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
Issued Date
2024-02-05
Resource Type
eISSN
23999772
Scopus ID
2-s2.0-85184814994
Pubmed ID
38316469
Journal Title
BMJ Paediatrics Open
Volume
8
Issue
1
Rights Holder(s)
SCOPUS
Bibliographic Citation
BMJ Paediatrics Open Vol.8 No.1 (2024)
Suggested Citation
Ducey J., Lansdale N., Gorst S., Bray L., Teunissen N., Cullis P., Faulkner J., Gray V., Gammino L.G., Slater G., Baird L., Adams A., Brendel J., Donne A., Folaranmi E., Hopwood L., Long A.M., Losty P.D., Benscoter D., de Vos C., King S., Kovesi T., Krishnan U., Nah S.A., Ong L.Y., Rutter M., Teague W.J., Zorn A.M., Hall N.J., Thursfield R. 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 Vol.8 No.1 (2024). doi:10.1136/bmjpo-2023-002262 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/97245
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
Author(s)
Ducey J.
Lansdale N.
Gorst S.
Bray L.
Teunissen N.
Cullis P.
Faulkner J.
Gray V.
Gammino L.G.
Slater G.
Baird L.
Adams A.
Brendel J.
Donne A.
Folaranmi E.
Hopwood L.
Long A.M.
Losty P.D.
Benscoter D.
de Vos C.
King S.
Kovesi T.
Krishnan U.
Nah S.A.
Ong L.Y.
Rutter M.
Teague W.J.
Zorn A.M.
Hall N.J.
Thursfield R.
Lansdale N.
Gorst S.
Bray L.
Teunissen N.
Cullis P.
Faulkner J.
Gray V.
Gammino L.G.
Slater G.
Baird L.
Adams A.
Brendel J.
Donne A.
Folaranmi E.
Hopwood L.
Long A.M.
Losty P.D.
Benscoter D.
de Vos C.
King S.
Kovesi T.
Krishnan U.
Nah S.A.
Ong L.Y.
Rutter M.
Teague W.J.
Zorn A.M.
Hall N.J.
Thursfield R.
Author's Affiliation
Faculty of Biology, Medicine and Health
Yeovil District Hospital NHS Foundation Trust
Cardiff & Vale University Health Board
Universiti Malaya
University of Ottawa
Royal Children's Hospital, Melbourne
University of Southampton, Faculty of Medicine
Alder Hey Children's NHS Foundation Trust
Hannover Medical School
Royal Manchester Children's Hospital
Edge Hill University
Hospital de Ninos Sor Maria Ludovica
University of Liverpool
Children's Hospital of Eastern Ontario, Ottawa
University of Cincinnati College of Medicine
Cambridge University Hospitals NHS Foundation Trust
KK Women's And Children's Hospital
Erasmus MC Sophia Children’s Hospital
Mahidol University
Leeds Teaching Hospitals NHS Trust
Murdoch Children's Research Institute
NHS Lothian
Stellenbosch University, Faculty of Medicine and Health Sciences
MRC/NIHR Trials Methodology Research Partnership
Sydney Children's Hospitals Network
TOFS
Yeovil District Hospital NHS Foundation Trust
Cardiff & Vale University Health Board
Universiti Malaya
University of Ottawa
Royal Children's Hospital, Melbourne
University of Southampton, Faculty of Medicine
Alder Hey Children's NHS Foundation Trust
Hannover Medical School
Royal Manchester Children's Hospital
Edge Hill University
Hospital de Ninos Sor Maria Ludovica
University of Liverpool
Children's Hospital of Eastern Ontario, Ottawa
University of Cincinnati College of Medicine
Cambridge University Hospitals NHS Foundation Trust
KK Women's And Children's Hospital
Erasmus MC Sophia Children’s Hospital
Mahidol University
Leeds Teaching Hospitals NHS Trust
Murdoch Children's Research Institute
NHS Lothian
Stellenbosch University, Faculty of Medicine and Health Sciences
MRC/NIHR Trials Methodology Research Partnership
Sydney Children's Hospitals Network
TOFS
Corresponding Author(s)
Other Contributor(s)
Abstract
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.