Development of a core outcome set for congenital pulmonary airway malformations: study protocol of an international Delphi survey

Sergei Hermelijn, Casper Kersten, Dhanya Mullassery, Nagarajan Muthialu, Nazan Cobanoglu, Silvia Gartner, Pietro Bagolan, Carmen Mesas Burgos, Alberto Sgro, Stijn Heyman, Holger Till, Janne Suominen, Maarten Schurink, Liesbeth Desender, Paul Losty, Kjetil Ertresvag, Harm A. W. M. Tiddens, Rene M. H. Wijnen, Marco Schnater*, CONNECT study consortium COS development groupMichiel Bannier

*Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review



A worldwide lack of consensus exists on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM) even though the incidence is increasing. Either a surgical resection is performed or a wait-and-see policy is employed, depending on the treating physician. Management is largely based on expert opinion and scientific evidence is scarce. Wide variations in outcome measures are seen between studies making comparison difficult thus highlighting the lack of universal consensus in outcome measures as well. We aim to define a core outcome set which will include the most important core outcome parameters for paediatric patients with an asymptomatic CPAM.

Methods and analysis

This study will include a critical appraisal of the current literature followed by a three-stage Delphi process with two stakeholder groups. One surgical group including paediatric as well as thoracic surgeons, and a non-surgeon group including paediatric pulmonologists, intensive care and neonatal specialists. All participants will score outcome parameters according to their level of importance and the most important parameters will be determined by consensus.

Ethics and dissemination

Electronic informed consent will be obtained from all participants. Ethical approval is not required. After the core outcome set has been defined, we intend to design an international randomised controlled trial: the COllaborative Neonatal NEtwork for the first CPAM Trial, which will be aimed at determining the optimal management of patients with asymptomatic CPAM.

Original languageEnglish
Article number044544
Number of pages5
JournalBMJ Open
Issue number4
Publication statusPublished - 2021


  • paediatric intensive &amp
  • critical care
  • neonatology
  • paediatric thoracic medicine
  • paediatric surgery
  • paediatric thoracic surgery

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