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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 J 1 , Lansdale N 2 , Gorst S 3 , Bray L 4 , Teunissen N 5 , Cullis P 6 Show all authors , Faulkner J 7 , Gray V 8 , Gutierrez Gammino L 9 , Slater G 10 , Baird L 11 , Adams A 12 , Brendel J 13 , Donne A 14 , Folaranmi E 15 , Hopwood L 16 , Long AM 17 , Losty PD 18 , Benscoter D 19 , de Vos C 20 , King S 21 , Kovesi T 22 , Krishnan U 23 , Nah SA 24 , Ong LY 25 , Rutter M 19 , Teague WJ 26 , Zorn AM 27 , Hall NJ 28 , Thursfield R 29

Affiliations 

  • 1 Department of Paediatric and Neonatal Surgery, Alder Hey Children's NHS Foundation Trust, Liverpool, Merseyside, UK
  • 2 Department of Paediatric and Neonatal Surgery, Royal Manchester Children's Hospital, Manchester, UK
  • 3 Department of Health Data Science, University of Liverpool, Liverpool, UK
  • 4 Evidence-based Practice Research Centre, Edge Hill University, Ormskirk, UK
  • 5 Department of Pediatric Surgery, Erasmus MC Sophia Children Hospital, Rotterdam, Zuid-Holland, The Netherlands
  • 6 Department of Paediatric and Neonatal Surgery, Royal Hospital for Children and Young People, Edinburgh, UK
  • 7 Department of Dietetics, Yeovil District Hospital NHS Foundation Trust, Yeovil, UK
  • 8 Department of Clinical Psychology, Alder Hey Children's NHS Foundation Trust, Liverpool, Merseyside, UK
  • 9 Department of Pediatric Surgery, Sor Maria Ludovica Hospital, La Plata, Argentina
  • 10 TOFS, Nottingham, UK
  • 11 Department of Speech and Language Therapy, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK
  • 12 Department of Respiratory Medicine, Leeds Teaching Hospitals NHS Trust, Leeds, UK
  • 13 Department of Pediatric Surgery, Hannover Medical School, Hannover, Germany
  • 14 Department of ENT Surgery, Alder Hey Children's NHS Foundation Trust, Liverpool, Merseyside, UK
  • 15 Department of Paediatric, Cardiff and Vale University Health Board, Cardiff, UK
  • 16 Department of Physiotherapy, Alder Hey Children's NHS Foundation Trust, Liverpool, Merseyside, UK
  • 17 Department of Paediatric and Neonatal Surgery, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK
  • 18 Department of Paediatric Surgery, Mahidol University, Salaya, Thailand
  • 19 University of Cincinnati College of Medicine, Cincinnati, Ohio, USA
  • 20 Faculty of Medicine and Health Sciences, Stellenbosch University, Cape Town, South Africa
  • 21 Paediatric Surgery, The Royal Children's Hospital Melbourne, Melbourne, Victoria, Australia
  • 22 Pediatrics, Division of Respirology, Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada
  • 23 Department of Pediatric Gastroenterology, Sydney Children's Hospitals Network, Westmead, New South Wales, Australia
  • 24 Department of Surgery, University of Malaya, Kuala Lumpur, Malaysia
  • 25 Department of Paediatric Surgery, KK Women's and Children's Hospital, Singapore
  • 26 Department of Paediatric Surgery, The Royal Children's Hospital Melbourne, Melbourne, Victoria, Australia
  • 27 Department of Pediatrics, College of Medicine, University of Cincinnati, Cincinnati, Ohio, USA
  • 28 University Surgery Unit, Faculty of Medicine, University of Southampton, Southampton, UK
  • 29 Respiratory Unit, Alder Hey Children's NHS Foundation Trust, Liverpool, UK rebecca.thursfield@alderhey.nhs.uk
BMJ Paediatr Open, 2024 Feb 05;8(1).
PMID: 38316469 DOI: 10.1136/bmjpo-2023-002262

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.

* Title and MeSH Headings from MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine.

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