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Abstract
Background Congenital diaphragmatic hernia (CDH) is a rare birth defect with substantial mortality. This study aims to generate a population-based overview of CDH care and outcomes in the Netherlands. Moreover, it assesses interhospital variations between the two Dutch CDH expert centres.
Methods This study uses data from the Dutch branch of the European Pediatric Surgical Audit, a prospective clinical audit for congenital anomalies. Data of all patients with CDH treated between 2014 and 2021 were included for epidemiological analysis. For comparative analyses, patients presenting after the neonatal period or not treated in the two CDH expert centres were excluded. Identified interhospital variations were assessed using regression analysis.
Results In the study period, 283 children with CDH were born, resulting in a national prevalence rate of 2.06/10 000 live births. The patient population, treatment and outcomes at 1 year were comparable between the hospitals, except for length of hospital stay. Regression analysis identified the treating hospital as the strongest significant predictor thereof. Other factors associated with longer length of stay include the presence of other malformations, intrathoracic liver position on prenatal ultrasound, extracorporeal membrane oxygenation treatment, patch repair, complicated postoperative course and discharge to home rather than to another care facility.
Conclusion Outcomes of CDH care throughout the Netherlands are comparable. However, the length of stay differed between the two hospitals, also when adjusting for other covariates. Further qualitative analysis to explain this interhospital variation is indicated. Our findings underscore the potential of clinical auditing as a quality measurement tool in rare conditions.
- Epidemiology
- Mortality
- Paediatrics
Data availability statement
Data may be obtained from a third party and are not publicly available. Upon request, the study protocol, EPSA’s data dictionary and statistical code used for analyses in this manuscript are available from the corresponding author at n.teunissen@erasmusmc.nl.
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Data availability statement
Data may be obtained from a third party and are not publicly available. Upon request, the study protocol, EPSA’s data dictionary and statistical code used for analyses in this manuscript are available from the corresponding author at n.teunissen@erasmusmc.nl.
Footnotes
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Contributors NMT drafted the manuscript, and is the guarantor. NMT, HD, RMHW and IdB developed the protocol. All authors contributed to the accurate implementation of the protocol and read, provided feedback on and approved the final manuscript.
Funding The European Commission funded the EPSA|ERNICA Registry in the 3rd and 4th Health Programs (HP-PJ-219 and EU4H-2022-ERN-IBA). Additionally, the Dutch branch of EPSA is continuously supported by Zorgverzekeraars Nederland (ZN), a collaborative institute comprising 10 healthcare insurance companies in the Netherlands.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.
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