Children (Jul 2022)
The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey
- Casper M. Kersten,
- Sergei M. Hermelijn,
- Dhanya Mullassery,
- Nagarajan Muthialu,
- Nazan Cobanoglu,
- Silvia Gartner,
- Pietro Bagolan,
- Carmen Mesas Burgos,
- Alberto Sgrò,
- Stijn Heyman,
- Holger Till,
- Janne Suominen,
- Maarten Schurink,
- Liesbeth Desender,
- Paul Losty,
- Henri Steyaert,
- Suzanne Terheggen-Lagro,
- Martin Metzelder,
- Arnaud Bonnard,
- Rony Sfeir,
- Michael Singh,
- Iain Yardley,
- Noor R. V. M. Rikkers-Mutsaerts,
- Cornelis K. van der Ent,
- Niels Qvist,
- Des W. Cox,
- Robert Peters,
- Michiel A. G. E. Bannier,
- Lucas Wessel,
- Marijke Proesmans,
- Michael Stanton,
- Edward Hannon,
- Marco Zampoli,
- Francesco Morini,
- Harm A. W. M. Tiddens,
- René M. H. Wijnen,
- Johannes M. Schnater
Affiliations
- Casper M. Kersten
- Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, 3015 GD Rotterdam, The Netherlands
- Sergei M. Hermelijn
- Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, 3015 GD Rotterdam, The Netherlands
- Dhanya Mullassery
- Department of Pediatric Surgery, Great Ormond Street Hospital, London WC1N 3JH, UK
- Nagarajan Muthialu
- Department of Pediatric Surgery, Great Ormond Street Hospital, London WC1N 3JH, UK
- Nazan Cobanoglu
- Department of Pediatric Pulmonology, Ankara University School of Medicine, 06620 Ankara, Turkey
- Silvia Gartner
- Department of Pediatric Pulmonology and Cystic Fibrosis, Hospital Universitari Vall d’Hebron, 08035 Barcelona, Spain
- Pietro Bagolan
- Department of Fetus, Newborn and Infant, Bambino Gesù Pediatric Hospital, Tor Vergata University, 00165 Rome, Italy
- Carmen Mesas Burgos
- Department of Pediatric Surgery, Karolinska Institutet, 171 77 Stockholm, Sweden
- Alberto Sgrò
- Department of Pediatric Surgery, University Hospital Padua, 35128 Padua, Italy
- Stijn Heyman
- Department of Pediatric Surgery, ZNA-GZA Children’s Hospital, 2020 Antwerp, Belgium
- Holger Till
- Department of Pediatric Surgery, Medical University of Graz, 8036 Graz, Austria
- Janne Suominen
- Department of Pediatric Surgery, Helsinki Children’s Hospital, 00290 Helsinki, Finland
- Maarten Schurink
- Department of Pediatric Surgery, Radboud University Medical Centre, Amalia Children’s Hospital, 3525 GA Nijmegen, The Netherlands
- Liesbeth Desender
- Department of Pediatric Surgery, Ghent University Hospital, 9000 Ghent, Belgium
- Paul Losty
- Department of Pediatric Surgery, Institute of Life Course and Medical Sciences, University of Liverpool, Liverpool L69 72B, UK
- Henri Steyaert
- Department of Pediatric Surgery, Queen Fabiola Children’s University Hospital, 1020 Brussels, Belgium
- Suzanne Terheggen-Lagro
- Department of Pediatric Pulmonology, Emma Children’s Hospital Amsterdam University Medical Centers, 1105 AZ Amsterdam, The Netherlands
- Martin Metzelder
- Department of Pediatric Surgery, Medizinische Universität Wien, 1090 Vienna, Austria
- Arnaud Bonnard
- Department of Pediatric Surgery, Hôpital Universitaire Robert Debré, 75019 Paris, France
- Rony Sfeir
- Department of Pediatric Surgery, Hôpital Jeanne de Flandre, 59000 Lille, France
- Michael Singh
- Department of Pediatric Surgery, Birmingham Children’s Hospital, Birmingham B4 6NH, UK
- Iain Yardley
- Department of Pediatric Surgery, Evelina London Children’s Hospital, London SE1 7EH, UK
- Noor R. V. M. Rikkers-Mutsaerts
- Department of Pediatric Pulmonology, Leiden University Medical Centre, 2033 ZA Leiden, The Netherlands
- Cornelis K. van der Ent
- Department of Pediatric Pulmonology, University Medical Centre Utrecht, 3584 CX Utrecht, The Netherlands
- Niels Qvist
- Department of Pediatric Surgery, Odense University Hospital, 5000 Odense, Denmark
- Des W. Cox
- Department of Pediatric Pulmonology, Children’s Health Ireland at Crumlin, D12 N512 Dublin, Ireland
- Robert Peters
- Department of Pediatric Surgery, Royal Manchester Children’s Hospital, Manchester M13 9WL, UK
- Michiel A. G. E. Bannier
- Department of Pediatric Pulmonology, Maastricht University Medical Centre, 6229 HX Maastricht, The Netherlands
- Lucas Wessel
- Department of Pediatric Surgery, University Hospital Mannheim, 68167 Mannheim, Germany
- Marijke Proesmans
- Department of Pediatric Pulmonology, University Hospital Leuven, 3000 Leuven, Belgium
- Michael Stanton
- Department of Pediatric Surgery, University Hospital Southampton, Southampton SO16 6YD, UK
- Edward Hannon
- Department of Pediatric Surgery, Leeds Children’s Hospital, Leeds LS1 3EX, UK
- Marco Zampoli
- Department of Pediatric Pulmonology, Red Cross War Memorial Children’s Hospital, Cape Town 7700, South Africa
- Francesco Morini
- Department of Neonatal Surgery, Azienda Ospedaliero Universitaria Meyer, 50139 Florence, Italy
- Harm A. W. M. Tiddens
- Department of Pediatric Pulmonology and Allergology, Department of Radiology, Erasmus MC Sophia Children’s Hospital, 3015 GD Rotterdam, The Netherlands
- René M. H. Wijnen
- Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, 3015 GD Rotterdam, The Netherlands
- Johannes M. Schnater
- Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, 3015 GD Rotterdam, The Netherlands
- DOI
- https://doi.org/10.3390/children9081153
- Journal volume & issue
-
Vol. 9,
no. 8
p. 1153
Abstract
Consensus on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM) is lacking, and comparison between studies remains difficult due to a large variety in outcome measures. We aimed to define a core outcome set (COS) for pediatric patients with an asymptomatic CPAM. An online, three-round Delphi survey was conducted in two stakeholder groups of specialized caregivers (surgeons and non-surgeons) in various European centers. Proposed outcome parameters were scored according to level of importance, and the final COS was established through consensus. A total of 55 participants (33 surgeons, 22 non-surgeons) from 28 centers in 13 European countries completed the three rounds and rated 43 outcome parameters. The final COS comprises seven outcome parameters: respiratory insufficiency, surgical complications, mass effect/mediastinal shift (at three time-points) and multifocal disease (at two time-points). The seven outcome parameters included in the final COS reflect the diversity in priorities among this large group of European participants. However, we recommend the incorporation of these outcome parameters in the design of future studies, as they describe measurable and validated outcomes as well as the accepted age at measurement.
Keywords
- congenital lung abnormalities
- congenital pulmonary airway malformation
- core outcome set
- outcome parameters
- consensus
