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

CONNECT study consortium COS development group, Sergei Hermelijn, Casper Kersten, S Mullasery, Nagarajan Muthialu, A Cobanoglu, S Gartner, Pietro Bagolan, Carmen M. Burgos, A Sgro, Stijn Heyman, H Till, M Schurink, Liesbeth Desender, Paul Losty, K Ertresvag, H.A.W.M. Tiddens, Rene Wijnen, Marco Schnater*

*Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review

4 Citations (Scopus)
7 Downloads (Pure)


Introduction 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
Methods and analysis This study will include a critical
appraisal of the current literature followed by a threestage 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 numbere044544
Pages (from-to)1-5
Number of pages5
JournalBMJ Open
Issue number4
Publication statusPublished - 12 Apr 2021


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