Core outcome set for perinatal interventions for congenital diaphragmatic hernia

S. Vergote, F. R. De Bie, J. M.N. Duffy, J. Bosteels, A. Benachi, B. Power, F. Meijer, H. L. Hedrick, C. J. Fernandes, I. K.M. Reiss, P. De Coppi, K. P. Lally, J. A. Deprest*

*Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review

1 Citation (Scopus)

Abstract

Objective: To develop a core set of prenatal and neonatal outcomes for clinical studies evaluating perinatal interventions for congenital diaphragmatic hernia, using a validated consensus-building method. Methods: An international steering group comprising 13 leading maternal–fetal medicine specialists, neonatologists, pediatric surgeons, patient representatives, researchers and methodologists guided the development of this core outcome set. Potential outcomes were collected through a systematic review of the literature and entered into a two-round online Delphi survey. A call was made for stakeholders with experience of congenital diaphragmatic hernia to review the list and score outcomes based on their perceived relevance. Outcomes that fulfilled the consensus criteria defined a priori were discussed subsequently in online breakout meetings. Results were reviewed in a consensus meeting, during which the core outcome set was defined. Finally, the definitions, measurement methods and aspirational outcomes were defined in online and in-person definition meetings by a selection of 45 stakeholders. Results: Overall, 221 stakeholders participated in the Delphi survey and 198 completed both rounds. Fifty outcomes met the consensus criteria and were discussed and rescored by 78 stakeholders in the breakout meetings. During the consensus meeting, 93 stakeholders agreed eventually on eight outcomes, which constituted the core outcome set. Maternal and obstetric outcomes included maternal morbidity related to the intervention and gestational age at delivery. Fetal outcomes included intrauterine demise, interval between intervention and delivery and change in lung size in utero around the time of the intervention. Neonatal outcomes included neonatal mortality, pulmonary hypertension and use of extracorporeal membrane oxygenation. Definitions and measurement methods were formulated by 45 stakeholders, who also added three aspirational outcomes: duration of invasive ventilation, duration of oxygen supplementation and use of pulmonary vasodilators at discharge. Conclusions: We developed with relevant stakeholders a core outcome set for studies evaluating perinatal interventions in congenital diaphragmatic hernia. Its implementation should facilitate the comparison and combination of trial results, enabling future research to better guide clinical practice.

Original languageEnglish
Pages (from-to)374-382
Number of pages9
JournalUltrasound in Obstetrics and Gynecology
Volume62
Issue number3
Early online date26 Apr 2023
DOIs
Publication statusPublished - Sept 2023

Bibliographical note

ACKNOWLEDGMENTS:
The core outcome set for congenital diaphragmatic hernia steering group thank the stakeholders who completed the Delphi surveys and attended the breakout meetings, consensus meetings and definition meetings. We also acknowledge ERNICA for allowing us to present and discuss our data during their annual meetings. S.V. is supported by the Flanders Research Foundation (Fonds Wetenschappelijk Onderzoek Vlaanderen T002618N) and F.R.D.B. is supported by the Flanders Research Foundation (1S31720N). J.A.D. is supported by Great Ormond Street Hospital Charity fund.

Publisher Copyright:
© 2023 International Society of Ultrasound in Obstetrics and Gynecology.

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