Variability in the Reporting of Baseline Characteristics, Treatment, and Outcomes in Esophageal Atresia Publications: A Systematic Review

Nadine Teunissen; Julia Brendel; Simon Eaton; Nigel Hall; Rebecca Thursfield; Ernest L.W. van Heurn; Benno Ure; Rene Wijnen

doi:10.1055/s-0042-1758828

European Journal of Pediatric Surgery, Table of Contents

Eur J Pediatr Surg 2023; 33(02): 129-137
DOI: 10.1055/s-0042-1758828

Review Article

Variability in the Reporting of Baseline Characteristics, Treatment, and Outcomes in Esophageal Atresia Publications: A Systematic Review

Nadine Teunissen

¹Department of Pediatric Surgery and Pediatric Intensive Care Unit, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands

²Dutch Institute for Clinical Auditing, European Pediatric Surgical Audit, Leiden, the Netherlands

,

Julia Brendel

³Department of Pediatric Surgery, Hannover Medical School, Hannover, Niedersachsen, Germany

,

Simon Eaton

¹Department of Pediatric Surgery and Pediatric Intensive Care Unit, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands

⁴Developmental Biology and Cancer Research and Teaching Programme, UCL Great Ormond Street Institute of Child Health, London, United Kingdom of Great Britain and Northern Ireland

,

Nigel Hall

⁵University Surgery Unit, Faculty of Medicine, University of Southampton, Southampton, United Kingdom of Great Britain and Northern Ireland

,

Rebecca Thursfield

⁶Department of Pediatric Respiratory Medicine, Alder Hey Children's NHS Foundation Trust, Liverpool, United Kingdom of Great Britain and Northern Ireland

,

Ernest L.W. van Heurn

⁷Department of Pediatric Surgery, Emma's Children's Hospital, Amsterdam UMC, Amsterdam, the Netherlands

⁸Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands

,

Benno Ure

³Department of Pediatric Surgery, Hannover Medical School, Hannover, Niedersachsen, Germany

,

Rene Wijnen

¹Department of Pediatric Surgery and Pediatric Intensive Care Unit, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands

› Author Affiliations

Abstract

Introduction As survival rates of infants born with esophageal atresia (EA) have improved considerably, research interests are shifting from viability to morbidity and longer-term outcomes. This review aims to identify all parameters studied in recent EA research and determine variability in their reporting, utilization, and definition.

Materials and Methods Following PRISMA guidelines, we performed a systematic review of literature regarding the main EA care process, published between 2015 and 2021, combining the search term “esophageal atresia” with “morbidity,” “mortality,” “survival,” “outcome,” or “complication.” Described outcomes were extracted from included publications, along with study and baseline characteristics.

Results From 209 publications that met the inclusion criteria, 731 studied parameters were extracted and categorized into patient characteristics (n = 128), treatment and care process characteristics (n = 338), and outcomes (n = 265). Ninety-two of these were reported in more than 5% of included publications. Most frequently reported characteristics were sex (85%), EA type (74%), and repair type (60%). Most frequently reported outcomes were anastomotic stricture (72%), anastomotic leakage (68%), and mortality (66%).

Conclusion This study demonstrates considerable heterogeneity of studied parameters in EA research, emphasizing the need for standardized reporting to compare results of EA research. Additionally, the identified items may help develop an informed, evidence-based consensus on outcome measurement in esophageal atresia research and standardized data collection in registries or clinical audits, thereby enabling benchmarking and comparing care between centers, regions, and countries.

Keywords

esophageal atresia - outcome - characteristics

Full Text

References

References
1 Nassar N, Leoncini E, Amar E. et al. Prevalence of esophageal atresia among 18 international birth defects surveillance programs. Birth Defects Res A Clin Mol Teratol 2012; 94 (11) 893-899
2 Bell JC, Baynam G, Bergman JEH. et al. Survival of infants born with esophageal atresia among 24 international birth defects surveillance programs. Birth Defects Res 2021; 113 (12) 945-957
3 Zimmer J, Eaton S, Murchison LE, De Coppi P, Ure BM, Dingemann C. State of play: eight decades of surgery for esophageal atresia. Eur J Pediatr Surg 2019; 29 (01) 39-48
4 Teunissen NM. ERNICA | EPSA Registry. Published 2022. Accessed June 10, 2022 at: https://ern-ernica.eu/registry/
5 Dingemann C, Eaton S, Aksnes G. et al. ERNICA consensus conference on the management of patients with esophageal atresia and tracheoesophageal fistula: diagnostics, preoperative, operative, and postoperative management. Eur J Pediatr Surg 2020; 30 (04) 326-336
6 Krishnan U, Mousa H, Dall'Oglio L. et al. ESPGHAN-NASPGHAN guidelines for the evaluation and treatment of gastrointestinal and nutritional complications in children with esophageal atresia-tracheoesophageal fistula. J Pediatr Gastroenterol Nutr 2016; 63 (05) 550-570
7 Feng X, Martynov I, Suttkus A, Lacher M, Mayer S. Publication trends and global collaborations on esophageal atresia research: a bibliometric study. Eur J Pediatr Surg 2021; 31 (02) 164-171
8 Ocelot study. Research projects endorsed by TOFS. Published 2022. . Accessed May 10, 2022 at: https://tofs.org.uk/oa-tof-information/oa-tof-research/ocelot-study/
9 Page MJ, Moher D, Bossuyt PM. et al. PRISMA 2020 explanation and elaboration: updated guidance and exemplars for reporting systematic reviews. BMJ 2021; 372: n160
10 Flieder S, Dellenmark-Blom M, Witt S. et al. Generic health-related quality of life after repair of esophageal atresia and its determinants within a German-Swedish cohort. Eur J Pediatr Surg 2019; 29 (01) 75-84
11 Bakal U, Ersoz F, Eker I, Sarac M, Aydin M, Kazez A. Long-term prognosis of patients with esophageal atresia and/or tracheoesophageal fistula. Indian J Pediatr 2016; 83 (05) 401-404
12 Gallo G, van Tuyll van Serooskerken ES, Tytgat SHAJ. et al. Quality of life after esophageal replacement in children. J Pediatr Surg 2021; 56 (02) 239-244
13 Hannon E, Eaton S, Curry JI, Kiely EM, Spitz L, De Coppi P. Outcomes in adulthood of gastric transposition for complex and long gap esophageal atresia. J Pediatr Surg 2020; 55 (04) 639-645
14 Mikkelsen A, Boye B, Diseth TH. et al. Traumatic stress, mental health, and quality of life in adolescents with esophageal atresia. J Pediatr Surg 2022; 57 (07) 1423-1431
15 Okuyama H, Tazuke Y, Uenoa T. et al. Long-term morbidity in adolescents and young adults with surgically treated esophageal atresia. Surg Today 2017; 47 (07) 872-876
16 Vergouwe FWT, IJsselstijn H, Biermann K. et al. High prevalence of Barrett's esophagus and esophageal squamous cell carcinoma after repair of esophageal atresia. Clin Gastroenterol Hepatol 2018; 16 (04) 513-521.e6
17 Kumari V, Joshi P, Dhua AK. et al. Developmental status of children operated for esophageal atresia with or without tracheoesophageal fistula along with maternal stress, their quality of life, and coping abilities at AIIMS, New Delhi. Eur J Pediatr Surg 2019; 29 (01) 125-131
18 di Natale A, Brestel J, Mauracher AA. et al. Long-term outcomes and health-related quality of life in a Swiss patient group with esophageal atresia. Eur J Pediatr Surg 2022; 32 (04) 334-345
19 Sreeram II, Ten Kate CA, van Rosmalen J. et al. Patient-reported outcome measures and clinical outcomes in children with foregut anomalies. Children (Basel) 2021; 8 (07) 587
20 Tannuri ACA, Angelo SS, Takyi P, da Silva AR, Tannuri U. Esophageal substitution or esophageal elongation procedures in patients with complicated esophageal atresia? Results of a comparative study. J Pediatr Surg 2021; 56 (05) 933-937
21 van Hoorn CE, van der Cammen-van Zijp MHM, Stolker RJ, van Rosmalen J, Wijnen RMH, de Graaff JC. Associations of perioperative characteristics with motor function in preschool children born with esophageal atresia. Paediatr Anaesth 2021; 31 (08) 854-862
22 Bal HS, Sen S, Karl S, Mathai J, Thomas RJ. An assessment of quality of life of operated cases of esophageal atresia in the community. J Indian Assoc Pediatr Surg 2016; 21 (03) 131-138
23 Dingemann J, Szczepanski R, Ernst G. et al. Transition of patients with esophageal atresia to adult care: results of a transition-specific education program. Eur J Pediatr Surg 2017; 27 (01) 61-67
24 Miyano G, Seo S, Nakamura H. et al. Changes in quality of life from infancy to school age after esophagoesophagostomy for tracheoesophageal fistula: thoracotomy versus thoracoscopy. Pediatr Surg Int 2017; 33 (10) 1087-1090
25 Amin R, Knezevich M, Lingongo M. et al. Long-term quality of life in neonatal surgical disease. Ann Surg 2018; 268 (03) 497-505
26 Svoboda E, Fruithof J, Widenmann-Grolig A. et al. A patient led, international study of long term outcomes of esophageal atresia: EAT 1. J Pediatr Surg 2018; 53 (04) 610-615
27 Youn JK, Park T, Kim SH. et al. Prospective evaluation of clinical outcomes and quality of life after gastric tube interposition as esophageal reconstruction in children. Medicine (Baltimore) 2018; 97 (52) e13801
28 Dellenmark-Blom M, Abrahamsson K, Quitmann JH. et al. Development and pilot-testing of a condition-specific instrument to assess the quality-of-life in children and adolescents born with esophageal atresia. Dis Esophagus 2017; 30 (07) 1-9
29 Ten Kate CA, Teunissen NM, van Rosmalen J. et al. Development and validation of a condition-specific quality of life instrument for adults with esophageal atresia: the SQEA questionnaire. Dis Esophagus. Published online December 2022
30 Williamson PR, Altman DG, Bagley H. et al. The COMET handbook: version 1.0. Trials 2017; 18 (Suppl. 03) 280

Supplementary Material

Supplementary Material