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Oral Presentation - 26
Is health-related quality of life in children born with esophageal atresia-tracheoesophageal fistula reduced compared with general population norms and what are the main associated factors? - a systematic literature review
A Abrahamsson*, U Krishnan**, C Faure***, T Kovesi****, A C Koumbourlis*****, R Wijnen******, L Dall’Oglio*******, D von Allmen********, F Gottrand*********, P Lobos**********, M van Wijk***********, J Dingemann************, J H Quitmann*************, M Aumar**************, A Widenmann***************, G Slater***************, T Soyer****************, C de Vos*****************, M Dellenmark-Blom******************
*Department of Pediatrics, Institute of Clinical Sciences, University of Gothenburg, Gothenburg, Sweden
**Department of Paediatric Gastroenterology, Sydney Children’s Hospital, Sydney, NSW, Australia; School of Women’s and Children’s Health, University of New South Wales, Sydney, Australia
***Department of Pediatric Gastroenterology, Sainte-Justine Hospital, Montreal, Quebec, Canada; Université de Montréal, Montréal, QC, Canada
****Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada
*****Division of Pulmonary & Sleep Medicine, Children's National Medical Center, Washington D.C, USA; George Washington University School of Medicine & Health Sciences, Washington D.C, USA
******Department of Pediatric Surgery, Erasmus Medical Center-Sophia Children's Hospital, Rotterdam, the Netherlands
*******Gastroenterology and Nutrition Unit, Bambino Gesù Children’s Hospital, Rome, Italy
********Department of Pediatric Surgery and Surgical Services, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA. Department of Surgery, University of Cincinnati College of Medicine, Cincinnati, OH, USA
*********Univ. Lille, Reference Centre for rare œsophageal diseases, CHU Lille, Lille, France Division of Pediatric Gastroenterology, Hepatology and Nutrition, CHU Lille, Lille, France
**********Department of Pediatric Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
***********Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital Amsterdam UMC, Amsterdam, the Netherlands
************Centre of Pediatric Surgery, Hannover Medical School, Hannover, Germany
*************Hochschule für Angewandte Wissenschaften Hamburg (HAW Hamburg), Germany; Department of Medical Psychology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
**************Univ. Lille, Reference Centre for rare œsophageal diseases, CHU Lille, Lille, France
***************EAT (Esophageal Atresia Global Support Groups), Sommerrainstr. 61, 70374 Stuttgart, Germany
****************Department of Pediatric Surgery, Faculty of Medicine, Hacettepe University, Ankara, Turkiye
*****************Tygerberg Hospital, Stellenbosch University, Cape Town, South Africa
******************Department of Pediatric Surgery, Queen Silvia Children’s Hospital, Gothenburg, Sweden Department of Women's and Children's Health, Karolinska Institutet, Stockholm, Sweden
Purpose: to review the literature on health-related quality of life (HRQoL) in children born with esophageal atresia-tracheoesophageal fistula (EA-TEF) compared with general norms and the main associated factors.
Method: Utilizing a PRISMA-compliant protocol, a literature search of articles using valid and reliable generic HRQoL questionnaires in EA-TEF children was conducted in five databases (PubMed/CINAHL/PsychINFO/Embase/Cochrane) from inception to September 2024. A meta-analysis of overall HRQoL in EA-TEF children compared with general norms was performed using a random-effects model, Cohen’s d for effect sizes (ESs) and I² statistics. Significant level was p<0.05.
Results: Sixteen articles (published 2010-2023) described generic HRQoL in 948 EA-TEF children (median 63/study, range 11-192). Five different HRQoL questionnaires were employed. Fourteen studies originated from Europe. 10/12 studies reported reduced levels of HRQoL in at least one domain (social/school/physical/psychological domains) and/or in overall HRQoL among EA-TEF children compared with general norms. In the metaanalysis, seven studies could be included. The pooled estimate showed that overall generic HRQoL scores were significantly lower in EA-TEF children compared with general norms in parent-reports (736 EA-TEF children vs 10513 controls, mean difference-3.34 [95%-CI -4.54;-2.14], I2=18.4%, p<0.01) and child-reports (460 EA-TEF children vs 6303 controls, mean difference -3.28 [95%-CI; -5.14; -1.23], I2=38.9%, p<0.01), with small ES (Cohen’s d<0.5). Concerning HRQoL domains, the children's school functioning was most frequently described as reduced (4/8 studies). The most common factors associated with worse HRQoL were gastro-esophageal reflux disease (GERD) in 5/5 studies investigating GERD, associated anomalies (3/4 studies) and prematurity (4/6 studies).
Conclusion: Generic HRQoL in EA-TEF children is significantly reduced compared with the general population, and associated with GERD, associated anomalies and prematurity. This needs consideration in clinical follow-up of children with EA-TEF.