Graded exercise therapy compared to activity management for paediatric chronic fatigue syndrome/myalgic encephalomyelitis: pragmatic randomized controlled trial.

The MAGENTA pragmatic parallel groups randomized controlled trial compared graded exercise therapy (GET) with activity management (AM) in treating paediatric myalgic encephalomyelitis or chronic fatigue syndrome (ME/CFS). Children aged 8-17 years with mild/moderate ME/CFS and presenting to NHS specialist paediatric services were allocated at random to either individualised flexible treatment focussing on physical activity (GET, 123 participants) or on managing cognitive, school and social activity (AM, 118 participants) delivered by NHS therapists. The primary outcome was the self-reported short-form 36 physical function subscale (SF-36-PFS) after 6 months, with higher scores indicating better functioning. After 6 months, data were available for 201 (83%) participants who received a mean of 3.9 (GET) or 4.6 (AM) treatment sessions. Comparing participants with measured outcomes in their allocated groups, the mean SF-36-PFS score changed from 54.8 (standard deviation 23.7) to 55.7 (23.3) for GET and from 55.5 (23.1) to 57.7 (26.0) for AM giving an adjusted difference in means of -2.02 (95% confidence interval -7.75, 2.70). One hundred thirty-five participants completed the mean SF-36-PFS at 12 months, and whilst further improvement was observed, the difference between the study groups remained consistent with chance. The two study groups showed similar changes on most of the secondary outcome measures: Chalder Fatigue, Hospital Anxiety and Depression Scale: Depression, proportion of full-time school attended, a visual analogue pain scale, participant-rated change and accelerometer measured physical activity, whether at the 6-month or 12-month assessment. There was an isolated finding of some evidence of an improvement in anxiety in those allocated to GET, as measured by the Hospital Anxiety and Depression Scale at 6 months, with the 12-month assessment, and the Spence Children's Anxiety scale being aligned with that finding. There was weak evidence of a greater risk of deterioration with GET (27%) than with AM (17%; p = 0.069). At conventional UK cost per QALY thresholds, the probability that GET is more cost-effective than AM ranged from 18 to 21%. Whilst completion of the SF-36-PFS, Chalder Fatigue Scale and EQ-5D-Y was good at the 6-month assessment point, it was less satisfactory for other measures, and for all measures at the 12-month assessment.  Conclusion: There was no evidence that GET was more effective or cost-effective than AM in this setting, with very limited improvement in either study group evident by the 6-month or 12-month assessment points.  Trial registration: The study protocol was registered at www.isrctn.com (3rd September 2015; ISRCTN 23962803) before the start of enrolment to the initial feasibility phase.

Management of severe ME/CFS in children and young people in the UK: a British Paediatric Surveillance Unit study.

OBJECTIVE: Severe myalgic encephalomyelitis or chronic fatigue syndrome (ME/CFS) in children and young people (CYP) is a little-understood condition which significantly impacts education, development and quality of life. We used data from a population-wide surveillance study to explore the screening investigation, referral and management of suspected cases of paediatric severe ME/CFS. METHODS: A British Paediatric Surveillance Unit (BPSU) study reported cases of CYP with suspected severe ME/CFS between February 2018 and February 2019. Paediatricians reporting cases to BPSU and allied healthcare professionals in two large specialist paediatric ME/CFS centres were invited to complete questionnaires for CYP meeting the surveillance case definition. The study focused primarily on CYP with confirmed severe ME/CFS and the extent to which their care met NICE (The National Institute for Health and Care Excellence) recommendations but also considered separately those with probable or possible severe ME/CFS. RESULTS: This study includes a total of 92 CYP with suspected severe ME/CFS; 33 meeting criteria for severe ME/CFS and an additional 59 classified as probable or possible severe ME/CFS. For 16 possible cases, incomplete investigation to exclude alternative diagnoses prevented confirmation of a severe ME/CFS diagnosis. Only 21 of 33 (64%) confirmed severe ME/CFS cases had been referred to specialist services. The management provided varied considerably between patients and four received nothing at all. Of the management provided, the most frequent approaches were medication (67%), activity management (61%) and physiotherapy (61%). Domiciliary assessments and support, and social services referrals were received by 12% and 6% of confirmed severe cases. Similar proportions of management approaches were seen in probable/possible severe ME/CFS. CONCLUSION: Full investigation is frequently incomplete in CYP with suspected severe ME/CFS and recommendations for referral and management are poorly implemented, in particular the needs of CYP who are unable to leave their home might be poorly met.

"not one size fits all" The challenges of measuring paediatric health-related quality of life and the potential role of digital ecological momentary assessment: a qualitative study.

PURPOSE: To explore the views of clinicians and researchers about the challenges of measuring health-related quality of life (HRQoL) in children (5-11 years) and to explore whether digital ecological momentary assessment (EMA) could enhance HRQoL measurement. METHODS: Semi-structured qualitative interviews with 18 professionals (10 academics/researchers, four clinicians, four with both professional backgrounds) experienced in child HRQoL measurement. We analysed data thematically. RESULTS: Theme One describes the uncertainty around conceptualising HRQoL for children and which domains to include; the greater immediacy and sensitivity of children's reflections on their HRQoL, leading to high variability of the construct; and the wide individual differences across childhood, incongruent with fixed HRQoL measures. Theme Two describes the challenges of proxy reporting, questioning whether proxies can meaningfully report a child's HRQoL and reflecting on discrepancies between child and proxy reporting. Theme Three covers the challenge of interpreting change in HRQoL over time; does a change in HRQoL reflect a change in health, or does this reflect developmental changes in how children report HRQoL. Theme Four discusses digital EMA for HRQoL data capture. In-the-moment, repeated measurement could provide rich data and address challenges of recall, ecological validity and variability; passive data could provide objective markers to supplement subjective responses; and technology could enable personalisation and child-centred design. However, participants also raised methodological, practical and ethical challenges of digital approaches. CONCLUSION: Digital EMA may address some of the challenges of HRQoL data collection with children. We conclude by discussing potential future research to explore and develop this approach.

Investigating the factors associated with meaningful improvement on the SF-36-PFS and exploring the appropriateness of this measure for young people with ME/CFS accessing an NHS specialist service: a prospective cohort study.

OBJECTIVES: Paediatric myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is relatively common and disabling, but little is known about the factors associated with outcome. We aimed to describe the number and characteristics of young people reaching the 10-point minimal clinically important difference (MCID) of SF-36-Physical Function Subscale (SF-36-PFS) and to investigate factors associated with reaching the MCID. DESIGN: Prospective observational cohort study. SETTING: A specialist UK National Health Service ME/CFS service, Southwest England; recruitment between March 2014 and August 2015. PARTICIPANTS: 193 eligible patients with ME/CFS aged 8-17 years reported baseline data. 124 (65%) and 121 (63%) with outcome data at 6 and 12 months, respectively. OUTCOME MEASURES: SF-36-PFS (primary outcome). Chalder Fatigue Questionnaire, school attendance, visual analogue pain scale, Hospital Anxiety and Depression Scale, Spence Young People Anxiety Scale, Clinical Global Impression scale and EQ-5D-Y (secondary). RESULTS: At 6 months 48/120 (40%) had reached the MCID for SF-36-PFS. This had increased to 63/117 (54%) at 12 months. On the Clinical Global Impressions, 77% and 79% reported feeling either a little better, much better or very much better. Those with worse SF-36-PFS at baseline assessment were more likely to achieve the MCID for SF-36-PFS at 6 months (odds ratio 0.97, 95% confidence interval 0.96 to 0.99, p value 0.003), but there was weaker evidence of effect at 12 months (OR 0.98, 95% CI 0.97 to 1.00, p value 0.038). No other factors at baseline were associated with the odds of reaching the MCID at 6 months. However, at 12 months, there was strong evidence of an effect of pain on MCID (OR 0.97, 95% CI 0.95 to 0.99, p value 0.001) and SF-36-PFS on MCID (OR 0.96, 95% CI 0.94 to 0.98, p value 0.001). CONCLUSIONS: 40% and 54% of young people reached the MCID at 6 and 12 months, respectively. No factors at assessment (other than SF-36-PFS at 6 months, and pain and SF-36-PFS at 12 months) are associated with MCID of SF-36-PFS at either 6 or 12 months. Further work is needed to explore the most appropriate outcome measure for capturing clinical meaningful improvement for young people with ME/CFS.

Views and Needs of Students, Parents, and Teachers on Closed-Circuit Television, Proximity Trackers, and Access Cards to Facilitate COVID-19 Contact Tracing in Schools: Thematic Analysis of Focus Groups and Interviews.

BACKGROUND: Contact tracing is considered a key measure in preventing the spread of infectious diseases. Governments around the world adopted contact tracing to limit the spread of COVID-19 in schools. Contact tracing tools utilizing digital technology (eg, GPS chips, Bluetooth radios) can increase efficiency compared to manual methods. However, these technologies can introduce certain privacy challenges in relation to retention, tracking, and the using and sharing of personal data, and little is known about their applicability in schools. OBJECTIVE: This is the second of two studies exploring the potential of digital tools and systems to help schools deal with the practical challenges of preventing and coping with an outbreak of COVID-19. The aim was to explore the views, needs, and concerns among secondary school stakeholders (parents, teachers, pupils) regarding the implementation of three digital tools for contact tracing: access cards, proximity tracking, and closed-circuit television (CCTV). METHODS: Focus groups and interviews were conducted with secondary school students, parents, and teachers. The topic guide was informed by the Unified Theory of Technology and Acceptance. Data-driven and theory-driven approaches were combined to identify themes and subthemes. RESULTS: We recruited 22 participants. Findings showed that there is no single solution that is suitable for all schools, with each technology option having advantages and limitations. Existing school infrastructure (eg, CCTV and smart/access cards technology) and the geography of each school would determine which tools would be optimal for a particular school. Concerns regarding the cost of installing and maintaining equipment were prominent among all groups. Parents and teachers worried about how the application of these solutions will affect students' right to privacy. Parents also appeared not to have adequate knowledge of the surveillance technologies already available in schools (eg, CCTV). Students, who were mostly aware of the presence of surveillance technologies, were less concerned about any potential threats to their privacy, while they wanted reassurances that any solutions would be used for their intended purposes. CONCLUSIONS: Findings revealed that there is not one tool that would be suitable for every school and the context will determine which tool would be appropriate. This study highlights important ethical issues such as privacy concerns, balancing invasions of privacy against potential benefits, transparency of communication around surveillance technology and data use, and processes of consent. These issues need to be carefully considered when implementing contact tracing technologies in school settings. Communication, transparency, and consent within the school community could lead to acceptance and engagement with the new tools.

Severe myalgic encephalomyelitis/chronic fatigue syndrome in children and young people: a British Paediatric Surveillance Unit study.

OBJECTIVES: Primary objective: to determine the point prevalence and incidence rate of severe myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) in children aged 5-16 years over 13 months. SECONDARY OBJECTIVES: to describe the demographic features, symptoms, impact on activities of daily living, school attendance and time to diagnosis. DESIGN: Prospective surveillance study conducted by the British Paediatric Surveillance Unit. Paediatricians was asked if they had assessed a child with severe ME/CFS (screening definition for prevalence and incidence: children (5-16 years) diagnosed with ME/CFS so severe that they are unable to attend school for more than 1 hour a week during the last 6 weeks of the school term). PARTICIPANTS: Patients 5-16 years of age, seen by paediatricians and two large ME/CFS specialist services across the UK and Ireland. OUTCOME MEASURES: Paediatrician-completed questionnaires describing demographics, symptoms, function and treatment, (applying National Institute for Health and Care Excellence (NICE)-recommended criteria to assess severity of ME/CFS). Diagnosis of severe, probable severe or possible severe ME/CFS was made only with evidence of NICE-recommended screening blood tests. RESULTS: 285 cases were reported, of which of which 33 were severe, 4 probable severe and 55 possible severe. Estimated prevalence was 3.2 per million children (95% CI 2.2 to 4.5). Including possible/probable severe ME/CFS gave 8.9 per million children (95% CI 7.2 to 11). The incidence rate was 0.90 per million children-years (95% CI 0.43 to 1.65) (1.97 per million children-years (95% CI 1.24 to 2.99)). Median age was 13 years and 58% of cases were female. Median time to diagnosis was 0.47 years. CONCLUSIONS: Although the incidence of children presenting with severe ME/CFS is low, all were very disabled. In addition, the majority receive little or no education. Paediatricians need to consider how to provide rehabilitation and education for these disabled young people.

Maternal vaccines during the Covid-19 pandemic:A qualitative interview study with UK pregnant women.

BACKGROUND: There is suboptimal uptake of recommended maternal vaccines (pertussis and influenza) during pregnancy in the UK. The Covid-19 pandemic has impacted healthcare services, and potentially vaccine coverage, and brought the need for new vaccines to be tested and rolled out. OBJECTIVES: To explore: i) the impact of the Covid-19 pandemic on pregnant women's access to, and attitudes towards, routine maternal vaccines and; ii) women's attitudes towards testing Covid-19 vaccines on pregnant women and their personal willingness to take part in such a trial. DESIGN: Qualitative interview study with pregnant women in the Bristol and surrounding area (UK). METHODS: Semi-structured telephone/videoconference interviews were conducted (following a topic guide), transcribed verbatim and subjected to thematic analysis. RESULTS: Thirty-one pregnant women (selected for demographic range) were interviewed in April/May 2020. Participants felt the pandemic had elevated the importance of routine maternal vaccines, though women were concerned about safety management around appointment attendance. Women were wary of receiving a new Covid-19 vaccine, with most perceiving it as riskier than Covid-19 itself. CONCLUSIONS: It is important to maximise the safety and efficiency of maternity appointments to encourage uptake of routine maternal vaccines, and to communicate this well. For pregnant women to gain a new vaccine or participate in a vaccine trial, they need to be convinced that the risk posed by the virus is greater than any risk of receiving a new vaccine.

Pregnant women's experiences of social distancing behavioural guidelines during the Covid-19 pandemic 'lockdown' in the UK, a qualitative interview study.

BACKGROUND: Covid-19 triggered the rapid roll-out of mass social distancing behavioural measures for infection control. Pregnant women were categorised as 'at risk' requiring extra vigilance with behavioural guidelines. Their understanding and ability to adhere to recommendations was unknown. OBJECTIVES: To complete a behavioural analysis of the determinants of recommended social distancing behaviour in pregnant women, according to the 'capability, opportunity, motivation and behaviour' ('COM-B') model to inform the development of recommendations/materials to support pregnant women in understanding and adhering to behavioural guidelines. DESIGN: Qualitative interview study with pregnant women in the Bristol area (UK). METHODS: Semi-structured telephone/videoconference interviews were conducted following a topic guide informed by the COM-B model, transcribed verbatim and subjected to framework analysis. Infographic materials were iteratively produced with stakeholder consultation, to support pregnant women. RESULTS: Thirty-one women participated (selected for demographic range). Women reported adhering to social distancing recommendations and intended to continue. COM-B analysis identified gaps in understanding around risk, vulnerability, and the extent of required social distancing, as well as facilitators of social distancing behaviour (e.g. social support, motivation to stay safe, home environment/resources). Additional themes around detrimental mental health effects and changes to maternity healthcare from the social distancing measures were identified. Infographic resources (plus midwife report) addressing women's key concerns were produced and disseminated. CONCLUSIONS: The COM-B model provided useful details of determinants of pregnant women's adherence to social distancing behaviours. The confusion of what being 'at risk' meant and varying interpretation of what was expected indicates a need for greater clarity around categories and guidance. The loss of maternity care and negative mental health effects of social distancing suggest a growing area of unmet health needs to be addressed in future.

Recovery from chronic fatigue syndrome: a systematic review-heterogeneity of definition limits study comparison.

BACKGROUND: Paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is a common illness with a major impact on quality of life. Recovery is poorly understood. Our aim was to describe definitions of recovery in paediatric CFS/ME, the rate of recovery and the time to recovery. METHODS: This systematic review included a detailed search of MEDLINE, EMBASE, PsycInfo and Cochrane Library between 1994 and July 2018. Inclusion criteria were (1) clinical trials and observational studies, (2) participants aged <19 years with CFS/ME, (3) conducted in Western Healthcare systems and (4) studies including a measure of recovery and time taken to recover. RESULTS: Twelve papers (10 studies) were identified, involving 826 patients (range 23-135). Recovery rates were highly varied, ranging between 4.5% and 83%.Eleven distinct definitions of recovery were used; six were composite outcomes while five used unidimensional outcomes. Outcome measures used to define recovery were highly heterogeneous. School attendance (n=8), fatigue (n=6) and physical functioning (n=4) were the most common outcomes included in definition of recovery. Only five definitions included a personal measure of recovery. IMPLICATIONS: Definitions of recovery are highly variable, likely secondary to differences in study design, outcomes used, follow-up and study populations. Heterogeneous definitions of recovery limit meaningful comparison between studies, highlighting the need for a consensus definition going forward. Recovery is probably best defined from the child's own perspective with a single self-reported measure. If composite measures are used for research, there should be agreement on the core outcome set used.

"it's a medical condition you need to support as much as possible": a qualitative analysis of teachers' experiences of chronic fatigue syndrome / myalgic encephalomyelitis (CFS/ME).

BACKGROUND: An increasing number of children with complex health needs are being educated in mainstream classes. CFS/ME is a complex and disabling condition, and there is little guidance on how primary school teachers can support younger children with this condition. To improve care, it is important to understand what these children need in the school setting, and the barriers and facilitators to teachers providing this support. The aims for this qualitative study were to explore teachers' views about CFS/ME, their experiences of supporting a pupil with CFS/ME and their perspectives on the barriers and facilitators to providing support. METHODS: We recruited families from an NHS specialist paediatric CFS/ME service and families were eligible if the child was aged between 5 and 11 years and had a diagnosis of CFS/ME. We gained written consent/assent from families to invite the child's teacher to participate in a qualitative interview. We contacted these teachers, gained written consent and then carried out semi-structured qualitative interviews. Interviews were audio-recorded, transcribed, anonymised and analysed thematically. Interviews took place between July 2018 and December 2018. RESULTS: We interviewed 11 teachers; their pupil's age ranged from 5 to 11 years and school attendance ranged from 0 to 80%. Theme 1: Most teachers provided rich descriptions of their pupil's CFS/ME; they consistently described cognitive dysfunction and significant fatigue, but beyond this the symptoms varied from one account to the next (from mobility problems, to aches and pains, digestive problems, headaches, nausea and hypersensitivity). These teachers noted the ripple effects on their pupil's social, emotional and academic functioning. Two of the eleven teachers said that they did not observe symptoms of CFS/ME, expressing a degree of scepticism about the diagnosis. Theme 2: Teachers described a close relationship with their pupil. They said they understood the individual needs of the child and portrayed positive and proactive attitudes towards providing support. The type of support provided included facilitating rest breaks and limiting strenuous activities; using practical strategies to address cognitive, physical, social and emotional difficulties; maintaining a connection with the child during their absences from school; and encouraging the child to talk about their health and wellbeing. Teachers noted that receiving formal confirmation of the child's diagnosis enabled them to put this support in place. Theme 3: The adaptations they described were often intuitive, rather than being based on a knowledge of CFS/ME. Teachers wanted more resources to increase their understanding of the condition and its management. CONCLUSIONS: Primary school teachers want to provide effective support for children with CFS/ME. Clinical services should consider working in collaboration with teachers to equip them with evidence-based strategies for CFS/ME management in the primary school setting.

Chronic fatigue syndrome/myalgic encephalomyelitis in children aged 5 to 11 years: A qualitative study.

Treatments for paediatric chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) have not been designed or evaluated for younger children (5-11-years). The development of a complex intervention for this population requires an in-depth understanding of the perspectives and psychosocial context of children and families. Children with CFS/ME (5-11-years) and their families were recruited from a specialist CFS/ME service, and interviewed using semi-structured topic guides. Data were analysed thematically. Twenty-two participants were interviewed; eight parents, two children (aged nine and ten) and six parent-child dyads (aged 5-11-years). Theme 1: CFS/ME in younger children is complex and disabling. Theme 2: Children aged eight and over (in comparison to those under eight) were more able to describe their illness, engage in clinical consultation, understand diagnosis and self-manage. Theme 3: Parents of children under eight took full responsibility for their child's treatment. As children got older, this increasingly became a joint effort between the parent and child. Parents felt unsupported in their caring role. Clinicians should consider different treatment approaches for children under eight, focusing on: parent-only clinical sessions, training parents to deliver treatment, and increasing support for parents. Children over eight may benefit from tools to help them understand diagnosis, treatment and aids for self-management.

Digital Behavior Change Interventions for Younger Children With Chronic Health Conditions: Systematic Review.

BACKGROUND: The prevalence of chronic health conditions in childhood is increasing, and behavioral interventions can support the management of these conditions. Compared with face-to-face treatment, the use of digital interventions may be more cost-effective, appealing, and accessible, but there has been inadequate attention to their use with younger populations (children aged 5-12 years). OBJECTIVE: This systematic review aims to (1) identify effective digital interventions, (2) report the characteristics of promising interventions, and (3) describe the user's experience of the digital intervention. METHODS: A total of 4 databases were searched (Excerpta Medica Database [EMBASE], PsycINFO, Medical Literature Analysis and Retrieval System Online [MEDLINE], and the Cochrane Library) between January 2014 and January 2019. The inclusion criteria for studies were as follows: (1) children aged between 5 and 12 years, (2) interventions for behavior change, (3) randomized controlled trials, (4) digital interventions, and (5) chronic health conditions. Two researchers independently double reviewed papers to assess eligibility, extract data, and assess quality. RESULTS: Searches run in the databases identified 2643 papers. We identified 17 eligible interventions. The most promising interventions (having a beneficial effect and low risk of bias) were 3 targeting overweight or obesity, using exergaming or social media, and 2 for anxiety, using web-based cognitive behavioral therapy (CBT). Characteristics of promising interventions included gaming features, therapist support, and parental involvement. Most were purely behavioral interventions (rather than CBT or third wave), typically using the behavior change techniques (BCTs) feedback and monitoring, shaping knowledge, repetition and substitution, and reward. Three papers included qualitative data on the user's experience. We developed the following themes: parental involvement, connection with a health professional is important for engagement, technological affordances and barriers, and child-centered design. CONCLUSIONS: Of the 17 eligible interventions, digital interventions for anxiety and overweight or obesity had the greatest promise. Using qualitative methods during digital intervention development and evaluation may lead to more meaningful, usable, feasible, and engaging interventions, especially for this underresearched younger population. The following characteristics could be considered when developing digital interventions for younger children: involvement of parents, gaming features, additional therapist support, behavioral (rather than cognitive) approaches, and particular BCTs (feedback and monitoring, shaping knowledge, repetition and substitution, and reward). This review suggests a model for improving the conceptualization and reporting of behavioral interventions involving children and parents.

Rapid Systematic Review: The Impact of Social Isolation and Loneliness on the Mental Health of Children and Adolescents in the Context of COVID-19.

OBJECTIVE: Disease containment of COVID-19 has necessitated widespread social isolation. We aimed to establish what is known about how loneliness and disease containment measures impact on the mental health in children and adolescents. METHOD: For this rapid review, we searched MEDLINE, PsycInfo, and Web of Science for articles published between January 1, 1946, and March 29, 2020. Of the articles, 20% were double screened using predefined criteria, and 20% of data was double extracted for quality assurance. RESULTS: A total of 83 articles (80 studies) met inclusion criteria. Of these, 63 studies reported on the impact of social isolation and loneliness on the mental health of previously healthy children and adolescents (n = 51,576; mean age 15.3 years). In all, 61 studies were observational, 18 were longitudinal, and 43 were cross-sectional studies assessing self-reported loneliness in healthy children and adolescents. One of these studies was a retrospective investigation after a pandemic. Two studies evaluated interventions. Studies had a high risk of bias, although longitudinal studies were of better methodological quality. Social isolation and loneliness increased the risk of depression, and possibly anxiety at the time at which loneliness was measured and between 0.25 and 9 years later. Duration of loneliness was more strongly correlated with mental health symptoms than intensity of loneliness. CONCLUSION: Children and adolescents are probably more likely to experience high rates of depression and most likely anxiety during and after enforced isolation ends. This may increase as enforced isolation continues. Clinical services should offer preventive support and early intervention where possible and be prepared for an increase in mental health problems.

"The child's got a complete circle around him". The care of younger children (5-11 years) with CFS/ME. A qualitative study comparing families', teachers' and clinicians' perspectives'.

Society needs to improve the care of children with complex needs. Guidelines recommend integrating care across health and educational settings, however, there is little research on whether this is achieved or how this can be done in practice. Our aim was to address this gap by examining how the care of children (aged 5-11 years) with Chronic Fatigue Syndrome/ Myalgic Encephalomyelitis (CFS/ME) is shared across home, education and health settings, in order to generate recommendations for integrating care. We undertook semi-structured interviews with families (22 participants), teachers (11 participants) and healthcare providers (9 participants), analysing the data thematically and comparatively. Our analysis of the data was informed by a socio-ecological perspective as we sought to understand the complexity of the relationships and systems around the child. The first theme focuses on the child ("individual level"); child-centred care is seen as essential whilst acknowledging that the child has limited capacity to manage their own care. The second theme presents the distinct roles of parents, teachers and clinicians ("interpersonal and organisational levels"). The third describes how these three levels interact in the management of the child's care, in the context of the health and education systems and policies ("policy levels"). The fourth explores optimal ways to integrate care across home, school and clinical settings. In conclusion, there is opportunity to support a child with complex health needs by targeting the systems around the child; parents, teachers and clinicians, as well as education and health policy that can enable shared-care. Involving schools in assessment, communicating diagnosis across settings and using a stepped-care approach to integrated care may be beneficial. Further work is needed to explore these recommendations, with attention to the policy factors that may act as barriers and enablers.

Physical activity patterns among children and adolescents with mild-to-moderate chronic fatigue syndrome/myalgic encephalomyelitis.

OBJECTIVE: Chronic fatigue syndromemyalgic encephalomyelitis (CFS/ME) is relatively common among children and adolescents; however, little is known about the physical activity levels and patterns of this population. The aim of this study was to examine the underlying patterns of physical activity among youth with mild-to-moderate CFS/ME. Cross-sectional associations between physical activity patterns with self-reported physical function, pain, fatigue, anxiety and depression were also examined. DESIGN: Baseline cross-sectional data from the Managed Activity Graded Exercise iN Teenagers and pre-Adolescents randomised controlled trial. PATIENTS: Children and adolescents (aged 8-17 years) diagnosed with mild-to-moderate CFS/ME who wore an accelerometer for at least three valid weekdays. ANALYSES: Latent profile analysis was used to identify physical activity patterns. Linear regression models examined associations between physical activity classes and self-reported physical function, pain, fatigue, anxiety and depression. RESULTS: 138 children and adolescents (72.5% females) had valid data. Overall, participants did less than half the government recommended level of physical activity for children and adolescents, but not all were inactive: three (2.2%) did more than 1 hour of physical activity every day, and 13 (9.4%) achieved an average of 60 min a day. Adolescents (≥12 years) were less active than younger children, but activity levels were similar between genders. Three latent classes emerged from the data: 'active', 'light' and 'inactive'. Compared with being 'inactive', being in the 'light' class was associated with greater self-reported physical function (10.35, 95% CI 2.32 to 18.38) and lower fatigue (-1.60, 95% CI -3.13 to -0.06), while being 'active' was associated with greater physical function (15.26, 95% CI 0.12 to 30.40), but also greater anxiety (13.79, 95% CI 1.73 to 25.85). CONCLUSIONS: Paediatricians need to be aware that physical activity patterns vary widely before recommending treatment. CLINICAL TRIAL REGISTRATION: ISRCTN registry: 23 962 803.

Results of the feasibility phase of the managed activity graded exercise in teenagers and pre-adolescents (MAGENTA) randomised controlled trial of treatments for chronic fatigue syndrome/myalgic encephalomyelitis.

BACKGROUND: Chronic fatigue syndrome (CFS) also known as myalgic encephalomyelitis (ME) is relatively common in young people and causes significant disability. Graded exercise therapy (GET) and activity management are recommended by the National Institute for Health and Care Excellence (NICE) despite a limited evidence-base for either treatment in paediatric CFS/ME. This paper reports on feasibility and acceptability measures from the feasibility phase of the ongoing MAGENTA randomised controlled trial (RCT) investigating GET versus activity management for young people with CFS/ME. METHODS: Setting: Three specialist secondary care National Health Service (NHS) Paediatric CFS/ME services (Bath, Cambridge and Newcastle).Participants: Young people aged 8-17 years with a diagnosis of mild to moderate CFS/ME. Young people were excluded if they were severely affected, referred to cognitive behavioural therapy (CBT) at initial assessment or unable to attend clinical sessions.Interventions: GET and activity management delivered by physiotherapists, occupational therapists, nurses and psychologists. Families and clinicians decided the number (typically 8-12) and frequency of appointments (typically every 2-6 weeks).Outcome Measures: Recruitment and follow-up statistics. We used integrated qualitative methodology to explore the feasibility and acceptability of the trial processes and the interventions. RESULTS: 80/161 (49.7%) of eligible young people were recruited at two sites between September 2015 and August 2016, indicating recruitment to the trial was feasible. Most recruitment (78/80; 97.5%) took place at one centre. Recruitment consultations, online consent and interventions were acceptable, with less than 10% in each arm discontinuing trial treatment. Response rate to the primary outcome (the SF36-PFS at 6 months) was 91.4%. Recruitment, treatment and data collection were not feasible at one centre. The site was withdrawn from the study.In response to data collected, we optimised trial processes including using Skype for recruitment discussions; adapting recruiter training to improve recruitment discussions; amending the accelerometer information leaflets; shortening the resource use questionnaires; and offering interventions via Skype. These amendments have been incorporated into the full trial protocol. CONCLUSIONS: Conducting an RCT investigating GET versus activity management is feasible and acceptable for young people with CFS/ME. TRIAL REGISTRATION: ISRCTN23962803 10.1186/ISRCTN23962803, date of registration: 03 September 2015.

How are behavioural interventions delivered to children (5-11 years old): a systematic mapping review.

CONTEXT: Behavioural interventions are used to prevent, manage and treat a wide variety of conditions including obesity, diabetes, chronic pain, asthma and emotional difficulties. There has been inadequate attention to the delivery of behavioural interventions to younger children (5-11 years old). OBJECTIVE: Our objectives were to describe the characteristics of behavioural interventions for children aged 5-11 years. DATA SOURCES: We searched five databases: CINAHL, EMBASE, PsycINFO, MEDLINE and Cochrane Library, from January 2005 to August 2019. STUDY SELECTION: The inclusion criteria were (1) children aged 5-11, (2) cognitive and/or behavioural interventions, (3) randomised controlled trials and (4) 2005 onward. Two researchers independently identified studies for inclusion. DATA EXTRACTION: Two researchers independently extracted data from eligible papers. RESULTS: The search identified 10 541 papers. We extracted information on 117 interventions (from 152 papers). Many of the interventions were categorised as complex. This was particularly true for clinical populations; 78.7% were delivered to both the child and parent, and 33.9% took place across multiple settings, typically health and school settings. Most (70.9%) were 'First Wave' (behavioural) interventions, and few (4.3%) were 'Third Wave' (characterised by metacognition, acceptance and mindfulness). Thirty-nine per cent used interactive techniques (play, arts, story and/or games). Purely digital and paper-based interventions were rare, but around a third used these tools as supplements to face-face delivery. There were differences in interventions for younger (5-7 years) and older (8-11 years) children. CONCLUSIONS: Interventions designed and delivered to children should be developmentally sensitive. This review highlights characteristics of interventions delivered to children 5-11 years old: the involvement of the child's parent, using behavioural (rather than cognitive) modalities, using interactive techniques and some interventions were delivered across multiple settings.

Defining the minimally clinically important difference of the SF-36 physical function subscale for paediatric CFS/ME: triangulation using three different methods.

BACKGROUND: Defining the minimally clinically important difference (MCID) is important for the design and analysis of clinical trials and ensures that findings are clinically meaningful. Studies in adult populations have investigated the MCID of The Short Form 36 physical function sub-scale (SF-36-PFS). However, to our knowledge no studies have defined the MCID of the SF-36-PFS in a paediatric population. We aimed to triangulate findings from distribution, anchor and qualitative methods to identify the MCID of the SF-36-PFS for children and adolescents with CFS/ME. METHODS: Quantitative methods: We analysed routinely-collected data from a specialist paediatric CFS/ME service in South-West England using: 1) the anchor method, based on Clinical Global Impression (CGI) outcomes at 6 months' follow-up; 2) the distribution method, based on the standard deviation of baseline SF-36-PFS scores. Qualitative methods: Young people (aged 12-17 years) and parents were asked to complete the SF-36-PFS, marking each question twice: once for where they would currently rate themselves/their child and a second time to show what they felt would be the smallest amount of change for them/their child to feel treatment had made a difference. Semi-structured interviews were designed to explore what factors were deemed important to patients and to what extent an improvement was considered satisfactory. We thematically analysed qualitative interviews from 21 children and their parents. RESULTS: Quantitative results: Six-month follow-up data were available for 198 children with a mean age of 14 years. Most were female (74%, 146/198) and 95% gave their ethnicity as "White British". Half the standard deviation of the baseline SF-36-PFS scores was 11.0. "A little better" on the CGI equated to a mean difference on the SF-36-PFS from baseline to 6-month follow-up of 9.0. Qualitative results: Twenty-one children with CFS/ME participated: 16 females (76.2%) with a mean age of 14.4 years. Twenty mothers and two fathers were also interviewed. The median minimal improvement in the SF-36-PFS was 10. Participants indicated that small changes in physical function can lead to important improvements in valued social and family function. Patients and parents were positive about improvement even in the presence of persisting symptoms. Triangulation: The MCID based on the mean score from the three methods was 10. CONCLUSIONS: Converging evidence indicates future studies in paediatric CFS/ME should use an MCID of 10 on the SF-36-PFS.

Using the internet to cope with chronic fatigue syndrome/myalgic encephalomyelitis in adolescence: a qualitative study.

BACKGROUND: Adolescents are increasingly using online resources for health purposes. Previous studies suggest that online provision of information about chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is neither balanced nor consistent with evidence-based practice. However, little is known about how adolescents with CFS/ME use the internet for their condition and whether this is helpful or harmful. METHODS: Nine indepth, semistructured, qualitative interviews were conducted with young people (aged 12-17) recruited from a specialist paediatric CFS/ME service. Interviews explored the types of online resources accessed, motivations for doing so and how resource use related to patterns of coping. RESULTS: Around the time of diagnosis, participants focused on gathering facts about CFS/ME and therefore used official resources (eg, National Health Service sites) that were considered reliable. This transitioned to exploring patient-led and peer-led spaces: health forums, Facebook and YouTube. Participants accessed these regularly, over the long term, and valued these sites for the personal stories, emotional content and interactive technology. Patient-led and peer-led sites supported coping, encouraging active behavioural management, providing social support and addressing stigmatised aspects of the condition. CFS/ME put a strain on normal adolescent life, such as identity and friendships. Online resources allowed participants to adapt and maintain a sense of normality. CONCLUSIONS: Adolescents who use the internet find online resources helpful in seeking information and social support for their condition. Healthcare services should improve their online resources to meet the needs of younger users, providing evidence-based content in ways that are relevant to adolescents and that can meet the needs for social support, as well as providing information.