Title: Developing an effective intervention for younger children (5-11 years) with Chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME)
Funder: Department of Health and Social Care

Abstract: Background Paediatric Chronic fatigue syndrome (CFS), or myalgic encephalomyelitis (ME) is relatively common (0.4-2.4%) and can lead to prolonged illness and disability with negative impacts on children, their families and high healthcare costs. CFS/ME is characterised by persistent, disabling fatigue and is associated with a range of symptoms.Paediatric and specialist CFS/ME services see patients below the age of 12, however little is known about how to treat this age group. Disability in the under-12 age group is high, with low levels of school attendance, high levels of fatigue, anxiety, functional disability and pain. Research exploring epidemiology and treatments have focused on children aged 12 and over. Preparatory work for this application suggests younger age predicts worse physical function 12 months after assessment (change in SF-36 physical function subscale is 1.44 points (95% CI 0.03 to 2.86) per chronological year (p=5 to ageAim To develop an intervention for CFS/ME in 5-11 year olds, in line with MRC guidance.Research Questions What are the characteristics of 5-11 year olds with CFS/ME? How are cognitive and/or behavioural interventions delivered to 5-11 year olds? Is there evidence to suggest that these are acceptable to children, parents/carers and clinicians? What are the views of families affected by CFS/ME in 5-11 year olds? What are the views of professionals on how to deliver intervention for CFS/ME in 5-11 year olds? Plan of Investigation Project 1 (Research Question (RQ1): I will use secondary data on children aged 8-11 from one trial and one cohort study. I will deliver a cohort study as part of this PhD to collect data on 5-7 year olds. I will combine these datasets to describe the clinical and demographic characteristics of 5-11years olds, as well as reporting recovery rates at 6 and 12 months and risk factors to poorer recovery. Project 2 (RQ 2): I will carry out a mixed-methods systematic review exploring cognitive and/or behavioural Interventions for 5-11year olds. The mode of delivery for each intervention will be described detailing aspects such as: what was done and what was needed, and by coding interventions using the Behaviour Change Techniques Taxonomy. In addition, I will present mixed-methods data on whether children, parents/carers and clinicians find these interventions acceptable. Project 3(RQ 3): I will carry out qualitative interviews with children and parents/carers. Semi-structured topic guides will explore: the experience of families; perspectives on effective intervention; views on acceptable outcome measures. I will audio record interviews, keep visual images, take observations notes and analyse all of these using thematic analysis. Project 4 (RQ 4): I will synthesise projects 1-3 to develop an intervention for 5-11year with CFS/ME. In the fourth year, I will convene an expert focus group to: review evidence (from studies 1-3), gather the group's perspectives and finalise a logic model of the intervention.Benefits Within 5 years of project completion I will have developed and evaluated an intervention that can be delivered within NHS services. Paediatric CFS/ME Patients and families affected by CFS/ME, clinicians and NHS commissioners will benefit from knowing the epidemiology of CFS/ME in 5-11year olds and effective treatment. Wider Paediatric Services Other clinical conditions may benefit from understanding effective ways to adapt behavioural interventions for 5-11 year olds, including chronic pain services.

Lay abstract: Background Paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is relatively common and disabling; it affects schooling, friendships and family life. Although we know how we should treat teenagers, there are no studies which show us what we should do for younger (primary school age) children. Children with CFS/ME aged 5-11 are usually given the same treatments, but they don't seem to work as well in young children. Aims My research aims to understand more about CFS/ME in primary school children (5-11 year olds) and to develop effective treatment.Plan This research will: Project 1: Describe children with CFS/ME aged 5-11 years. I will use information from two existing studies looking at CFS/ME in 8-11 year olds. I will also carry out a new study recruiting children with CFS/ME aged 5-7 years. Children will be recruited from the Bath specialist CFS/ME service. I will ask children and families to complete questionnaires when they first join the study, then at 6 months and 12months. I will use information from these studies to describe things like: the age and gender of the children; the types of symptoms the children have; how severe their symptoms are; how much it affects their friendships; and how much school they miss. I will investigate how many children get better at 6 months and 12 months and whether there are factors that predict recovery. Project 2: Look at what is already known about health interventions for children aged 5-11 years. I will look at existing research about how treatments are delivered to this age group, and I will summarise this information. I will describe each treatment, giving information such as: what was done and what was needed. I will also present any information which indicates whether children, parents/carers and clinicians liked these treatments. Project 3: Speak to children and families affected by CFS/ME in 5-11 year olds. I will recruit children from the specialist paediatric CFS/ME service in Bath. I will talk to children and their family together and also talk to parents/ carers on their own. I will use activities with the children, such as drawing and card sorting, to make the discussion more fun. I will ask families what they think about the questionnaires we are using in project 1, and will make changes to these based on their opinions. I also want to understand the experience of children and families affected by CFS/ME at this young age, and their views on CFS/ME treatments for younger children. I will use this information to guide the development of the new treatment, and as this develops I will ask families what they think about it. Project 4: Combine the findings from the above three projects to develop a treatment for children with CFS/ME aged 5-11 years of age. I will ask a panel of experts in child health and CFS/ME to help me review all the information and help me develop better treatments. I will also work with a patient group throughout, and I will ask for their opinion as the new treatment is developed. Benefits for the NHS Research into chronic illnesses is childhood is important for the NHS. The NIHR brought attention to this issue in its previous theme long-term conditions in children and young people.We think that this work is important for NHS paediatric CFS/ME services because we don't know how to treat younger children with CFS/ME. This project should improve treatment and therefore outcomes for this age group. If successful, NHS services will deliver better treatments within 5 years of the project completion. Secondly, we think that learning about effective ways to adapt treatments for 5-11 year olds will be helpful for other child health services, for example chronic pain services.Sharing this information Throughout this project, I will work closely with the Association of Young People with ME (AYME) to share the findings with children, young people and families affected by CFS/ME by various means e.g. social media and the AYME website. The research findings will be written up in a way that is easy for families to understandand, and will be shared widely with professionals and patients as the project progresses. In addition, articles will be published in journals that are read by people working with children who have CFS/ME and other health conditions. I will also share findings at conferences.

Research activity codes: 3.1 PRIMARY PREVENTION INTERVENTIONS TO MODIFY BEHAVIOURS OR PROMOTE WELL-BEING
Health categories: DISPUTED AETIOLOGY AND OTHER