Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol

Introduction Heterogeneity in reported outcomes of infants with oesophageal atresia (OA) with or without tracheo-oesophageal fistula (TOF) prevents effective data pooling. Core outcome sets (COS) have been developed for many conditions to standardise outcome reporting, facilitate meta-analysis and i...

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Main Authors: Ducey, Jonathan, Lansdale, Nick, Gorst, Sarah, Bray, Lucy, Teunissen, Nadine, Cullis, Paul, Faulkner, Julia, Gray, Victoria, Gammino, Lucia Gutierrez, Slater, Graham, Baird, Laura, Adams, Alex, Brendel, Julia, Donne, Adam, Folaranmi, Eniola, Hopwood, Laura, Long, Anna-May, Losty, Paul D., Benscoter, Dan, de Vos, Corné, King, Sebastian, Kovesi, Tom, Krishnan, Usha, Nah, Shireen Anne, Ong, Lin Yin, Rutter, Mike, Teague, Warwick J., Zorn, Aaron M., Hall, Nigel J., Thursfield, Rebecca
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Published: BMJ Publishing Group 2024
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Online Access:http://eprints.um.edu.my/44812/
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spelling my.um.eprints.448122024-07-04T04:52:17Z http://eprints.um.edu.my/44812/ Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol Ducey, Jonathan Lansdale, Nick Gorst, Sarah Bray, Lucy Teunissen, Nadine Cullis, Paul Faulkner, Julia Gray, Victoria Gammino, Lucia Gutierrez Slater, Graham Baird, Laura Adams, Alex Brendel, Julia Donne, Adam Folaranmi, Eniola Hopwood, Laura Long, Anna-May Losty, Paul D. Benscoter, Dan de Vos, Corné King, Sebastian Kovesi, Tom Krishnan, Usha Nah, Shireen Anne Ong, Lin Yin Rutter, Mike Teague, Warwick J. Zorn, Aaron M. Hall, Nigel J. Thursfield, Rebecca RD Surgery RJ Pediatrics Introduction Heterogeneity in reported outcomes of infants with oesophageal atresia (OA) with or without tracheo-oesophageal fistula (TOF) prevents effective data pooling. Core outcome sets (COS) have been developed for many conditions to standardise outcome reporting, facilitate meta-analysis and improve the relevance of research for patients and families. Our aim is to develop an internationally-agreed, comprehensive COS for OA-TOF, relevant from birth through to transition and adulthood. Methods and analysis A long list of outcomes will be generated using (1) a systematic review of existing studies on OA-TOF and (2) qualitative research with children (patients), adults (patients) and families involving focus groups, semistructured interviews and self-reported outcome activity packs. A two-phase Delphi survey will then be completed by four key stakeholder groups: (1) patients (paediatric and adult); (2) families; (3) healthcare professionals; and (4) researchers. Phase I will include stakeholders individually rating the importance and relevance of each long-listed outcome using a 9-point Likert scale, with the option to suggest additional outcomes not already included. During phase II, stakeholders will review summarised results from phase I relative to their own initial score and then will be asked to rescore the outcome based on this information. Responses from phase II will be summarised using descriptive statistics and a predefined definition of consensus for inclusion or exclusion of outcomes. Following the Delphi process, stakeholder experts will be invited to review data at a consensus meeting and agree on a COS for OA-TOF. Ethics and dissemination Ethical approval was sought through the Health Research Authority via the Integrated Research Application System, registration no. 297026. However, approval was deemed not to be required, so study sponsorship and oversight were provided by Alder Hey Children’s NHS Foundation Trust. The study has been prospectively registered with the COMET Initiative. The study will be published in an open access forum. © Author(s) (or their employer(s)) 2024. BMJ Publishing Group 2024 Article PeerReviewed Ducey, Jonathan and Lansdale, Nick and Gorst, Sarah and Bray, Lucy and Teunissen, Nadine and Cullis, Paul and Faulkner, Julia and Gray, Victoria and Gammino, Lucia Gutierrez and Slater, Graham and Baird, Laura and Adams, Alex and Brendel, Julia and Donne, Adam and Folaranmi, Eniola and Hopwood, Laura and Long, Anna-May and Losty, Paul D. and Benscoter, Dan and de Vos, Corné and King, Sebastian and Kovesi, Tom and Krishnan, Usha and Nah, Shireen Anne and Ong, Lin Yin and Rutter, Mike and Teague, Warwick J. and Zorn, Aaron M. and Hall, Nigel J. and Thursfield, Rebecca (2024) Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol. BMJ Paediatrics Open, 8 (1). ISSN 2399-9772, DOI https://doi.org/10.1136/bmjpo-2023-002262 <https://doi.org/10.1136/bmjpo-2023-002262>. 10.1136/bmjpo-2023-002262
institution Universiti Malaya
building UM Library
collection Institutional Repository
continent Asia
country Malaysia
content_provider Universiti Malaya
content_source UM Research Repository
url_provider http://eprints.um.edu.my/
topic RD Surgery
RJ Pediatrics
spellingShingle RD Surgery
RJ Pediatrics
Ducey, Jonathan
Lansdale, Nick
Gorst, Sarah
Bray, Lucy
Teunissen, Nadine
Cullis, Paul
Faulkner, Julia
Gray, Victoria
Gammino, Lucia Gutierrez
Slater, Graham
Baird, Laura
Adams, Alex
Brendel, Julia
Donne, Adam
Folaranmi, Eniola
Hopwood, Laura
Long, Anna-May
Losty, Paul D.
Benscoter, Dan
de Vos, Corné
King, Sebastian
Kovesi, Tom
Krishnan, Usha
Nah, Shireen Anne
Ong, Lin Yin
Rutter, Mike
Teague, Warwick J.
Zorn, Aaron M.
Hall, Nigel J.
Thursfield, Rebecca
Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
description Introduction Heterogeneity in reported outcomes of infants with oesophageal atresia (OA) with or without tracheo-oesophageal fistula (TOF) prevents effective data pooling. Core outcome sets (COS) have been developed for many conditions to standardise outcome reporting, facilitate meta-analysis and improve the relevance of research for patients and families. Our aim is to develop an internationally-agreed, comprehensive COS for OA-TOF, relevant from birth through to transition and adulthood. Methods and analysis A long list of outcomes will be generated using (1) a systematic review of existing studies on OA-TOF and (2) qualitative research with children (patients), adults (patients) and families involving focus groups, semistructured interviews and self-reported outcome activity packs. A two-phase Delphi survey will then be completed by four key stakeholder groups: (1) patients (paediatric and adult); (2) families; (3) healthcare professionals; and (4) researchers. Phase I will include stakeholders individually rating the importance and relevance of each long-listed outcome using a 9-point Likert scale, with the option to suggest additional outcomes not already included. During phase II, stakeholders will review summarised results from phase I relative to their own initial score and then will be asked to rescore the outcome based on this information. Responses from phase II will be summarised using descriptive statistics and a predefined definition of consensus for inclusion or exclusion of outcomes. Following the Delphi process, stakeholder experts will be invited to review data at a consensus meeting and agree on a COS for OA-TOF. Ethics and dissemination Ethical approval was sought through the Health Research Authority via the Integrated Research Application System, registration no. 297026. However, approval was deemed not to be required, so study sponsorship and oversight were provided by Alder Hey Children’s NHS Foundation Trust. The study has been prospectively registered with the COMET Initiative. The study will be published in an open access forum. © Author(s) (or their employer(s)) 2024.
format Article
author Ducey, Jonathan
Lansdale, Nick
Gorst, Sarah
Bray, Lucy
Teunissen, Nadine
Cullis, Paul
Faulkner, Julia
Gray, Victoria
Gammino, Lucia Gutierrez
Slater, Graham
Baird, Laura
Adams, Alex
Brendel, Julia
Donne, Adam
Folaranmi, Eniola
Hopwood, Laura
Long, Anna-May
Losty, Paul D.
Benscoter, Dan
de Vos, Corné
King, Sebastian
Kovesi, Tom
Krishnan, Usha
Nah, Shireen Anne
Ong, Lin Yin
Rutter, Mike
Teague, Warwick J.
Zorn, Aaron M.
Hall, Nigel J.
Thursfield, Rebecca
author_facet Ducey, Jonathan
Lansdale, Nick
Gorst, Sarah
Bray, Lucy
Teunissen, Nadine
Cullis, Paul
Faulkner, Julia
Gray, Victoria
Gammino, Lucia Gutierrez
Slater, Graham
Baird, Laura
Adams, Alex
Brendel, Julia
Donne, Adam
Folaranmi, Eniola
Hopwood, Laura
Long, Anna-May
Losty, Paul D.
Benscoter, Dan
de Vos, Corné
King, Sebastian
Kovesi, Tom
Krishnan, Usha
Nah, Shireen Anne
Ong, Lin Yin
Rutter, Mike
Teague, Warwick J.
Zorn, Aaron M.
Hall, Nigel J.
Thursfield, Rebecca
author_sort Ducey, Jonathan
title Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
title_short Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
title_full Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
title_fullStr Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
title_full_unstemmed Developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: OCELOT task group study protocol
title_sort developing a core outcome set for the health outcomes for children and adults with congenital oesophageal atresia and/or tracheo-oesophageal fistula: ocelot task group study protocol
publisher BMJ Publishing Group
publishDate 2024
url http://eprints.um.edu.my/44812/
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