TY - JOUR
T1 - Development of a core outcome set for congenital pulmonary airway malformations
T2 - study protocol of an international Delphi survey
AU - Hermelijn, Sergei
AU - Kersten, Casper
AU - Mullassery, Dhanya
AU - Muthialu, Nagarajan
AU - Cobanoglu, Nazan
AU - Gartner, Silvia
AU - Bagolan, Pietro
AU - Mesas Burgos, Carmen
AU - Sgro, Alberto
AU - Heyman, Stijn
AU - Till, Holger
AU - Suominen, Janne
AU - Schurink, Maarten
AU - Desender, Liesbeth
AU - Losty, Paul
AU - Ertresvag, Kjetil
AU - Tiddens, Harm A W M
AU - Wijnen, Rene M H
AU - Schnater, Marco
AU - CONNECT study consortium COS development group
AU - Haarman, EG
N1 - Publisher Copyright: © 2021 BMJ Publishing Group. All rights reserved.
PY - 2021/4/12
Y1 - 2021/4/12
N2 - 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.
AB - 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.
KW - neonatology
KW - paediatric intensive & critical care
KW - paediatric surgery
KW - paediatric thoracic medicine
KW - paediatric thoracic surgery
UR - http://www.scopus.com/inward/record.url?scp=85104258208&partnerID=8YFLogxK
U2 - https://doi.org/10.1136/bmjopen-2020-044544
DO - https://doi.org/10.1136/bmjopen-2020-044544
M3 - Article
C2 - 33846152
SN - 2044-6055
VL - 11
SP - e044544
JO - BMJ Open
JF - BMJ Open
IS - 4
M1 - e044544
ER -