Core Outcome Set for Hirschsprung’s Disease – The NETS1HD Study

By Benjamin Allin

Edited by Ewelina Rogozińska

Hirschsprung’s disease affects approximately 300-350 children in the UK each year and is caused by the failure of development of the nerves within the intestines. Affected children require an operation to remove part of the bowel early in infancy, and there are several different ways of performing this operation. At the moment, when comparing treatments, researchers usually report on short-term markers of success such as operative time, as opposed to longer term, patient-centered outcomes such as fecal incontinence. There is also a great variation in the outcomes reported by individual studies (Allin et al. 2016). Both of these factors have made it impossible to reliably identify which operation has the best outcomes.

Developing and implementing a core outcome set (COS) will increase the ability of researchers to identify whether there are differences in outcome between the operations, thereby allowing development of evidence-based management guidelines. Ultimately this will help to improve the care offered to children with the condition and lead to better health outcomes.

The aim of the NETS1HD study was to develop a core outcome set that would address these issues, and increase the likelihood of researchers being able to identify a gold standard treatment for children with Hirschsprung’s disease.

This is the first pediatric surgical core outcome set that has been developed, and as a result, the biggest challenge we faced was in gaining initial ‘buy in’ from the clinical community. Engaging with groups such as the British Association of Paediatric Surgeons and the UK Paediatric Colorectal Group helped to overcome initial skepticism, and we hope that upcoming presentations of the study, along with its planned used in a UK-wide cohort study, will help to demonstrate the utility of core outcome sets to the wider clinical community.  

The key to achieving a useful COS is ensuring that participants fully understand the factors that should be considered when deciding how important each outcome is. Throughout the process, we have honed the language that we use to explain the different concepts that people should consider, particularly when balancing frequency of an outcome against the severity of the outcome. Throughout the NETS1HD study we used written material to explain the scoring process to participants, but after undertaking a review of the process have decided that it would be beneficial to present this material using video clips embedded within the Delphi process.

The NETS1HD core outcome set is a completed project and its results will be published in due course. 



Mr Benjamin Allin is a National Institute for Health Research Doctoral Research Fellow and Paediatric Surgical Registrar based at National Perinatal Epidemiology Unit, Nuffield Department of Population Health and Nuffield Department of Surgical Sciences, University of Oxford

Twitter: @NPEU_NETS


Posted in Core Outcome Set, Researcher Perspective