospital Department of Child and Adolescent Psychiatry\/Psychology, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9103-9757","authenticated-orcid":false,"given":"Manon C W","family":"Spaander","sequence":"additional","affiliation":[{"name":"Erasmus University Medical Centre Department of Gastroenterology and Hepatology, , Rotterdam, The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2022,12,22]]},"reference":[{"issue":"3","key":"2023052911454930800_ref1","doi-asserted-by":"crossref","first-page":"219","DOI":"10.1055\/s-0033-1347912","article-title":"Long-term outcome of esophageal anastomosis","volume":"23","author":"Rintala","year":"2013","journal-title":"Eur J Pediatr Surg"},{"issue":"4","key":"2023052911454930800_ref2","doi-asserted-by":"crossref","DOI":"10.1093\/bjsopen\/zrab069","article-title":"Barrett\u2019s oesophagus and oesophageal cancer following oesophageal atresia repair: a systematic review","volume":"5","author":"Tullie","year":"2021","journal-title":"BJS Open"},{"volume-title":"WHOQOL: Measuring Quality of Life","year":"1997","author":"World Health Organization 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