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Abstract Pediatric chronic pain places a significant burden on children, their families, and healthcare services. Effective pain measurement is needed for both clinical management and research. Digital pain measurement tools have been developed for adult and adolescent populations however less is known about measurement in younger children. In this systematic review, we aimed to identify, describe, and evaluate (in terms of acceptability) digital tools for the assessment of chronic pain in children (5–11 years). We searched five databases (Cochrane Library, EMBASE, MEDLINE, PsycINFO, and CINAHL), between January 2014 and January 2022. We included empirical studies which included digital tool/s to assess pain in children aged between 5–11 years with chronic pain conditions. We independently double‐screened the papers to determine eligibility. We followed PRISMA guidelines for reporting. A total of five papers, covering four digital tools, were included. The digital tools used ranged from a static online survey to a highly interactive, personalized tablet application. Two studies were cross‐sectional and two collected longitudinal pain data via electronic devices outside the clinical setting. Digital features of the tools included: dynamic testing (n = 2), notifications/prompts (n = 1), data transmission (n = 1), remote monitoring (n = 1), accessibility (n = 1), data visualization/feedback (n = 1), personalization/customization (n = 1), gamification (n = 1) and data labeling (n = 1). Qualitative usability data was only available for one of the tools, which indicated its acceptability and highlighted preferred features/functions by child users (creative and personalizable features, gamification features), and parental users (symptom tracking). This review has highlighted the limited number of digital assessment tools available for children with chronic pain aged 5–11. This review identified some examples of technology enabling the capture of longitudinal, repeated measurement of multiple dimensions of pain (intensity, location, quality). We suggest directions for future research.

Design Parallel-group randomised controlled trial. Methods Participants Adolescents aged 11–17 years, diagnosed with myalgic encephalomyelitis/chronic fatigue syndrome and with no local specialist treatment centre, were referred to a specialist service in South West England. Interventions Fatigue In Teenagers on the interNET in the National Health Service is a web-based myalgic encephalomyelitis/chronic fatigue syndrome-focused cognitive–behavioural therapy programme for adolescents, supported by individualised written, asynchronous electronic consultations with a clinical psychologist/cognitive–behavioural therapy practitioner. The comparator was videocall-delivered activity management with a myalgic encephalomyelitis/chronic fatigue syndrome clinician. Both treatments were intended to last 6 months. Objectives Estimate the effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management for paediatric myalgic encephalomyelitis/chronic fatigue syndrome. Estimate the effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management for those with mild/moderate comorbid mood disorders. From a National Health Service perspective, estimate the cost-effectiveness of Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management over a 12-month horizon. Primary Outcome 36-item Short Form Health Survey Physical Function subscale at 6 months post randomisation. Randomisation Web-based, using minimisation with a random component to balance allocated groups by age and gender. Blinding While the investigators were blinded to group assignment, this was not possible for participants, parents/carers and therapists. Results The treatment of 314 adolescents was randomly allocated, 155 to Fatigue In Teenagers on the interNET in the National Health Service. Mean age was 14 years old and 63% were female. Primary outcome At 6 months, participants allocated to Fatigue In Teenagers on the interNET in the National Health Service were more likely to have improved physical function (mean 60.5, standard deviation 29.5, n = 127) compared to Activity Management (mean 50.3, standard deviation 26.5, n = 138). The mean difference was 8.2 (95% confidence interval 2.7 to 13.6, p = 0.003). The result was similar for participants meeting the National Institute for Health and Care Excellence 2021 diagnostic criteria. Secondary outcomes Fatigue In Teenagers on the interNET in the National Health Service participants attended, on average, half a day more school per week at 6 months than those allocated Activity Management, and this difference was maintained at 12 months. There was no strong evidence that comorbid mood disorder impacted upon the relative effectiveness of the two interventions. Similar improvement was seen in the two groups for pain and the Clinical Global Impression scale, with a mixed picture for fatigue. Both groups continued to improve, and no clear difference in physical function remained at 12 months [difference in means 4.4 (95% confidence interval −1.7 to 10.5)]. One or more of the pre-defined measures of a worsening condition in participants during treatment, combining therapist and patient reports, were met by 39 (25%) participants in the Fatigue In Teenagers on the interNET in the National Health Service group and 42 (26%) participants in the Activity Management group. A small gain was observed for the Fatigue In Teenagers on the interNET in the National Health Service group compared to Activity Management in quality-adjusted life-years (0.002, 95% confidence interval −0.041 to 0.045). From an National Health Service perspective, the costs were £1047.51 greater in the Fatigue In Teenagers on the interNET in the National Health Service group (95% confidence interval £624.61 to £1470.41). At a base cost-effectiveness threshold of £20,000 per quality-adjusted life-year, the incremental cost-effectiveness ratio was £457,721 with incremental net benefit of −£1001 (95% confidence interval −£2041 to £38). Conclusion At 6 months post randomisation, compared with Activity Management, Fatigue In Teenagers on the interNET in the National Health Service improved physical function and school attendance. The additional cost of Fatigue In Teenagers on the interNET in the National Health Service and limited sustained impact mean it is unlikely to be cost-effective. Trial registration This trial is registered as ISRCTN18020851. Funding This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: 14/192/109) and is published in full in Health Technology Assessment; Vol. 28, No. 70. See the NIHR Funding and Awards website for further award information. Plain language summary Why did we do the study? The best evidence for the treatment of adolescents with myalgic encephalomyelitis/chronic fatigue syndrome is cognitive–behavioural therapy for fatigue delivered in person. In the United Kingdom, most adolescents with myalgic encephalomyelitis/chronic fatigue syndrome cannot get this specialist treatment where they live. Fatigue In Teenagers on the interNET in the National Health Service is an online treatment using cognitive–behavioural therapy designed for myalgic encephalomyelitis/chronic fatigue syndrome, which has been shown to work in the Netherlands. To find out if Fatigue In Teenagers on the interNET in the National Health Service would be beneficial in the United Kingdom, we compared Fatigue In Teenagers on the interNET in the National Health Service to Activity Management. Activity Management is the treatment most often offered to children and young people with myalgic encephalomyelitis/chronic fatigue syndrome in the United Kingdom, and aims to avoid peaks in activity (sometimes called ‘pacing’). What was the question? Does Fatigue In Teenagers on the interNET in the National Health Service lead to greater improvements in children and young people with myalgic encephalomyelitis/chronic fatigue syndrome when compared to Activity Management, when both interventions are delivered remotely? What did we do? We compared Fatigue In Teenagers on the interNET in the National Health Service and Activity Management in two comparable groups of children, and measured physical function at 6 months as the main indication of improvement. We measured how much the treatments cost and we asked children and young people, their parents and treatment providers what they thought about the two interventions. What did we find? At 6 months, adolescents saw greater improvements in physical function, and attended half a day more school per week, with Fatigue In Teenagers on the interNET in the National Health Service compared to Activity Management. Both interventions were associated with improvements over 12 months, with there being no clear difference between them after that time. However, the Fatigue In Teenagers on the interNET in the National Health Service treatment was more expensive. What does this mean? We have shown that cognitive–behavioural therapy for fatigue can be provided online to children as Fatigue In Teenagers on the interNET in the National Health Service, leading to faster improvement in physical function and greater school attendance compared to Activity Management. However, Fatigue In Teenagers on the interNET in the National Health Service is expensive and is unlikely to be good value for money. Scientific summary Objectives Primary objective To investigate whether cognitive–behavioural therapy (CBT) specifically designed for myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and delivered over the internet [Fatigue In Teenagers on the interNET in the National Health Service (FITNET-NHS)] is effective and cost-effective compared to Activity Management for children with ME/CFS who do not have access to a local specialist ME/CFS service. Secondary objectives (1) Estimate the effectiveness of FITNET-NHS compared to Activity Management for those with mild/moderate comorbid mood disorders (anxiety/depression). (2) Estimate the cost-effectiveness of FITNET-NHS compared to Activity Management. (3) Estimate the cost-effectiveness of FITNET-NHS compared to Activity Management for those with mild/moderate comorbid mood disorders (anxiety/depression). Methods Trial design Randomised controlled trial (RCT) comparing FITNET-NHS with Activity Management. Participants were allocated in a 1 : 1 ratio, minimised by age and gender. We conducted an internal pilot (first 12 months) and integrated qualitative methods to optimise recruitment and retention. Participants Adolescents aged 11–17 years with a diagnosis of ME/CFS [defined using National Institute for Health and Care Excellence (NICE) 2007 Criteria], who did not have a local specialist ME/CFS service. To have a confirmed diagnosis, potential participants were required to have been assessed by a paediatrician (or equivalent specialist doctor), and to have had screening bloods taken. We excluded adolescents if: they were not disabled by fatigue; their fatigue was due to another cause (including primary anxiety/depression); they had access to a local specialist service; they were unable to complete video calls or FITNET-NHS treatment modules; they were pregnant. Setting We identified potentially eligible adolescents at referral to the specialist paediatric ME/CFS service at the Royal United Hospital, Bath. Patients were referred to the service by their general practitioner (GP) or paediatrician (or equivalent specialist). Between September 2018 and March 2020, we offered GP surgeries the opportunity to become patient identification centres. In these sites, database searches were conducted to identify potentially eligible adolescents, who were then offered referral to the Bath Royal United Hospital for eligibility assessment. Recruitment Potentially eligible adolescents were contacted by the clinical team by telephone to discuss the opportunity to take part in the FITNET-NHS trial. Interested adolescents were sent study information, including a link to the Revised Children’s Anxiety and Depression Scale (RCADS) questionnaire. Eligibility assessment Adolescents who were interested were invited to an eligibility assessment conducted by a specialist nurse. The nurse checked if: (1) they had debilitating persistent or relapsing fatigue for at least 3 months, but not life-long; (2) fatigue was not the result of ongoing exertion and not substantially alleviated by rest; (3) they had post-exertional malaise (and increase in fatigue and other symptoms after exertion); and (4) their fatigue was severe enough to cause substantial reduction in previous levels of occupational, educational, social or personal activities. The recruiting nurse checked that the screening blood tests had been done within the previous 12 months and were normal. The nurse then assessed whether the adolescent had comorbid mental health problems or a primary mental health problem. Patients were asked to complete the RCADS. Those scoring within the borderline or above the age-/gender-validated threshold answered further questions to determine if they were at risk of harm and whether their anxiety/depression was sufficiently severe to explain the fatigue. This enabled us to ensure that those recruited had a diagnosis of ME/CFS from a specialist but also had 3 months of disabling fatigue plus one further symptom and did not have an exclusionary diagnosis and therefore had ME/CFS according to the contemporary United Kingdom (UK) guideline. Consent If adolescents were eligible and interested in taking part in the FITNET-NHS trial, consent and assent (patients aged 11–15 years) were obtained using an online form. Randomisation We used an automated web randomisation service operated by Bristol Trials Centre. Participants were allocated in a 1 : 1 ratio. Allocation used minimisation to balance by age and gender with a random component to preserve allocation concealment. Blinding It was not possible to blind the participant, family or the clinical service. The investigators (including the senior statistician who wrote the statistical analysis plan) were blinded to treatment allocation. The study statistician was unblinded as they reported to the Data Safety and Monitoring Committee. Interventions Fatigue In Teenagers on the interNET in the National Health Service (FITNET-NHS) This is a web-based modular specialist CBT programme designed to be used by adolescents with ME/CFS and their parents. We adapted the Dutch FITNET platform for UK adolescents. FITNET-NHS is delivered using asynchronous individualised e-consultations within the web-based platform. Contact is weekly initially and then becomes less frequent. The therapist works with patients and parents separately. FITNET-NHS has psycho-educational and CBT chapters for patients and a parallel programme for parents. There are 19 chapters. Chapters 1–3 introduce ME/CFS, CBT and the role of therapists; Chapter 4 discusses treatment goals; Chapters 5–19 are about cognitive and behavioural strategies. There are diaries for patients that are visible to the therapist. FITNET-NHS is individualised for patients. Chapters 5–19 are unlocked (made available) by the therapist according to clinical presentation, needs and formulation. Fidelity was assessed in clinical supervisions. Activity Management Participants and their families received information on ME/CFS, Activity Management, sleep and symptoms management. Participants could use an online app to record their activity. Activity Management was delivered via videocall. In the initial assessment, participants had a detailed assessment of physical and cognitive activity. The participant and therapist agreed a ‘baseline’ of activity, which is the average level of activity. Participants were asked to record activity, and then, when activity was stable, to increase activity gradually in a flexible and individual way. The initial assessment was 90 minutes (but this could be split into two shorter sessions). Follow-up video calls (60 minutes each) were organised 2–6 weeks apart. In November 2017, the number of follow-up sessions was increased from 3 to 6 in response to participant feedback. Fidelity was assessed using a checklist of mandatory, flexible and prohibited elements. Data sources Adolescent- and parent-completed measures, collected online using research electronic data capture. Therapist reports. Routinely collected data from local systems and from NHS Digital. Qualitative interviews with adolescents, parents and therapists. Measures Primary outcome Disability measured using the 36-item Short Form Health Survey Physical Function subscale (SF-36-PFS) measured 6 months after randomisation. We included outcome data returned in a 5-to-9-month window post randomisation. Secondary outcomes (Measured at 3, 6 and 12 months unless specified): Physical function (3 and 12 months); fatigue [Chalder fatigue scale and checklist individual strength (CIS) fatigue scale]; school attendance (self-reported days per week attending school, or whether receiving home tuition); mental health {RCADS; pain [visual analogue scale (VAS)]; Clinical Global Impression Scale; quality of life [EuroQol-5 Dimensions Youth (EQ-5D-Y)]; parent-completed resource use questionnaire; parent-completed work productivity and activity impairment questionnaire general health (WPAI:GH)}. Demographic data were collected at recruitment and included: age, sex, postcode, ethnicity, symptoms, months since illness onset, presence of comorbid illnesses. Harms/adverse events We prospectively collected the following data: (1) clinician-reported serious deterioration in health, (2) a decrease of ≥ 20 in SF-36-PFS between baseline and 3, 6 or 12 months or scores of ‘much’ or ‘very much’ worse on the Clinical Global Impression scale or (3) withdrawal from treatment because of feeling worse. Safety outcomes were reported to the Data Safety Monitoring Committee. Resource use data In addition to study documentation and patient/parent questionnaires, we used patient-level data recorded on the Royal United Hospitals Bath NHS Trusts electronic patient record system (Millennium). We obtained secondary care data on outpatient visits, inpatient visits and emergency department attendances from NHS Digital. Costs were valued using 2019/20 prices. Analysis Sample size Our sample size of 314 participants (assuming 15% attrition) gives 90% power at 5% significance to detect a 10-point (0.4 standard deviation) difference for the SF-36-PFS for our primary outcome. The original sample size was powered to detect a difference in the subgroup with mental health problems, but this was revised during the study. Statistical methods We used an intention-to-treat analysis in study participants who completed the primary outcome. We used multivariable linear regression analysis adjusting for baseline values of the outcome, age and gender. The treatment effect was estimated as an adjusted difference between sample means. We conducted the following pre-planned sensitivity analyses: we adjusted for variation across participants in the time between randomisation and the 6-month outcome; the primary analysis was repeated with an additional binary covariate distinguishing participants recruited before or after 1 September 2019 (distinguishing those with a 6-month assessment before or during the COVID-19 pandemic); we repeated the analysis in those who completed one or more modules/sessions of their allocated intervention. We conducted a sensitivity analysis for participants with ME/CFS according to the NICE (2021) criteria. We adapted the regression model used for the primary analysis to the secondary outcome variables. We estimated the effectiveness of FITNET-NHS compared with Activity Management in participant subgroups defined by the presence or absence of anxiety or depression (defined by the RCADS). Health economic analysis We used an intention-to-treat approach and multiple imputation by chained equation to minimise bias due to missing data. We combined cost and quality-adjusted life-year (QALY) data to calculate an incremental cost-effectiveness ratioand an incremental net monetary benefit (iNMB) statistic. We performed a subgroup analysis to explore the interaction between comorbid anxiety/depression and cost-effectiveness. Prespecified sensitivity analyses include conducting a complete-case analysis and repeating the primary analysis using the tariff paid by Clinical Commissioning Groups instead of reference costs. Qualitative methods We integrated qualitative methods into the pilot and main phase of the trial to explore trial conduct, recruitment and intervention acceptability. We analysed recruitment to trial consultations and conducted in-depth interviews with recruiters, trial therapists and participants. Results were used to improve recruitment and make small changes to the interventions. Qualitative analysis was ongoing and iterative, commencing soon after data collection. Audio recordings were transcribed, checked and imported into NVivo (QSR International, Warrington, UK). The data were systematically assigned codes and analysed thematically using techniques of constant comparison. Results Of 892 referrals between 1 November 2016 and 31 October 2020, 550 were eligible, of which 155 were allocated to FITNET-NHS and 159 to Activity Management. 265 adolescents were included in the primary analysis (127 for FITNET-NHS and 138 for Activity Management). The baseline characteristics were similar between the treatment arms. 147 participants had either comorbid depression or anxiety (145 depression, 34 anxiety and 32 both). The number completing 80% or more of expected modules/sessions was lower for FITNET-NHS participants (58, 38%) compared to Activity Management participants (124, 78%). Participants in the FITNET-NHS group had a greater improvement in physical function compared to the Activity Management group at 6 months [mean difference 8.2, 95% confidence interval (CI) 2.7 to 13.6]. The sensitivity analyses confirmed the primary analysis. This was true for participants with ME/CFS defined using the NICE (2021) criteria. At 6 months, those allocated to FITNET-NHS were, on average, attending half a day more of school per week compared to Activity Management, and this difference was maintained at 12 months. They experienced less fatigue (with the CIS fatigue measure) at both 6 and 12 months. There was no strong evidence that either treatment was more effective in those with comorbid depression/anxiety, and similar levels of improvement were noted for pain, the Clinical Global Improvement Score, and fatigue measured using the Chalder Fatigue Scale. Participants continued to improve between 6 and 12 months in both treatment arms, and there was little difference in physical function between the two treatment arms at 12 months. Fatigue In Teenagers on the interNET in the National Health Service participants had a small gain in QALYs (0.002, 95% CI −0.041 to 0.045) compared to Activity Management but substantially higher mean costs (£1047.51, 95% CI £624.61 to £1470.41). In the primary analysis, from an NHS perspective, at a threshold of £20,000 per QALY, the iNMB was −£1001.74 (95% CI −£2041.31 to £37.83), indicating that FITNET-NHS is unlikely to be cost-effective. The wide Cis show there is considerable uncertainty in this result. The subgroup analysis suggests that FITNET-NHS is more likely to be cost-effective among those with comorbid anxiety and depression at baseline. In the qualitative interviews, families felt online treatment could fit around everyday life and reduce the increase in symptoms that can accompany travelling to face-to-face appointments. However, some participants still preferred in-person treatment and found it difficult to build a rapport with therapists online. Personalised contact with a therapist was valued in both treatments and remains essential even in online treatment. The reading and writing required on the FITNET-NHS platform was difficult for younger children and those with cognitive symptoms such as brain fog; therefore, adaptations may be needed for these groups. Therapists felt they had to develop a different set of skills to engage patients online. Conclusions Despite the fact that adolescents are more likely to have better physical function at 6 months and attend more school (at 6 and 12 months) after receiving FITNET-NHS compared to Activity Management, FITNET-NHS is unlikely to be cost-effective. This study is consistent with previous RCTs that demonstrated the effectiveness of CBT for children and young people with ME/CFS. It is the first study to attempt to look at cost-effectiveness. The high additional cost of FITNET-NHS and limited substantial sustained impact mean that it may not be a cost-effective use of NHS funds. Alternatively, it is possible that the EQ-5D-Y is not sensitive enough to appropriately reflect the improvements in physical function and school attendance. Most participants in the FITNET-NHS group did not complete 80% or more of the recommended treatment modules. While our qualitative data suggest that FITNET-NHS was acceptable to most patients, it was considered burdensome by some. Implications for health care This study strengthens the evidence that CBT is effective for adolescents with ME/CFS and should be the first line of treatment offered. FITNET-NHS is an intensive treatment approach and is unlikely to be cost-effective. However, the online approach was popular with patients and families. Recommendations for research Would a shorter intervention with less intensive therapist input increase retention and reduce costs without jeopardising effectiveness? We need a validated health economic measure for children and young people that is sensitive to change. Further research needs to be conducted on the best method to deliver remote treatment. A large implementation study in the Netherlands suggested that many patients (and therapists) prefer a mixture of face-to-face and online treatment. Trial registration This trial is registered as ISRCTN18020851. Funding This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: 14/192/109) and is published in full in Health Technology Assessment; Vol. 28, No. 70. See the NIHR Funding and Awards website for further award information.

Abstract Background The factors which influence participant retention in paediatric randomised controlled trials are under-researched. Retention may be more challenging due to child developmental stages, involving additional participants, and proxy-reporting of outcomes. This systematic review and meta-analysis explores the factors which may influence retention in paediatric trials. Methods Using the MEDLINE database, paediatric randomised controlled trials published between 2015 and 2019 were identified from six general and specialist high-impact factor medical journals. The review outcome was participant retention for each reviewed trial’s primary outcome. Context (e.g. population, disease) and design (e.g. length of trial) factors were extracted. Retention was examined for each context and design factor in turn, with evidence for an association being determined by a univariate random-effects meta-regression analysis. Results Ninety-four trials were included, and the median total retention was 0.92 (inter-quartile range 0.83 to 0.98). Higher estimates of retention were seen for trials with five or more follow-up assessments before the primary outcome, those less than 6 months between randomisation and primary outcome, and those that used an inactive data collection method. Trials involving children aged 11 and over had the higher estimated retention compared with those involving younger children. Those trials which did not involve other participants also had higher retention, than those where they were involved. There was also evidence that a trial which used an active or placebo control treatment had higher estimated retention, than treatment-as-usual. Retention increased if at least one engagement method was used. Unlike reviews of trials including all ages of participants, we did not find any association between retention and the number of treatment groups, size of trial, or type of treatment. Conclusions Published paediatric RCTs rarely report the use of specific modifiable factors that improve retention. Including multiple, regular follow-ups with participants before the primary outcome may reduce attrition. Retention may be highest when the primary outcome is collected up to 6 months after a participant is recruited. Our findings suggest that qualitative research into improving retention when trials involve multiple participants such as young people, and their caregivers or teachers would be worthwhile. Those designing paediatric trials also need to consider the use of appropriate engagement methods. Research on Research (RoR) registry https://ror-hub.org/study/2561

While non-operative treatment has emerged as an alternative to surgery for the treatment of uncomplicated acute appendicitis in children, comparative patient-centred outcomes are not well documented. We investigated these in a feasibility randomised trial. Of 57 randomised participants, data were available for 26. Compared with appendicectomy, children allocated to non-operative treatment reported higher short-term quality of life scores, shorter duration of requiring analgesia, more rapid return to normal activities and shorter parental absence from work. These preliminary data suggest differences exist in recovery profile and quality of life between these treatments that are important to measure in a larger RCT. Trial registration number is ISRCTN15830435.

Abstract Background To update and internally validate a model to predict children and young people (CYP) most likely to experience long COVID (i.e. at least one impairing symptom) 3 months after SARS-CoV-2 PCR testing and to determine whether the impact of predictors differed by SARS-CoV-2 status. Methods Data from a nationally matched cohort of SARS-CoV-2 test-positive and test-negative CYP aged 11–17 years was used. The main outcome measure, long COVID, was defined as one or more impairing symptoms 3 months after PCR testing. Potential pre-specified predictors included SARS-CoV-2 status, sex, age, ethnicity, deprivation, quality of life/functioning (five EQ-5D-Y items), physical and mental health and loneliness (prior to testing) and number of symptoms at testing. The model was developed using logistic regression; performance was assessed using calibration and discrimination measures; internal validation was performed via bootstrapping and the final model was adjusted for overfitting. Results A total of 7139 (3246 test-positives, 3893 test-negatives) completing a questionnaire 3 months post-test were included. 25.2% (817/3246) of SARS-CoV-2 PCR-positives and 18.5% (719/3893) of SARS-CoV-2 PCR-negatives had one or more impairing symptoms 3 months post-test. The final model contained SARS-CoV-2 status, number of symptoms at testing, sex, age, ethnicity, physical and mental health, loneliness and four EQ-5D-Y items before testing. Internal validation showed minimal overfitting with excellent calibration and discrimination measures (optimism-adjusted calibration slope: 0.96575; C-statistic: 0.83130). Conclusions We updated a risk prediction equation to identify those most at risk of long COVID 3 months after a SARS-CoV-2 PCR test which could serve as a useful triage and management tool for CYP during the ongoing pandemic. External validation is required before large-scale implementation.

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.

This study emphasises the critical role of quality sleep in physical and mental well-being, exploring its impact on bodily recovery and cognitive function. Investigating poor sleep quality in approximately 40% of individuals with insomnia symptoms, the research delves into its potential diagnostic relevance for depression and anxiety, with a focus on intervention in mental health by understanding sleep patterns, especially in young individuals. This study includes an exploration of phone usage habits among young adults during PPI sessions, providing insights for developing the SleepTracker app. This pivotal tool utilises phone usage and movement data from mobile device sensors to identify indicators of anxiety or depression, with participant information organised comprehensively in a table categorising condition related to phone usage and movement data. The analysis compares this data with survey results, incorporating scores from the Sleep Condition Indicator (SCI), Patient Health Questionnaire-9 (PHQ-9), and Generalised Anxiety Disorder-7 (GAD-7). Generated confusion matrices offer a detailed overview of the relationship between sleep metrics, phone usage, and movement data. In summary, this study reveals the accurate detection of negative sleep disruption instances by the classifier. However, improvements are needed in identifying positive instances, reflected in the F1-score of 0.5 and a precision result of 0.33. While early intervention potential is significant, this study emphasises the need for a larger participant pool to enhance the model’s performance.

BackgroundSince the era of smartphones started in early 2007, they have steadily turned into an accepted part of our lives. Poor sleep is a health problem that needs to be closely monitored before it causes severe mental health problems, such as anxiety or depression. Sleep disorders (eg, acute insomnia) can also develop to chronic insomnia if not treated early. More specifically, mental health problems have been recognized to have casual links to anxiety, depression, heart disease, obesity, dementia, diabetes, and cancer. Several researchers have used mobile sensors to monitor sleep and to study changes in individual mood that may cause depression and anxiety. ObjectiveExtreme sleepiness and insomnia not only influence physical health, they also have a significant impact on mental health, such as by causing depression, which has a prevalence of 18% to 21% among young adults aged 16 to 24 in the United Kingdom. The main body of this narrative review explores how passive data collection through smartphone sensors can be used in predicting anxiety and depression. MethodsA narrative review of the English language literature was performed. We investigated the use of smartphone sensors as a method of collecting data from individuals, regardless of whether the data source was active or passive. Articles were found from a search of Google Scholar records (from 2013 to 2020) with keywords including “mobile phone,” “mobile applications,” “health apps,” “insomnia,” “mental health,” “sleep monitoring,” “depression,” “anxiety,” “sleep disorder,” “lack of sleep,” “digital phenotyping,” “mobile sensing,” “smartphone sensors,” and “sleep detector.” ResultsThe 12 articles presented in this paper explain the current practices of using smartphone sensors for tracking sleep patterns and detecting changes in mental health, especially depression and anxiety over a period of time. Several researchers have been exploring technological methods to detect sleep using smartphone sensors. Researchers have also investigated changes in smartphone sensors and linked them with mental health and well-being. ConclusionsThe conducted review provides an overview of the possibilities of using smartphone sensors unobtrusively to collect data related to sleeping pattern, depression, and anxiety. This provides a unique research opportunity to use smartphone sensors to detect insomnia and provide early detection or intervention for mental health problems such as depression and anxiety if insomnia is detected.

Abstract 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.

Abstract Background Mental health conditions have been shown to disproportionately affect those from Black, Asian and Minority Ethnic (BAME) communities. Somali communities globally have relatively high levels of mental illness, but low levels of mental health service use, with numerous barriers to care identified. This study was conducted in an established UK Somali community in the South West of England and aimed to explore community beliefs and views about the causes of mental illness, treatment for mental illness, and access to medical services in general. Participants were asked about how mental health and illness are understood and conceptualised, along with the cultural meaning of mental illness and its manifestations in relation to men, women and young people. Design Using a community-based participatory research design, in partnership with local Somali community organisations, the research team conducted four focus groups with a total of 23 participants aged over 18. Open-ended questions were used to facilitate discussion. Transcripts were analysed thematically. Results The participants discussed the role of migration and associated stress from the civil war and how that could contribute to mental illness. Participants tended to view the symptoms of mental illness as physical manifestations such as headaches and to describe a strong community stigma where those with mental health conditions were viewed as “crazy” by others. Barriers to accessing healthcare included language barriers, waiting times and a mistrust of doctors. Various ideas for improvements were discussed, including ideas to reduce stigma and ideas for community initiatives. Conclusion Cultural considerations and reducing stigma are vital in improving understanding of mental illness and improving access to mental health services, along with building relationships and trust between the Somali community and health care workers.

Abstract Background Chronic fatigue syndrome (CFS, also known as myalgic encephalomyelitis (ME)) is defined as fatigue that is disabling, is accompanied by additional symptoms and persists for ≥ 4 months. Treatment of CFS/ME aims to help patients manage their symptoms and make lifestyle adjustments. We do not know whether intervening early in primary care (

Background Paediatric chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is disabling and relatively common. Although evidenced-based treatments are available, at least 15% of children remain symptomatic after one year of treatment. Acceptance and commitment therapy (ACT) is an alternative therapy option; however, little is known about whether it is an acceptable treatment approach. Our aim was to find out if adolescents who remain symptomatic with CFS/ME after 12 months of treatment would find ACT acceptable, to inform a randomised controlled trial (RCT) of ACT.Methods We recruited adolescents (diagnosed with CFS/ME; not recovered after one year of treatment; aged 11–17 years), their parent/carer and healthcare professionals (HCPs) from one specialist UK paediatric CFS/ME service. We conducted semi-structured interviews to explore barriers to recovery; views on current treatments; acceptability of ACT; and feasibility of an effectiveness RCT. Thematic analysis was used to identify patterns in data.Results Twelve adolescents, eleven parents and seven HCPs were interviewed. All participants thought ACT was acceptable. Participants identified reasons why ACT might be efficacious: pragmatism, acceptance and compassion are valued in chronic illness; values-focussed treatment provides motivation and direction; psychological and physical needs are addressed; normalising difficulties is a useful life-skill. Some adolescents preferred ACT to cognitive behavioural therapy as it encouraged accepting (rather than challenging) thoughts. Most adolescents would consent to an RCT of ACT but a barrier to recruitment was reluctance to randomisation. All HCPs deemed ACT feasible to deliver. Some were concerned patients might confuse ‘acceptance’ with ‘giving up’ and called for clear explanations. All participants thought the timing of ACT should be individualised.Conclusions All adolescents with CFS/ME, parents and HCPs thought ACT was acceptable, and most adolescents were willing to try ACT. An RCT needs to solve issues around randomisation and timing of the intervention.

Abstract 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 https://doi.org/10.1186/ISRCTN23962803, date of registration: 03 September 2015

Abstract Background There is a lack of patient derived, child specific outcome measures to capture what health outcomes are important to children with Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME). We developed a new Patient Reported Outcome Measure (PROM) for paediatric CFS/ME through qualitative research with children. This study aimed to pre-test the new measure through cognitive interviews with children with CFS/ME. Methods Cognitive interviews were undertaken in children’s homes or over Skype. The Three-Step Test-Interview (TSTI) method was used to assess the quality of the draft PROM with children with CFS/ME to identify problems with initial content and design and test modifications over subsequent interview rounds. Children were purposively sampled from a single specialist paediatric CFS/ME service in England. Results Twenty-four children and their parents took part. They felt the new measure captured issues relevant to their condition and preferred it to the generic measures they completed in clinical assessment. Changes were made to item content and phrasing, timeframe and response options and tested through three rounds of interviews. Conclusions Cognitive interviews identified problems with the draft PROM, enabling us to make changes and then confirm acceptability in children aged 11–18. Further cognitive interviews are required with children 8–10 years old to examine the acceptability and content validity and provide evidence for age related cut offs of the new PROM to meet FDA standards. This study demonstrates the content validity of the new measure as relevant and acceptable for children with CFS/ME. The next stage is to undertake a psychometric evaluation to support the reduction of items, confirm the structure of the PROM and provide evidence of the data quality, reliability and validity.

Abstract Background Studies in both paediatric and psychiatric settings often experience problems in recruitment. This can compromise the ability of the study to recruit to target, meaning studies are potentially underpowered. It can also result in a biased sample if a non-representative group are selectively recruited. Recruitment to studies in health contexts often depends on healthcare professionals, who act as gatekeepers by screening patients for eligibility and obtaining consent for the research team to contact them. The experience of health professionals as gatekeepers in paediatric studies is poorly understood and may affect whether recruitment is successful or not. Methods Six out of seven eligible healthcare professionals from a specialist paediatric chronic fatigue syndrome (CFS) team were interviewed. All participants were undertaking initial clinical assessments within which they were asked to identify eligible patients for an observational study of co-morbid mental health problems in adolescents with confirmed CFS/ME. This study had experienced particular recruitment problems, more so than other studies in the same service. Interview questions were designed to explore perceptions of research, and barriers and facilitators of recruitment. Interviews were audio recorded and transcribed verbatim. Thematic analysis was used. Results Participants espoused their commitment to the value of research. However, they perceived there to be a number of barriers to recruitment. Barriers within the clinical context included time pressures and the emotional nature of initial clinical assessments. Barriers posed by the wider research context included recruiting to multiple studies at the same time. Factors specific to the observational study of mental health in CFS/ME included aspects of the study design, such as the name and nature of the study, as well as the focus of the study itself. Participants made a number of recommendations about how recruitment barriers could be overcome. Conclusions The current study highlights the need to carefully consider, at design stage, how to overcome potential barriers to recruitment. Gatekeepers should be actively involved at this stage to ensure that the study is set up in such a way to best enable recruitment activities within the clinical setting.

Introduction There is uncertainty surrounding the diagnosis, prevalence, phenotype, duration and treatment of Long COVID. This study aims to (A) describe the clinical phenotype of post-COVID symptomatology in children and young people (CYP) with laboratory-confirmed SARS-CoV-2 infection compared with test-negative controls, (B) produce an operational definition of Long COVID in CYP, and (C) establish its prevalence in CYP.Methods and analysis A cohort study of SARS-CoV-2-positive CYP aged 11–17 years compared with age, sex and geographically matched SARS-CoV-2 test-negative CYP. CYP aged 11–17 testing positive and negative for SARS-CoV-2 infection will be identified and contacted 3, 6, 12 and 24 months after the test date. Consenting CYP will complete an online questionnaire. We initially planned to recruit 3000 test positives and 3000 test negatives but have since extended our target. Data visualisation techniques will be used to examine trajectories over time for symptoms and variables measured repeatedly, separately by original test status. Summary measures of fatigue and mental health dimensions will be generated using dimension reduction methods such as latent variables/latent class/principal component analysis methods. Cross-tabulation of collected and derived variables against test status and discriminant analysis will help operationalise preliminary definitions of Long COVID.Ethics and dissemination Research Ethics Committee approval granted. Data will be stored in secure Public Health England servers or University College London’s Data Safe Haven. Risks of harm will be minimised by providing information on where to seek support. Results will be published on a preprint server followed by journal publication, with reuse of articles under a CC BY licence. Data will be published with protection against identification when there are small frequencies involved.Trial registration number ISRCTN34804192; Pre-results.

Background: Although non-operative treatment is known to be effective for the treatment of uncomplicated acute appendicitis in children, randomised trial data comparing important outcomes of non-operative treatment with those of appendicectomy are lacking. Objectives: The objectives were to ascertain the feasibility of conducting a multicentre randomised controlled trial comparing the clinical effectiveness and cost-effectiveness of a non-operative treatment pathway with appendicectomy for the treatment of uncomplicated acute appendicitis in children. Design: This was a mixed-methods study, which included a feasibility randomised controlled trial, embedded and parallel qualitative and survey studies, a parallel health economic feasibility study and the development of a core outcome set. Setting: This study was set in three specialist NHS paediatric surgical units in England. Participants: Children (aged 4–15 years) clinically diagnosed with uncomplicated acute appendicitis participated in the feasibility randomised controlled trial. Children, their families, recruiting clinicians and other health-care professionals involved in caring for children with appendicitis took part in the qualitative study. UK specialist paediatric surgeons took part in the survey. Specialist paediatric surgeons, adult general surgeons who treat children, and children and young people who previously had appendicitis, along with their families, took part in the development of the core outcome set. Interventions: Participants in the feasibility randomised controlled trial were randomised to a non-operative treatment pathway (broad-spectrum antibiotics and active observation) or appendicectomy. Main outcome measures: The primary outcome measure was the proportion of eligible patients recruited to the feasibility trial. Data sources: Data were sourced from NHS case notes, questionnaire responses, transcribed audio-recordings of recruitment discussions and qualitative interviews. Results: Overall, 50% (95% confidence interval 40% to 59%) of 115 eligible patients approached about the trial agreed to participate and were randomised. There was high acceptance of randomisation and good adherence to trial procedures and follow-up (follow-up rates of 89%, 85% and 85% at 6 weeks, 3 months and 6 months, respectively). More participants had perforated appendicitis than had been anticipated. Qualitative work enabled us to communicate about the trial effectively with patients and families, to design and deliver bespoke training to optimise recruitment and to understand how to optimise the design and delivery of a future trial. The health economic study indicated that the main cost drivers are the ward stay cost and the cost of the operation; it has also informed quality-of-life assessment methods for future work. A core outcome set for the treatment of uncomplicated acute appendicitis in children and young people was developed, containing 14 outcomes. There is adequate surgeon interest to justify proceeding to an effectiveness trial, with 51% of those surveyed expressing a willingness to recruit with an unchanged trial protocol. Limitations: Because the feasibility randomised controlled trial was performed in only three centres, successful recruitment across a larger number of sites cannot be guaranteed. However, the qualitative work has informed a bespoke training package to facilitate this. Although survey results suggest adequate clinician interest to make a larger trial possible, actual participation may differ, and equipoise may have changed over time. Conclusions: A future effectiveness trial is feasible, following limited additional preparation, to establish appropriate outcome measures and case identification. It is recommended to include a limited package of qualitative work to optimise recruitment, in particular at new centres. Future work: Prior to proceeding to an effectiveness trial, there is a need to develop a robust method for distinguishing children with uncomplicated acute appendicitis from those with more advanced appendicitis, and to reach agreement on a primary outcome measure and effect size that is acceptable to all stakeholder groups involved. Trial registration: Current Controlled Trials ISRCTN15830435. Funding: This project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 25, No. 10. See the NIHR Journals Library website for further project information.

Objective This systematic review aims to synthesise and evaluate structural MRI (sMRI) and functional MRI (fMRI) studies in chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME).Methods We systematically searched Medline and Ovid and included articles from 1991 (date of Oxford diagnostic criteria for CFS/ME) to first April 2019. Studies were selected by predefined inclusion and exclusion criteria. Two reviewers independently reviewed the titles and abstracts to determine articles for inclusion, full text and quality assessment for risk of bias.Results sMRI studies report differences in CFS/ME brain anatomy in grey and white matter volume, ventricular enlargement and hyperintensities. Three studies report no neuroanatomical differences between CFS/ME and healthy controls. Task-based fMRI investigated working memory, attention, reward and motivation, sensory information processing and emotional conflict. The most consistent finding was CFS/ME exhibited increased activations and recruited additional brain regions. Tasks with increasing load or complexity produced decreased activation in task-specific brain regions.Conclusions There were insufficient data to define a unique neural profile or biomarker of CFS/ME. This may be due to inconsistencies in finding neuroanatomical differences in CFS/ME and the variety of different tasks employed by fMRI studies. But there are also limitations with neuroimaging. All brain region specific volumetric differences in CFS/ME were derived from voxel-based statistics that are biased towards group differences that are highly localised in space. fMRI studies demonstrated both increases and decreases in activation patterns in CFS/ME, this may be related to task demand. However, fMRI signal cannot differentiate between neural excitation and inhibition or function-specific neural processing. Many studies have small sample sizes and did not control for the heterogeneity of this clinical population. We suggest that with robust study design, subgrouping and larger sample sizes, future neuroimaging studies could potentially lead to a breakthrough in our understanding of the disease.

Background: There are few specialist paediatric Chronic Fatigue Syndrome (CFS/ME) services in the UK. Therefore, the distance some families have to travel to reach these services can be a barrier to accessing evidence-based treatment. Videoconferencing technology such as Skype provides a means of delivering sessions remotely. This study aimed to explore the views of children and young people, their parents, and healthcare professionals of treatment delivered by videoconferencing in a specialist paediatric CFS/ME team. Method: To explore the experiences of the participants, a qualitative design was selected. Twelve young people (age 9–18), and 6 parents were interviewed about their experience of treatment sessions delivered via videoconferencing within a specialist CFS/ME service. A focus group explored the views of healthcare professionals (N = 9) from the service. Thematic analysis was used. Results: Three themes were identified from the data: “Challenges and concerns”, “Benefits” and “Treatment provision”. Challenges and concerns that participants identified were; difficulties experienced with technology; a sense of a part of communication being lost with virtual connections; privacy issues with communicating online and feeling anxious on a screen. Participants felt that benefits of videoconferencing were; improving access to the chronic fatigue service; convenience and flexibility of treatment provision; a sense of being more open online and being in the comfort of their own home. In terms of treatment provision participants talked about videoconferencing as a part of a hierarchy of communication; the function of videoconferencing within the context of the chronic fatigue service; additional preparation needed to utilise videoconferencing and an assumption that videoconferencing is “part of young people's lives”. Conclusions: Although the experience of sessions provided by videoconferencing was different to sessions attended in person, participants tended to be positive about videoconferencing as an alternative means of accessing treatment, despite some barriers. Videoconferencing could be an additional option within an individualised care plan, but should not be an alternative to face to face support. Keywords: Chronic fatigue syndrome, Myalgic encephalomyelitis, Skype, Video-conferencing, Adolescents

Abstract 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.

Abstract The FITNET-NHS Trial is a UK, national, trial investigating whether an online cognitive behavioural therapy program (FITNET-NHS) for treating chronic fatigue syndrome/ME in adolescents is clinically effective and cost-effective in the NHS. At the time of writing (September 2019), the trial was recruiting participants. This article presents an update to the planned sample size and data collection duration previously published within the trial protocol. Trial registration ISRCTN, ID: 18020851. Registered 8 April 2016.

Abstract Background NHS specialist chronic fatigue syndrome (CFS/ME) services in England treat approximately 8000 adult patients each year. Variation in therapy programmes and treatment outcomes across services has not been described. Methods We described treatments provided by 11 CFS/ME specialist services and we measured changes in patient-reported fatigue (Chalder, Checklist Individual Strength), function (SF-36 physical subscale, Work & Social Adjustment Scale), anxiety and depression (Hospital Anxiety & Depression Scale), pain (visual analogue rating), sleep (Epworth, Jenkins), and overall health (Clinical Global Impression) 1 year after the start of treatment, plus questions about impact of CFS/ME on employment, education/training and domestic tasks/unpaid work. A subset of these outcome measures was collected from former patients 2–5 years after assessment at 7 of the 11 specialist services. Results Baseline data at clinical assessment were available for 952 patients, of whom 440 (46.2%) provided 1-year follow-up data. Treatment data were available for 435/440 (98.9%) of these patients, of whom 175 (40.2%) had been discharged at time of follow-up. Therapy programmes varied substantially in mode of delivery (individual or group) and number of sessions. Overall change in health 1 year after first attending specialist services was ‘very much’ or ‘much better’ for 27.5% (115/418) of patients, ‘a little better’ for 36.6% (153/418), ‘no change’ for 15.8% (66/418), ‘a little worse’ for 12.2% (51/418), and ‘worse’ or ‘very much worse’ for 7.9% (33/418). Among former patients who provided 2- to 5-year follow-up (30.4% (385/1265)), these proportions were 30.4% (117/385), 27.5% (106/385), 11.4% (44/385), 13.5% (52/385), and 17.1% (66/385), respectively. 85.4% (327/383) of former patients responded “Yes” to “Do you think that you are still suffering from CFS/ME?” 8.9% (34/383) were “Uncertain”, and 5.7% (22/383) responded “No”. Conclusions This multi-centre NHS study has shown that, although one third of patients reported substantial overall improvement in their health, CFS/ME is a long term condition that persists for the majority of adult patients even after receiving specialist treatment.

Abstract Background Few studies have explored patients’ experiences of treatment for CFS/ME. This study aims to fill this gap by capturing the perspective of patients who have been treated by NHS specialist CFS/ME services in England. Methods Semi-structured interviews were conducted during the period June–September 2014 with 16 adults who were completing treatment at one of three outpatient NHS specialist CFS/ME services. Interviews were analysed thematically using constant comparison techniques, with particular attention paid to contrasting views. Results Three themes were identified: ‘Journey to specialist services’; ‘Things that help or hinder treatment’; and ‘Support systems’. Within these themes nine sub-themes were identified. A wide range of factors was evident in forming participants’ experiences, including personal characteristics such as perseverance and optimism, and service factors such as flexibility and positive, supportive relationships with clinicians. Participants described how specialist services played a unique role, which was related to the contested nature of the condition. Many participants had experienced a lack of validation and medical and social support before attending a specialist service. Patients’ experiences of life before referral, and the concerns that they expressed about being discharged, highlighted the hardship and obstacles which people living with CFS/ME continue to experience in our society. Conclusions The experiences of CFS/ME patients in our study showed that NHS specialist CFS/ME services played a vital role in patients’ journeys towards an improved quality of life. This improvement came about through a process which included validation of patients’ experiences, acceptance of change, practical advice and support, and therapeutic outcomes.

Abstract Background Our aim was to investigate patterns of health care resource use by patients before and after a diagnosis of CFS/ME, as recorded by Clinical Practice Research Datalink (CPRD) GP practices in the UK. Methods We used a case–control study design in which patients who had a first recorded diagnosis of CFS/ME during the period 01/01/2001 to 31/12/2013 were matched 1:1 with controls by age, sex, and GP practice. We compared rates of GP consultations, diagnostic tests, prescriptions, referrals, and symptoms between the two groups from 15 years (in adults) or 10 years (in children) before diagnosis to 10 years after diagnosis. Results Data were available for 6710 adult and 916 child (age

Background Paediatric chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is common (prevalence 1%–2%). Two-thirds of children experience moderate or severe pain, which is associated with increased fatigue and poorer physical function. However, we do not know if treatment for CFS/ME improves pain.Objective Identify whether specialist treatment of paediatric CFS/ME improves pain.Methods We conducted a detailed search in MEDLINE, EMBASE, PsycINFO and the Cochrane Library. Two researchers independently screened texts published between 1994 and 24 January 2019 with no language restrictions. Inclusion criteria were (1) randomised controlled trials and observational studies; (2) participants aged

Objectives Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is common in children and adolescents; however, little is known about how we should define recovery. This study aims to explore perceptions of recovery held by paediatric patients with CFS/ME and their parents.Methods Children with CFS/ME and their parents were recruited through a single specialist paediatric CFS/ME service. Data were collected through semistructured interviews with children and parents. The interview questions explored how participants would know if they/their child had recovered from CFS/ME. Thematic analysis was used to identify patterns within the data.Results Twenty-one children with CFS/ME, twenty mothers and two fathers were interviewed. Some children found it hard to define recovery as the illness had become a ‘new normal’. Others thought recovery would indicate returning to pre-morbid levels of activity or achieving the same activity level as peers (socialising, education and leisure activities). Increased flexibility in routines and the absence of payback after activities were important. The interviews highlighted the concept of recovery as highly individual with wide variation in symptoms experienced, type and level of activity that would signify recovery. Parents describe how changes in mood and motivation would signify their child’s recovery, but children did not reflect on this.Conclusion Some parents and children struggle to define what would constitute complete recovery. However, signs of recovery were more easily identifiable. Definitions of recovery went far beyond symptom reduction and were focused towards rebuilding lives.

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.

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

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.

Objective As part of a larger qualitative study to explore outcomes important in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME) and what improvements in fatigue and disability are key, interviews were undertaken with adolescents and their parents. This paper focuses on their descriptions of fatigue, fluctuation of symptoms and payback.Design and setting Semistructured qualitative interviews were undertaken between December 2014 and February 2015. Adolescents and parents were interviewed separately. Participants were recruited from a single specialist paediatric chronic fatigue service. Interviews were audio recorded, transcribed verbatim and analysed using thematic analysis.Participants We interviewed 21 adolescents and their parents (20 mothers and 2 fathers). The adolescents were aged between 12 and 17 years of age (mean age 14.4 years), mild to moderately affected by CFS/ME (not housebound) and the majority were female (16/21).Results All adolescents with CFS/ME reported fatigue, a natural fluctuation of the condition, with good days and bad days as well as an increase in symptoms after activity (payback). However, adolescent’s descriptions of fatigue, symptoms and the associated impact on their daily lives differed. The variations included: fatigue versus a collection of symptoms, constant versus variable symptoms and variable symptom severity. There were differences between participants in the amount of activity taken to cause payback. The impact of fatigue and symptoms on function ranged from: limiting the duration and amount of leisure activities, struggling with daily activities (eg, self-care) to no activity (sedentary).Conclusions Fatigue, fluctuation of the condition and payback after activity are described by all adolescents with CFS/ME in this study. However, the individual experience in terms of how they describe it and the degree and impact varies.

Objective To investigate differences between young children, adolescents and adults with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME).Study design Comparison of clinical cohorts from 8 paediatric and 27 adult CFS/ME services in the UK and a paediatric randomised controlled trial from the Netherlands. Outcome measures include: fatigue (the UK—Chalder Fatigue Scale); Disability (the UK—SF-36 physical function subscale; the Netherlands—CHQ-CF87); school attendance, pain, anxiety and depression (the UK—Hospital Anxiety & Depression Scale, Spence Children's Anxiety Scale; the Netherlands—Spielberger State-Trait Anxiety Inventory for Children, Children's Depression Inventory); symptoms; time-to-assessment; and body mass index. We used multinomial regression to compare younger (aged