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 group, CONNECT study consortium COS development group, S M Hermelijn, C M Kersten, J M Schnater, R M H Wijnen, H A W M Tiddens, S C M Cochius, J Suominen, M Pakarinen, L Martelius, S Heyman, D Vervloessem, H Steyaert, A Sgrò, P Gamba, M Schurink, S van der Heide, J Roukema, N Rikkers-Mutsaerts, S Terheggen-Lagro, S de Beer, E Haarman, H Till, G Singer, M Metzelder, P Sezen, L Desender, H Schaballie, N Cobanoglu, G Gollu, M Stanton, A Bonnard, R Sfeir, N Muthialu, D Mullassery, C Wallis, D Cox, P Bagolan, F Morini, C Mesas Burgos, P Conner, E Caffrey Osvald, C Bitkover, H Decaluwé, M Proesmans, M Boon, J Deprest, S Gartner, A Lain, P D Losty, I Sinha, I Yardley, M Singh, L Wessel, K Zahn, T Schaible, N Qvist, M Zampoli, G Aksnes, C K van der Ent, K M Winter-de Groot, R Peters, E Hannon, Q Jöbsis, M Bannier, 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 group, CONNECT study consortium COS development group, S M Hermelijn, C M Kersten, J M Schnater, R M H Wijnen, H A W M Tiddens, S C M Cochius, J Suominen, M Pakarinen, L Martelius, S Heyman, D Vervloessem, H Steyaert, A Sgrò, P Gamba, M Schurink, S van der Heide, J Roukema, N Rikkers-Mutsaerts, S Terheggen-Lagro, S de Beer, E Haarman, H Till, G Singer, M Metzelder, P Sezen, L Desender, H Schaballie, N Cobanoglu, G Gollu, M Stanton, A Bonnard, R Sfeir, N Muthialu, D Mullassery, C Wallis, D Cox, P Bagolan, F Morini, C Mesas Burgos, P Conner, E Caffrey Osvald, C Bitkover, H Decaluwé, M Proesmans, M Boon, J Deprest, S Gartner, A Lain, P D Losty, I Sinha, I Yardley, M Singh, L Wessel, K Zahn, T Schaible, N Qvist, M Zampoli, G Aksnes, C K van der Ent, K M Winter-de Groot, R Peters, E Hannon, Q Jöbsis, M Bannier

Abstract

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 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.

Keywords: neonatology; paediatric intensive & critical care; paediatric surgery; paediatric thoracic medicine; paediatric thoracic surgery.

Conflict of interest statement

Competing interests: None declared.

© Author(s) (or their employer(s)) 2021. Re-use permitted under CC BY. Published by BMJ.

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Source: PubMed

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