Core Outcome Measures in Effectiveness Trials

NETS1HD study: development of a Hirschsprung's disease core outcome set

General Information

Objective The objective of this study was to develop a Hirschsprung's disease (HD) core outcome set (COS).

Methods Candidate outcomes were identified from a systematic review and stakeholder nomination. A three-phase Delphi process and consensus meeting were used to prioritise candidate outcomes based on scores assigned by stakeholder participants using a nine-point scale. In phases two and three, participants were shown graphical representations of their panel's scores and all panels’ scores respectively for each outcome from the previous phase. After the third phase, outcomes prioritised by two or three panels were taken forward to the consensus meeting. The COS was formed from the 10 highest scoring outcomes meeting the threshold for inclusion (=70% 7–9 and <15% 1–3).

Results Eighty-nine stakeholders (82%) completed all three phases of the Delphi process. Seventy-four outcomes were assessed in phase one of the Delphi process, the following 10 of which met criteria for inclusion in the COS: (1) death with cause specified, (2) long-term faecal incontinence, (3) long-term voluntary bowel movements without need for enemas, or rectal or colonic irrigation, (4) long-term psychological stress for the individual with Hirschsprung's disease, (5) long-term urinary incontinence, (6) objective score of quality of life, (7) objective score of bowel function, (8) unplanned reoperation, (9) >need for a permanent stoma, (10) enterocolitis.

Conclusions This HD COS is formed of 10 outcomes deemed important by key stakeholders. Use of this COS in research will reduce outcome reporting heterogeneity and increase our ability to identify gold standard treatments for HD.

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The objective of this study was to develop a Hirschsprung's disease (HD) core outcome set (COS).

PI – Benjamin Allin, NIHR funded Doctoral Research Fellow, National Perinatal Epidemiology Unit, and Paediatric Surgical Registrar, John Radcliffe Hospital, Oxford.

Marian Knight, NIHR Professor of Maternal and Child Population Health, Honorary Consultant in Public Health, National Perinatal Epidemiology Unit, Nuffield Department of Population Health


BMJ Archives of Disease in Childhood
Further Study Information

August 2015 - July 2017
Funding source(s):
National Institute for Health Research

Health Area

Disease Category

Disease Name
Hirschsprung's disease

Target Population

Age Range
0 - 100


Nature / type of Intervention


Consensus meeting
Delphi process
Systematic review

1. Candidate outcome measures for inclusion in the Delphi process will be identified through a systematic review of the existing literature, which has already been completed.
2. Participants with either expertise in the management of Hirschsprung’s Disease, or lived experience of Hirschsprung’s Disease will be recruited from parents/patients, as well as the full paediatric multi-disciplinary team in order to complete an online three-phase Delphi process. Best practice as established by the COMET initiative will be followed throughout the process.
3. The identified core outcome set will be discussed and ratified at a face-face consensus meeting following completion of the online Delphi process.
4. Results will be disseminated through relevant publications and presentations.

Stakeholders Involved

Clinical experts
Consumers (caregivers)
Consumers (patients)
Service commissioners

Study Type

COS for clinical trials or clinical research
COS for practice

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