{
  "abstract": "Background Despite anatomical correction, people born with oesophageal atresia±tracheoesophageal fistula (OA-TOF) experience lifelong morbidity. Core outcome sets (COSs) are recognised as a means of improving research quality and, as a consequence, improving patient outcomes; one was not available for this population.Objective The scope of the study was to develop a COS for people born with OA-TOF that would be applicable regardless of age or geographic location.Study design Patient input was paramount to this study. For long-list generation, in addition to the systematic review (SR), patients and representatives were invited to participate in focus groups, interviews or complete activity packs to ascertain outcomes that matter most to them. International consensus was then sought using a two-step Delphi survey followed by an online consensus meeting.Results Eight outcomes were identified through patient events that had not been picked up from SR. 175 people completed the Delphi survey from 26 countries and health care professionals from 13 different disciplines. 24 outcomes met predefined criteria for inclusion and following discussion and voting in the consensus meeting, and 14/24 outcomes were agreed for inclusion in the COS.Conclusion 14 outcomes have been agreed on to form the COS. 12 of these outcomes are relevant to people of all ages, 1 to paediatric population and 1 to adult cohorts. The COS is, therefore, truly applicable lifelong, which was the scope of the project. This COS will help reduce research heterogeneity, enabling better quality research outcomes and more comparable data.",
  "authors": [
    {
      "affiliations": [
        "Respiratory Medicine, Alder Hey Children’s NHS Foundation Trust, Liverpool, UK",
        "Women and Children’s Health, University of Liverpool Faculty of Health and Life Sciences, Liverpool, UK"
      ],
      "name": "Rebecca Thursfield"
    },
    {
      "affiliations": [
        "Department of Health Data Science, University of Liverpool, Liverpool, UK"
      ],
      "name": "Sarah Gorst"
    },
    {
      "affiliations": [
        "Department of Pediatric Surgery, Erasmus MC Universitair Medisch Centrum Rotterdam, Rotterdam, The Netherlands"
      ],
      "name": "Nadine Teunisson"
    },
    {
      "affiliations": [
        "Department of Paediatric and Neonatal Surgery, Royal Manchester Children’s Hospital, Manchester, UK",
        "Developmental Biology and Medicine, School of Medical Sciences, The University of Manchester Faculty of Biology Medicine and Health, Manchester, UK"
      ],
      "name": "Nick Lansdale"
    },
    {
      "affiliations": [
        "Department of Dietetics, Yeovil District Hospital NHS Foundation Trust, Yeovil, UK",
        "TOFS Charity, Nottingham, UK"
      ],
      "name": "Julia Faulkner"
    },
    {
      "affiliations": [
        "Department of Pediatric Gastroenterology, Sydney Children’s Hospital Randwick, Randwick, New South Wales, Australia",
        "School of Women's and Children’s Health, University of New South Wales, Sydney, New South Wales, Australia"
      ],
      "name": "Usha Krishnan"
    },
    {
      "affiliations": [
        "Department of Pediatrics, Division of Respirology, Children’s Hospital of Eastern Ontario, Ottawa, Ontario, Canada",
        "Department of Pediatrics, University of Ottawa, Ottawa, Ontario, Canada"
      ],
      "name": "Tom Kovesi"
    },
    {
      "affiliations": [
        "TOFS Charity, Nottingham, UK"
      ],
      "name": "Graham Slater"
    },
    {
      "affiliations": [
        "Department of Paediatric and Neonatal Surgery, Royal Hospital for Children and Young People, Edinburgh, UK"
      ],
      "name": "Paul Cullis"
    },
    {
      "affiliations": [
        "Evidence-Based Practice Research Centre, Edge Hill University Faculty of Health and Social Care, Ormskirk, UK",
        "Children’s Nursing Research Unit, Alder Hey Children’s NHS Foundation Trust, Liverpool, UK"
      ],
      "name": "Lucy Bray"
    },
    {
      "affiliations": [
        "Department of Otorhinolaryngology, Alder Hey Children’s NHS Foundation Trust, Liverpool, UK"
      ],
      "name": "A Donne"
    },
    {
      "affiliations": [
        "Department of Paediatric Surgery, Melbourne Children’s, Melbourne, Victoria, Australia",
        "Murdoch Children’s Research Institute, Parkville, Victoria, Australia"
      ],
      "name": "Warwick Teague"
    },
    {
      "affiliations": [
        "Department of Paediatric Surgery, Mahidol University Faculty of Medicine Ramathibodi Hospital, Bangkok, Thailand"
      ],
      "name": "Paul D Losty"
    },
    {
      "affiliations": [
        "Department of Paediatric Respiratory Medicine, Royal Brompton and Harefield NHS Foundation Trust, London, UK"
      ],
      "name": "Siobhan Carr"
    },
    {
      "affiliations": [
        "Department of Clinical Psychology, Alder Hey Children’s NHS Foundation Trust, Liverpool, UK"
      ],
      "name": "Victoria Gray"
    },
    {
      "affiliations": [
        "Department of Pediatric Surgery, Sor Maria Ludovia’s Hospital, La Plata, Argentina"
      ],
      "name": "Lucia Gutierrez-Gammino"
    },
    {
      "affiliations": [
        "Department of Surgery, University of Malaya, Kuala Lumpur, Malaysia"
      ],
      "name": "Shireen Anne Nah"
    },
    {
      "affiliations": [
        "Department of Paediatric Surgery, Southampton General Hospital, Southampton, UK"
      ],
      "name": "Nigel J Hall"
    }
  ],
  "title": "Development of a lifelong core outcome set for oesophageal atresia ± tracheoesophageal fistula: the OCELOT study",
  "uid": "3da98f5d-2d71-5ec9-82d1-17d3276da757"
}