NETS1HD: study protocol for development of a core outcome set for use in determining the overall success of Hirschsprung’s disease treatment

Abstract

BackgroundUse of core outcome sets in research has been proposed as a method for countering the problems caused by heterogeneity of outcome measure reporting. Heterogeneity of outcome measure reporting occurs in Hirschsprung’s disease (HD) research and is limiting the development of a robust evidence base to support clinical practice.MethodsCandidate outcome measures have been identified through a systematic review. These outcome measures will form the starting point for a three-phase online Delphi process to be carried out in parallel by three panels of experts. Panel 1 is a neonatal panel; panel 2 is a non-neonatal panel; and panel 3 is a lay panel. In round 1, experts will be asked to score the previously identified outcome measures from 1 to 9 based on how important they think the measures are in determining the overall success of their/their child’s/their patient’s HD. In round 2, experts will be presented with the same list of outcome measures and graphical representations of how their panel scored that outcome in round 1. They will be asked to re-score the outcome measure, taking into account how important other members of their panel felt it to be. In round 3, experts will again be asked to re-score each outcome measure, but this time they will receive a graphical representation of the distribution of scores from all three panels, which they should take into account when re-scoring. Following round 3 of the Delphi process, 40 experts will be invited to attend a face-to-face consensus meeting. Participants will be invited in a purposive manner to obtain balance between the different panels. Results of the Delphi process will be discussed, and outcomes will be re-scored. Outcome measures where >70% of participants at the meeting scored it 7–9 and <15% scored it 1–3 will form the core outcome set.DiscussionDevelopment of a core outcome set will help to reduce heterogeneity of outcome measure reporting in HD. This will increase the quality of research taking place and ultimately improve care provided to infants with HD.Electronic supplementary materialThe online version of this article (doi:10.1186/s13063-016-1693-6) contains supplementary material, which is available to authorized users.

NETS1HDstudy: development of a Hirschsprung’s disease core outcome set

“…The COS was developed according to a prospectively registered protocol, 11 using methodology recommended by the COMET initiative ( figure 1 ).…”

Comprehensive Geriatric Assessment in hospital and hospital-at-home settings: a mixed-methods study

“…Formal written consent was not sought and participation in the panel assumed consent; this is a common approach. 115,116 We sent a link via e-mail to a brief online questionnaire that asked participants to provide details of their experience of the CGA and their role and invited them to register to participate in the Delphi process. The online Delphi questionnaire was hosted on the secure servers of the University of Oxford and developed using LimeSurvey (LimeSurvey GmbH Survey Services & Consulting, Hamburg, Germany).…”

“…The online Delphi questionnaire was hosted on the secure servers of the University of Oxford and developed using LimeSurvey (LimeSurvey GmbH Survey Services & Consulting, Hamburg, Germany). 116…”

“…Participants were asked to score each of the CGA statements on a score from 1 to 9, where 1-3 were 'not important', 4-6 were 'important' and 7-9 were 'really important'. 116 Participants had 4 weeks to complete the exercise (24 August 2017 to 20 September 2017) and received one reminder. Completion of the exercise took, on average, 45 minutes.…”

“…Statements that ≥ 69% of participants in both panels scored as 7-9 (really important) and that < 15% scored as 1-3 (not important) were included in the final version. 116…”