Abstract
Long-term digestive, respiratory, and neurological morbidity is significant in children who have undergone surgery for esophageal atresia (EA), especially after staged repair for long-gap EA. Risk factors for morbidity after primary repair (non-long-gap populations) have been less documented. We investigated peri- and neonatal factors associated with unfavorable outcomes in children 2 years after primary esophageal anastomosis. This was a single-center retrospective study, based on neonatal, surgical, and pediatric records of children born between December 1, 2002, and December 31, 2018, and followed up to age 2 years. The primary endpoint was unfavorable outcome at 2 years of age, defined by death or survival with severe respiratory, digestive, or neurologic morbidity. Univariate analyses followed by logistic regression analyses were performed to identify the peri- and neonatal risk factors of unfavorable outcomes among survivors at discharge. A total of 150 neonates were included (mean birth weight 2520 ± 718 g, associated malformations 61%); at age 2, 45 (30%) had one or more severe morbidities and 11 had died during the neonatal stay and 2 after discharge (8.7% deaths). In multivariate analyses of the 139 survivors at discharge, duration of ventilatory support (invasive and non-invasive) for more than 8 days (OR 3.74; CI95% [1.68–8.60]; p = 0.001) and achievement of full oral feeding before hospital discharge (OR 0.20; CI95% [0.06–0.56]; p = 0.003) were independently associated with adverse outcome after adjustment for sex, preterm birth, associated heart defect, any surgical complication, and the occurrence of more than one nosocomial infections during the neonatal stay.
Conclusions: Post-operative ventilation and feeding management strategies may represent an opportunity for quality-of-care improvement to positively impact long-term outcomes after primary esophageal atresia repair.
What is Known: • Children operated on for esophageal atresia experience long-term digestive, respiratory, and neurologic morbidity, especially after multiple-stage esophageal repair. • Exclusive oral feeding at discharge is associated with a decreased risk of medical complications in the first years of life, in studies including all types of esophageal atresia repair. Outcomes of children after primary repair (non-long gap populations) have been less documented. | |
What is New: • In our retrospective cohort of children with one-stage esophageal atresia repair, ventilatory support for more than 8 days and inability to achieve full oral feeding before hospital discharge in the neonatal period were independently associated with adverse digestive, respiratory, and neurologic outcomes at 2 years in survivors. • Both these factors are potentially modifiable, representing an opportunity for quality-of-care improvement to positively impact long-term outcomes. These results might also help identify children at risk of unfavorable evolution, to customize a multi-disciplinary follow-up program. |
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The data that support the findings of this study are not publicly available due to their containing information that could compromise the privacy of research participants but are available from the corresponding author upon reasonable request.
Abbreviations
- EA:
-
Esophageal atresia
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Acknowledgements
The authors thank the “Association pour la Recherche et la Formation En Néonatologie (ARFEN)” for its technical support.
Funding
EKD received a grant from the Foundation Université Paris Cité, called “Sauver la Vie 2020.” The funders had no role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript.
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EKD, MLG, VR, and VA designed the study. NCL, MLG, and EKD collected and analyzed the data. NCL and EKD drafted the article. FL, AG, VA, VR, and AL critically revised the manuscript and provided significant scientific input. All authors read and approved the final manuscript.
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Patients and their families were informed by a note at the end of all reports that their child’s clinical data could be collected for research purposes and of their right to decline such research. According to French law, the study was declared to the National Data Protection Authority (Commission Nationale de l’Informatique et des Libertés) as meeting the reference methodology (MR-004) (https://www.cnil.fr/fr/declaration/mr-004-recherches-nimpliquant-pas-la-personne-humaine-etudes-et-evaluations-dans-le). The protocol was reviewed, approved, and registered in the institutional register of data processing of the Assistance Publique-Hopitaux de Paris under the number 2021–0312112531. The reference methodology MR-004 provides a framework for the processing of personal data for the purposes of study, evaluation, or research not involving the human person. More specifically, this concerns studies that do not meet the definition of research involving the human person, in particular, studies concerning the reuse of data, which is the case of our study. The data controller undertakes to collect only data that is strictly necessary and relevant to the objectives of the research. The reference methodology MR-004 does not require an approval by an ethics committee provided an information is given that clinical data is collected for research purposes and information of patient’s right to oppose to such research and right of access, rectification, or deletion of their data. Data were anonymized for processing. The processing of personal data was carried out in accordance with the EU Regulation 2016/679 of the European Parliament and of the Council of 27 April 2016 on the protection of individuals with regard to the processing of personal data and on the free circulation of such data.
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Lefèvre, N.C., Le Gouez, M., Lesage, F. et al. Peri- and neonatal factors influencing mortality and morbidity 2 years after esophageal atresia primary repair: a single center retrospective study. Eur J Pediatr 182, 889–897 (2023). https://doi.org/10.1007/s00431-022-04740-0
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DOI: https://doi.org/10.1007/s00431-022-04740-0