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Oral Presentation - 25
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 1, U Krishnan 2, C Faure 3, T Kovesi 4, A C Koumbourlis 5, R Wijnen 6, L Dall’Oglio 7, D von Allmen 8, F Gottrand 9, P Lobos 10, M van Wijk 11, J Dingemann 12, J H Quitmann 13, M Aumar 14, A Widenmann 15, G Slater 15, T Soyer 16, C de Vos 17, M Dellenmark-Blom 18
1 Department of Pediatrics, Institute of Clinical Sciences, University of Gothenburg, Gothenburg, Sweden
2 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
3 Department of Pediatric Gastroenterology, Sainte-Justine Hospital, Montreal, Quebec, Canada; Université de Montréal, Montréal, QC, Canada
4 Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada
5 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
6 Department of Pediatric Surgery, Erasmus Medical Center-Sophia Children's Hospital, Rotterdam, the Netherlands
7 Gastroenterology and Nutrition Unit, Bambino Gesù Children’s Hospital, Rome, Italy
8 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
9 Univ. Lille, Reference Centre for rare œsophageal diseases, CHU Lille, Lille, France Division of Pediatric Gastroenterology, Hepatology and Nutrition, CHU Lille, Lille, France
10 Department of Pediatric Surgery, Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
11 Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital Amsterdam UMC, Amsterdam, the Netherlands
12 Centre of Pediatric Surgery, Hannover Medical School, Hannover, Germany
13 Hochschule für Angewandte Wissenschaften Hamburg (HAW Hamburg), Germany; Department of Medical Psychology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
14 Univ. Lille, Reference Centre for rare œsophageal diseases, CHU Lille, Lille, France
15 EAT (Esophageal Atresia Global Support Groups), Sommerrainstr. 61, 70374 Stuttgart, Germany
16 Department of Pediatric Surgery, Faculty of Medicine, Hacettepe University, Ankara, Turkiye
17 Tygerberg Hospital, Stellenbosch University, Cape Town, South Africa
18 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.