NETS²ᴴᴰ - Hirschsprung’s Disease

Why are we running this study and what benefits might it have?

This study will help to identify whether the type of operation a baby has to treat their Hirschsprung’s Disease affects their health and wellbeing when they are six to seven years of age.

At present, there are many ways of doing the ‘pull-through’ operation to treat babies who are born with Hirschsprung’s Disease. Each operation is proposed to have its benefits and its drawbacks, but at present, we do not know whether one is best overall. In this study, we would like to start trying to answer that question. Results from this study can then be used by surgeons to help them give more reliable information to future parents of children born with Hirschsprung’s disease.

We are conducting this study, as parents of children with Hirschsprung’s Disease have told us that it is an important question to try and answer. Parent support groups including the Hirschsprung’s and Motility Disorders Support Network, and the CHAMPS appeal have helped us to design the study.

When will I hear from the study team

You will hear from the study team on up to eight occasions:

  • The first contact will be from someone working in the hospital that your child was originally treated in. They will either call you or send you a letter to ask if you are happy for your contact details to be shared with researchers working in the National Perinatal Epidemiology Unit.
  • If you are happy for your contact details to be shared with researchers in the National Perinatal Epidemiology Unit, you will receive a pack from us containing more information about the study and a consent form that you need to complete and send back if you are happy to take part.
  • Within a month of sending the consent form back, you should receive confirmation from the National Perinatal Epidemiology Unit that we have received your consent form. If you have not heard from us in that time, please contact Benjamin.allin@npeu.ox.ac.uk or call 01865 617824
  • Once your child has had their sixth birthday, you will be sent a text message and an email containing a link to the study questionnaire. If you have not received this link within a month of your child’s sixth birthday, please contact Benjamin.allin@npeu.ox.ac.uk or call 01865617824
  • The study team may call you two weeks and four weeks after sending the study packs and the questionnaires to see if you have any questions about the study.
  • If you haven’t heard from us in a while and want to find out what is happening with the study, please get in touch with us.

Which hospitals are taking part in the study?

Hospital Clinical Contacts
Addenbrooke's Hospital Mr Adil Aslam
Edinburgh Royal Hospital for Sick Children Ms Amanda McCabe
Norfolk & Norwich University Hospital Mr Richard England
Queen's Medical Centre Mr Sandeep Motiwale
Royal Aberdeen Children's Hospital Mr Christopher Driver
Southampton General Mr Charles Keys
Bristol Royal Hospital For Children Ms Janet McNally
Royal Belfast Hospital for Sick Children Mr David Marshall
Royal Manchester Children's Hospital Mr Ross Craigie
Birmingham Children's Hospital Mr Ingo Jester
Glasgow Royal Hospital for Sick Children Mr Gregor Walker
John Radcliffe Hospital Professor Paul Johnson
Leicester Royal Infirmary Mr Anthony Owen and Mr Bala Eradi
Leeds General Infirmary Mr Ian Sugarman
The Children's University Hospital Professor Alan Mortell, Mr Brendan O’Connor and Ms Dawn Deacy
Alder Hey Children's Hospital Mr Simon Kenny
King's College Hospital Mr Shailesh Patel
Royal Alexandra Children's Hospital To be confirmed
Evelina Childrens' Hospital Miss Dorothy Kufeji
St George's Hospital To be confirmed
University Hospital of Wales Ms Aruna Abhyankar
Hull Royal Infirmary Ms Sanja Besarovic and Ms Sarah Trufhitt
Great Ormond Street Hospital for Sick Children Mr Stefano Giuliani
Our Lady's Hospital for Sick Children Professor Alan Mortell
Royal London Hospital Mr Stewart Cleeve
Royal Victoria Infirmary Mr Bruce Jaffray
Chelsea and Westminster Hospital Mr Simon Clarke
Sheffield Children's Hospital Mr Govind Murthi

Which children can take part in the study?

In 2006, British surgeons began collecting anonymous information for a new national database called BAPS-CASS (www.npeu.ox.ac.uk/baps-cass).  The aim of this database was to improve the care given to children requiring surgery in early life.  Every hospital in the UK and Ireland that provides surgery for babies contributes to this, which means we have a great opportunity to look at different treatments and outcomes. Each hospital that contributes information is required to keep a list of which children’s data were added to the central database.

There are approximately 300 children who were born with Hirschsprung’s Disease between October 2010 and March 2012, whose anonymous data were contributed to this database, and the parents of these children will be asked if they wish to take part in the study. The Confidentiality Advisory Group and a Research Ethics Committee have reviewed the design of the NETS2HD study, and have given approval for research nurses, clinicians and researchers working from each paediatric surgical centre to contact these parents to ask permission for staff from the National Perinatal Epidemiology Unit in Oxford to discuss the study with them in more detail.

When will the study start and finish?

We hope to start collecting data late summer 2017, and will finish in summer 2018. We aim to publish the results of the study at the end of 2018 or beginning of 2019.

How is the study designed?

There are two parts to the study. In the first part, surgeons will be asked to fill in a clinical questionnaire about every child whose data contributed to the original BAPS-CASS Hirschsprung’s Disease database. In the second part of the study, parents of this same group of children will be asked to fill in one questionnaire. The questionnaire asks about symptoms their child may or may not be having, and about whether they have been to the doctors recently. There are also some general questions about how the child feels in themselves. It will take about twenty minutes to complete the questionnaire.

The questions we ask have been chosen because people with Hirschsprung’s Disease, their parents, and doctors and nurses who treat them, have told us these are the areas that are most important in trying to work out whether treatment of a child’s Hirschsprung’s Disease has been successful or not. There are three areas that are covered by the questionnaire; the child’s health in relation to their Hirschsprung’s Disease, their bowel function, and how the Hirschsprung’s Disease impacts on the child’s everyday life.

We would like to be able to understand whether the first operation a child has for their Hirschsprung’s Disease affects their health and wellbeing at six to seven years of age. In order to do this, it is important that we are able to understand in detail the medical treatments they have had since birth, and we will do this by linking the information collected from parents to information in the central BAPS-CASS database, and information supplied by surgeons. We would also like to keep any information you provide anonymously for a period of forty years, so that we can use it in future studies to better understand how the health and wellbeing for children with Hirschsprung’s Disease changes from the time they start school, through their teenage years, and into adulthood. 

If you would like more information, please download a copy of the full study protocol .

How to enter study data

Data entry for the NETS2HD study will be managed using an electronic system called OpenClinica. If you are a clinician in one of the participating hospitals, you will have instructions on how to use OpenClinica in your site investigator’s pack, and will be able to find more detailed instructions in the example video below. This video was made for the INOSS anaphylaxis in pregnancy study,  but the processes explained are identical to those that will be used in the NETS2HD study. 

If you are a parent, then you will have the option of completing a paper version of the questionnaire, or entering your answers straight into OpenClinica using either your mobile phone or an internet browser. You will have details on how to do this in your study information pack, and if you have any questions, you can call 01865 289700 and ask to speak to Dr Benjamin Allin who will be able to help you.

Direct link to MP4

Where can I find the results of the study?

The results of the study will be published in a journal and on our website (www.npeu.ox.ac.uk/nets). A report will also be distributed by various parent support groups including the Hirschsprung’s and Motility Disorders Support Network and the CHAMPS appeal. If you take part and give us your contact details we will also send you the results of the study. There will be no information in anything we write that could be traced to any individual parent or children.

What should I do if I have further questions or concerns?

If you would like more information, we would be delighted to talk to you about the study. Please call us on 01865 289714 during normal office hours and ask to speak to Beth Lawson, Benjamin Allin, or Marian Knight, or email Benjamin.allin@npeu.ox.ac.uk. You may also wish to contact one of the Hirschsprung’s Disease support groups who have helped with this study, including the Hirschsprung’s and Motility Disorders Support Network and the CHAMPS appeal.

Who is organising and funding the study?

The study is organised by researchers at the National Perinatal Epidemiology Unit, University of Oxford, jointly with the British Association of Paediatric Surgeons. It will be led by Dr Benjamin Allin and Professor Marian Knight. The National Institute for Health Research (NIHR) has funded the study. Dr Benjamin Allin is undertaking a PhD at the University of Oxford, and the NETS2HD study will contribute to his PhD.

More information about the core study team can be found below:

Study Materials

Investigator pack

Hospital poster

Short form participant information leaflet

Electronic appendix 1.0 record of eligible infants

Electronic appendix 1.0b

Alternative first contact letter

Excel sheet for transfer of contact details