Development of a gastroschisis core outcome set

Objective: Outcome reporting heterogeneity impedes identification of gold standard treatments for children born with gastroschisis. Use of core outcome sets (COSs) in research reduces outcome reporting heterogeneity and ensures that studies are relevant to patients. The aim of this study was to develop a gastroschisis COS. Design and setting: Systematic reviews and stakeholder nomination were used to identify candidate outcomes that were subsequently prioritised by key stakeholders in a three-phase online Delphi process and face-to-face consensus meeting using a 9-point Likert scale. In phases two and three of the Delphi process, participants were shown graphical and numerical representations of their own, and all panels scores for each outcome respectively and asked to review their previous score in light of this information. Outcomes were carried forward to the consensus meeting if prioritised by two or three stakeholder panels in the third phase of the Delphi process. The COS was formed from outcomes where =70% of consensus meeting participants scored the outcome 7-9 and <15% of participants scored it 1-3. Results: 71 participants (84%) completed all phases of the Delphi process, during which 87 outcomes were assessed. Eight outcomes, mortality, sepsis, growth, number of operations, severe gastrointestinal complication, time on parenteral nutrition, liver disease and quality of life for the child, met criteria for inclusion in the COS. Conclusions: Eight outcomes have been included in the gastroschisis COS as a result of their importance to key stakeholders. Implementing use of the COS will increase the potential for identification of gold standard treatments for the management of children born with gastroschisis.

Contributors

Benjamin Saul Raywood Allin
Nigel J Hall
Andrew R Ross
Sean S Marven
Jennifer J Kurinczuk
Marian Knight
on behalf of the NETS1G collaboration

Publication

Journal: Archives of Disease in Childhood: Fetal and Neonatal Edition
Volume:
Issue:
Pages: -
Year: 2018
DOI: 10.1136/ archdischild-2017-314560

Further Study Information

Current Stage: Not Applicable
Date: October 2014 - June 2017
Funding source(s): MK is funded by a National Institute for Health Research (NIHR) Professorship. BSRA is funded by an NIHR Doctoral Research Fellowship. NJH is supported by the NIHR through the NIHR Southampton Biomedical Research Centre in nutrition.


Health Area

Disease Category: Child health, Gastroenterology, Neonatal care

Disease Name: Gastroschisis

Target Population

Age Range: 0 - 18

Sex: Either

Nature of Intervention: Any, Surgery

Stakeholders Involved

- Clinical experts
- Consumers (caregivers)
- Families
- Patient/ support group representatives
- Researchers
- Consumers (patients)

Study Type

- COS for clinical trials or clinical research

Method(s)

- Consensus meeting
- Delphi process
- Systematic review

1) Systematic review of the existing literature relating to treatment interventions intended to improve outcome of infants born with gastroschisis. This will identify candidate outcomes for inclusion in the COS.
2) Delphi consensus process using multiple stakeholder groups including clinicians, researchers, parents, people born with gastroschisis, and representatives of support groups.
3) Consensus process amongst multiple stakeholder groups to define each outcome measure and how to measure them
4) Dissemination of COS