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NETS1HD: Development of a Hirschsprung’s Disease Core Outcome Set

Abstract:

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.

Publication status:
Published
Peer review status:
Peer reviewed

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Publisher copy:
10.1136/archdischild-2017-312901

Authors

More by this author
Institution:
University of Oxford
Division:
MSD
Department:
Nuffield Department of Population Health
Sub department:
NPEU
Role:
Author
More by this author
Institution:
University of Oxford
Division:
MSD
Department:
Nuffield Department of Population Health
Sub department:
NPEU
Role:
Author



Publisher:
BMJ Publishing Group
Journal:
Archives of Disease in Childhood More from this journal
Volume:
102
Issue:
12
Pages:
1-10
Publication date:
2017-08-07
Acceptance date:
2017-06-01
DOI:
EISSN:
1468-2044
ISSN:
0003-9888


Pubs id:
pubs:698480
UUID:
uuid:b9f1ec43-1948-4d3f-a669-5de629fd337b
Local pid:
pubs:698480
Source identifiers:
698480
Deposit date:
2017-06-05
ARK identifier:

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