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

doi:10.3390/children9081153

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 SfeirMichael 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^*

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

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.

Original language	English
Article number	1153
Number of pages	14
Journal	Children-Basel
Volume	9
Issue number	8
DOIs	https://doi.org/10.3390/children9081153
Publication status	Published - 30 Jul 2022

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10.3390/children9081153Licence: CC BY

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Kersten, C. M., Hermelijn, S. M., Mullassery, D., Muthialu, N., Cobanoglu, N., Gartner, S., Bagolan, P., Mesas Burgos, C., Sgrò, A., Heyman, S., Till, H., Suominen, J., Schurink, M., Desender, L., Losty, P., Steyaert, H., Terheggen-Lagro, S., Metzelder, M., Bonnard, A., ... Schnater, J. M. (2022). The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey. Children-Basel, 9(8), Article 1153. https://doi.org/10.3390/children9081153

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title = "The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey",

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.",

author = "Kersten, {Casper M} and Hermelijn, {Sergei M} and Dhanya Mullassery and Nagarajan Muthialu and Nazan Cobanoglu and Silvia Gartner and Pietro Bagolan and {Mesas Burgos}, Carmen and Alberto Sgr{\`o} and Stijn Heyman and Holger Till and Janne Suominen and Maarten Schurink and Liesbeth Desender and Paul Losty and Henri Steyaert and Suzanne Terheggen-Lagro and Martin Metzelder and Arnaud Bonnard and Rony Sfeir and Michael Singh and Iain Yardley and Rikkers-Mutsaerts, {Noor R V M} and {van der Ent}, {Cornelis K} and Niels Qvist and Cox, {Des W} and Robert Peters and Bannier, {Michiel A G E} and Lucas Wessel and Marijke Proesmans and Michael Stanton and Edward Hannon and Marco Zampoli and Francesco Morini and Tiddens, {Harm A W M} and Wijnen, {Ren{\'e} M H} and Schnater, {Johannes M}",

note = "Publisher Copyright: {\textcopyright} 2022 by the authors.",

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Kersten, CM, Hermelijn, SM, Mullassery, D, Muthialu, N, Cobanoglu, N, Gartner, S, Bagolan, P, Mesas Burgos, C, Sgrò, A, Heyman, S, Till, H, Suominen, J, Schurink, M, Desender, L, Losty, P, Steyaert, H, Terheggen-Lagro, S, Metzelder, M, Bonnard, A, Sfeir, R, Singh, M, Yardley, I, Rikkers-Mutsaerts, NRVM, van der Ent, CK, Qvist, N, Cox, DW, Peters, R, Bannier, MAGE, Wessel, L, Proesmans, M, Stanton, M, Hannon, E, Zampoli, M, Morini, F, Tiddens, HAWM, Wijnen, RMH & Schnater, JM 2022, 'The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey', Children-Basel, vol. 9, no. 8, 1153. https://doi.org/10.3390/children9081153

TY - JOUR

T1 - The Management of Asymptomatic Congenital Pulmonary Airway Malformation

T2 - Results of a European Delphi Survey

AU - Kersten, Casper M

AU - Hermelijn, Sergei M

AU - Mullassery, Dhanya

AU - Muthialu, Nagarajan

AU - Cobanoglu, Nazan

AU - Gartner, Silvia

AU - Bagolan, Pietro

AU - Mesas Burgos, Carmen

AU - Sgrò, Alberto

AU - Heyman, Stijn

AU - Till, Holger

AU - Suominen, Janne

AU - Schurink, Maarten

AU - Desender, Liesbeth

AU - Losty, Paul

AU - Steyaert, Henri

AU - Terheggen-Lagro, Suzanne

AU - Metzelder, Martin

AU - Bonnard, Arnaud

AU - Sfeir, Rony

AU - Singh, Michael

AU - Yardley, Iain

AU - Rikkers-Mutsaerts, Noor R V M

AU - van der Ent, Cornelis K

AU - Qvist, Niels

AU - Cox, Des W

AU - Peters, Robert

AU - Bannier, Michiel A G E

AU - Wessel, Lucas

AU - Proesmans, Marijke

AU - Stanton, Michael

AU - Hannon, Edward

AU - Zampoli, Marco

AU - Morini, Francesco

AU - Tiddens, Harm A W M

AU - Wijnen, René M H

AU - Schnater, Johannes M

PY - 2022/7/30

Y1 - 2022/7/30

N2 - 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.

AB - 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.

U2 - 10.3390/children9081153

DO - 10.3390/children9081153

M3 - Article

C2 - 36010044

SN - 2227-9067

VL - 9

JO - Children-Basel

JF - Children-Basel

IS - 8

M1 - 1153

ER -