Applications of the experience sampling method (ESM) in paediatric healthcare: a systematic review.

BACKGROUND: With the Experience Sampling Method (ESM) participants are asked to provide self-reports of their symptoms, feelings, thoughts and behaviours in daily life. This preregistered systematic review assessed how ESM is being used to monitor emotional well-being, somatic health, fatigue and pain in children and adolescents with a chronic somatic illness. METHODS: Databases were searched from inception. Studies were selected if they included children or adolescents aged 0-25 years with a chronic somatic illness and used ESM focussing on mental health or psychosocial wellbeing, biopsychosocial factors and/or somatic health. Two reviewers extracted data of the final 47 papers, describing 48 studies. RESULTS: Most studies evaluated what factors influence medical or psychological symptoms and how symptoms influence each other. Another common purpose was to study the feasibility of ESM or ESM as part of an app or intervention. Study methods were heterogeneous and most studies lack adequate reporting of ESM applications and results. CONCLUSIONS: While ESM holds great potential for providing results and feedback to patients and caregivers, little use is being made of this option. Future studies should consider what they report in their studies, conduct a priori power analyses and how ESM can be embedded in clinical practice. IMPACT: While ESM has many clinical applications, it is currently mostly used for research purposes. Current studies using ESM are heterogeneous and lack consistent, high-quality reporting. There is great potential in ESM for providing patients and parents with personalised feedback.

The Permutation Distancing Test for dependent single-case observational AB-phase design data: A Monte Carlo simulation study.

The Permutation Distancing Test (PDT) is a nonparametric test for evaluating treatment effects in dependent single-case observational design (SCOD) AB-phase data without linear trends. Monte Carlo methods were used to estimate the PDT power and type I error rate, and to compare them to those of the Single-Case Randomization Test (SCRT) assuming a randomly determined intervention point and the traditional permutation test assuming full exchangeability. Data were simulated without linear trends for five treatment effect levels (- 2, - 1, 0, 1, 2), five autocorrelation levels (0, .15, .30, .45, .60), and four observation number levels (30, 60, 90, 120). The power was calculated multiple times for all combinations of factor levels each generating 1000 replications. With 30 observations, the PDT showed sufficient power (≥ 80%) to detect medium treatment effects up to autocorrelation ≤ .45. Using 60 observations, the PDT showed sufficient power to detect medium treatment effects regardless of autocorrelation. With ≥ 90 observations, the PDT could also detect small treatment effects up to autocorrelation ≤ .30. With 30 observations, the type I error rate was 5-7%. With 60 observations and more, the type I error rate was ≤ 5% with autocorrelation < .60. The PDT outperformed the SCRT regarding power, particularly with a small number of observations. The PDT outperformed the traditional permutation test regarding type I error rate control, especially when autocorrelation increased. In conclusion, the PDT is a useful and promising nonparametric test to evaluate treatment effects in dependent SCOD AB-phase data without linear trends.

Fatigue mediates the relationship between emotional and cognitive functioning in children post-cancer treatment.

BACKGROUND/OBJECTIVES: Children treated for cancer are at risk to develop cognitive problems. Insight in underlying associations with emotional functioning and fatigue can be used to optimize interventions. We therefore aim to study emotional functioning, fatigue, and cognitive functioning in children postcancer treatment and investigate whether fatigue mediates the relationship between emotional and cognitive functioning. DESIGN/METHODS: Emotional functioning, fatigue, and cognitive functioning were assessed in children post-cancer treatment using subscales of the Pediatric Quality of Life Inventory (PedsQL) Generic Core Scales, Multidimensional Fatigue Scale and Cognitive Functioning Scale. A one sample t-test was used to compare outcomes with general population peers and mediation analysis was used to address the effect of fatigue on the relationship between emotional and cognitive functioning. RESULTS: A total of 137 children (mean age: 13.6, SD ± 3.3 years; mean time since end of treatment: 7.1 months, SD ± 5.9) participated. Lower scores on emotional functioning (Cohen's d [D]: 0.4), fatigue (D: 0.8) and cognitive functioning (D: 0.6) were found (p < .001) in children post-cancer treatment than in peers. A medium association was found between emotional and cognitive functioning (standardized regression coefficient [ß]: 0.27, p < .001), which was mediated by fatigue (ß = 0.16). CONCLUSIONS: Outcomes on emotional and cognitive functioning are decreased and fatigue is increased in children postcancer treatment. Fatigue mediates the relationship between emotional and cognitive functioning. Our results show the importance to focus on fatigue amongst stress as a target for intervention to improve cognitive functioning.

Longitudinal development of fatigue after treatment for childhood cancer: a national cohort study.

BACKGROUND: Fatigue is a distressing and prevalent long-term sequela of treatment for childhood cancer, and there is a need for longitudinal studies to investigate the development of fatigue over time. The objective of this study was to calculate growth-curves for the longitudinal development of fatigue after treatment for childhood cancer, and to investigate the effects of biopsychosocial predictors. MATERIALS AND METHODS: Participants were recruited from a patient monitoring program and data extracted from medical records. Parent-proxy and self-report versions of PedsQLTM Multidimensional Fatigue Scale were used to repeatedly assess fatigue up to 5 years after the end of treatment for childhood cancer. Fatigue was assessed 2440 times for 761 participants (median:3) with proxy-reports (age 2-8 years) and 2657 times for 990 participants with self-reports (above 8 years) (median:2). Mixed models were used to establish growth-curves and to analyze the effect of predictors separately for participants with solid tumors (ST), hemato-oncological malignancies and central nervous system-tumors (CNS). RESULTS: CNS-tumors were associated with more cognitive fatigue than ST at the end of treatment, for both proxy-reports (-11.30, p<.001) and self-reports (-6.78, p=.002), and for proxy-reports of general fatigue (-6.78, p=.002). The only significant difference in change over time was for self-reports of sleep-rest fatigue. The raw scores for the CNS-group decreased with -0.87 per year (95% CI -1.64; -0.81, p=.031) compared to the ST-group. Parental distress was overall the variable most associated with increased fatigue, while immunotherapy was the most frequent medical predictor. National centralization of childhood cancer care decreased fatigue for the CNS-group, but not for other diagnoses. DISCUSSION: Children and adolescents treated for CNS-tumors reported more fatigue than other participants after the end of treatment, and this difference remained over time. Results from this study may help to facilitate the early recognition of children with insufficient recovery of fatigue symptoms.

Self-reported quantity and quality of sleep in children and adolescents with a chronic condition compared to healthy controls.

To assess self-reported quantity and quality of sleep in Dutch children with a chronic condition compared to healthy controls and to the recommended hours of sleep for youth. Sleep quantity and quality were analyzed in children with a chronic condition (cystic fibrosis, chronic kidney disease, congenital heart disease, (auto-)immune disease, and medically unexplained symptoms (MUS); n = 291; 15 ± 3.1 years, 63% female. A subset of 171 children with a chronic condition were matched to healthy controls using Propensity Score matching, based on age and sex, ratio 1:4. Self-reported sleep quantity and quality were assessed with established questionnaires. Children with MUS were analyzed separately to distinguish between chronic conditions with and without an identified pathophysiological cause. Generally, children with a chronic condition met the recommended amount of sleep, however 22% reported poor sleep quality. No significant differences in sleep quantity and quality were found between the diagnosis groups. Children with a chronic condition and with MUS slept significantly more than healthy controls at ages 13, 15, and 16. Both at primary and secondary school, poor sleep quality was least frequent reported in children with a chronic condition and most often reported in children with MUS.  Conclusion: Overall, children with chronic conditions, including MUS, met the recommended hours of sleep for youth, and slept more than healthy controls. However, it is important to obtain a better understanding of why a substantial subset of children with chronic conditions, mostly children with MUS, still perceived their sleep quality as poor. What is Known: • According to the Consensus statement of the American Academy of Sleep medicine, typically developing children (6 to 12 years) should sleep 9 to 12 h per night, and adolescents (13 to 18 years) should sleep 8 to 10 h per night. • Literature on the optimal quantity and quality of sleep in children with a chronic condition is very limited. What is New: Our findings are important and provide novel insights: • In general, children with a chronic condition sleep according to the recommended hours of sleep. • A substantial subset of children with chronic conditions, perceived their sleep quality as poor. Although this was reported mostly by children with medically unexplained symptoms (MUS), the found poor sleep quality was independent of specific diagnosis.

Internet-delivered cognitive behavioural therapy for chronic fatigue among adolescents with a chronic medical condition: a single case study.

BACKGROUND: Severe fatigue is a prominent symptom among adolescents with a chronic medical condition, with major impact on their well-being and daily functioning. Internet-based cognitive behavioural therapy (I-CBT) is a promising treatment for severe fatigue among adolescents with a chronic medical condition, but its effectiveness has not been studied. AIMS: We developed an I-CBT intervention for disabling fatigue in a chronic medical condition and tested its feasibility and effectiveness in an adolescent with an immune dysregulation disorder (IDD), namely juvenile idiopathic arthritis (JIA). METHOD: The application of I-CBT is illustrated through a clinical case study of a 15-year-old girl with JIA and chronic severe fatigue. An A-B single case experimental design was used with randomization of the waiting period prior to start of the intervention. Outcomes were weekly measures of fatigue severity, physical functioning, school absence and pain severity. RESULTS: Fatigue severity significantly decreased following I-CBT. Improvements were observed towards increased school attendance and improved physical functioning following the intervention, but these effects were too small to become significant. CONCLUSIONS: The study provides preliminary support for the feasibility and effectiveness of the application of I-CBT for severe fatigue in adolescents with a long-term medical condition.

The PROactive cohort study: rationale, design, and study procedures.

Children with a chronic condition face more obstacles than their healthy peers, which may impact their physical, social-emotional, and cognitive development. The PROactive cohort study identifies children with a chronic disease at high risk of debilitating fatigue, decreased daily life participation and psychosocial problems, as well as children who are resilient and thrive despite the challenges of growing up with a chronic condition. Both groups will teach us how we can best support children, adolescents and parents to adapt to and manage a disease, as well as tailor interventions to their specific needs.This cohort follows a continuous longitudinal design. It is based at the Wilhelmina Children's Hospital (WKZ) in the Netherlands and has been running since December 2016. Children with a chronic condition (e.g. cystic fibrosis, juvenile idiopathic arthritis, chronic kidney disease, or congenital heart disease) as well children with medically unexplained fatigue or pain in a broad age range (2-18 years) are included, as well as their parent(s). Data are collected from parents (of children between 2 and 18 years) and children (8-18 years), as well as data from their electronic health record (EHR). Primary outcome measures are fatigue, daily life participation, and psychosocial well-being, all assessed via patient- and proxy-reported outcome measures. Generic biological/lifestyle, psychological, and social factors were assessed using clinical assessment tools and questionnaires. In the PROactive cohort study the research assessment is an integrated part of clinical care. Children are included when they visit the outpatient clinic and are followed up annually.

Psychosocial functioning in adolescents growing up with chronic disease: The Dutch HBSC study.

Many adolescents worldwide (indirectly) grow up with a chronic disease, which may impact their functioning and wellbeing. The objective of this study is to assess whether adolescents with a (family member with a) chronic disease differ from their healthy counterparts in terms of psychosocial functioning. Data from the Dutch 2013 HBSC-survey were used, including 7168 adolescents (Meanage = 13.7, SD = 1.57, 50.5% female). Participants indicated whether they or one of their family members had a long-term (> 3 months) disease or disability (mental/physical) and were categorized into four groups based on disease presence (none, other, self, both). Psychosocial functioning was assessed in terms of life satisfaction, self-rated health, psychosomatic health, mental health problems, support, substance use, physical exercise, screen time, and school liking. Chronically diseased adolescents (n = 162) reported lower life satisfaction, self-rated and psychosomatic health, more mental health problems, lower peer support, more substance use, and less physical exercise compared to healthy peers. Chronically diseased adolescents who also had a family member with a chronic disease (n = 74) showed comparable outcomes on these life domains, although they did not differ from their healthy peers regarding peer support, substance use, and physical activity. Healthy adolescents with a chronically diseased family member (n = 737) reported significantly lower life satisfaction, self-rated and psychosomatic health, more mental health problems, and less family support compared to healthy peers who grew up in healthy families; however, they reported more positive outcomes than adolescents who had a chronic disease themselves.Conclusion: Having a (family member with a) chronic disease is associated with impaired psychosocial functioning on various life domains. Our findings aid in understanding the psychosocial associates of chronic disease and imply that caregivers should be observant of psychosocial problems among vulnerable adolescents to provide appropriate guidance. What is Known: • Adolescents who grow up with a (family member with a) chronic disease encounter numerous challenges that may be related to poorer developmental outcomes on the long term. What is New: • This study adds a comprehensive overview of the psychosocial functioning of adolescents with a (family member with a) chronic disease, as compared to healthy counterparts that grow up in a healthy family.

Severe Fatigue Is Common Among Pediatric Patients with Primary Immunodeficiency and Is Not Related to Disease Activity.

PURPOSE: Fatigue is a distressing symptom commonly reported among pediatric patients with primary immunodeficiency (PID). However, the relationship between fatigue and disease activity is currently unknown. METHODS: In this cross-sectional study, we examined the prevalence of severe fatigue, the effect of fatigue on health-related quality of life (HRQoL), and the effects of disease activity and comorbidity on fatigue severity among pediatric patients 2-18 years of age with PID. Fatigue and HRQoL were assessed using the pediatric quality of life inventory multidimensional fatigue scale (PedsQL MFS) and generic core scales (PedsQL GCS), respectively. Linear regression analyses and an analysis of covariance were used to compare the fatigue scores with the scores obtained from a healthy control group. Data were adjusted for age and sex. RESULTS: Of the 91 eligible patients, 79 were assessed (87% participation rate), with a mean age of 10.4 ± 4.4 years. Pediatric patients with PID reported significantly higher fatigue levels compared to healthy peers, with an 18.9% prevalence of severe fatigue. Moreover, higher fatigue levels were inversely associated with HRQoL in all domains and directly associated with school absences. We found that severe fatigue was comparable between common variable immunodeficiency (CVID), combined immunodeficiency (CID), and selective immunoglobulin A deficiency (SIgAD) patients, but was not reported in the X-linked agammaglobulinemia (XLA) patients studied. Finally, fatigue severity was not significantly associated with disease activity or comorbidity. CONCLUSIONS: Nearly 20% of pediatric patients with PID reported experiencing severe fatigue, and fatigue was reported among a wide range of PID subcategories. In addition, severe fatigue negatively affected the patient's quality of life and daily functioning, but was not associated with disease activity or comorbidity. Thus, targeting severe fatigue might be a promising strategy for improving the overall well-being and quality of life of pediatric patients with PID.

Mental Well-being and General Health in Adolescents with Asthma: The Prevention and Incidence of Asthma and Mite Allergy Birth Cohort Study.

OBJECTIVES: To assess whether adolescents with asthma experience a lower mental well-being and lower general health than their peers without asthma. STUDY DESIGN: Data from the Prevention and Incidence of Asthma and Mite Allergy study were used. At the ages of 11, 14, 17, and 20 years, 2651, 2522, 2094, and 2206 participants, respectively, completed questionnaires. Their parents completed questionnaires at the ages of 11 (n = 2660), 14 (n = 2338), and 17 years (n = 1872). Asthma was defined according to the Mechanisms of the Development of Allergy criteria. Mental well-being was measured using the Mental Health Index-5 and was reported by the adolescents. General health, measured on a 4-point Likert scale, was reported by the adolescents and their parents. We estimated associations of asthma with mental well-being and perceived general health using generalized estimating equations. RESULTS: At ages 11, 14, 17, and 20 years, 6.7%, 6.9%, 5.0%, and 6.6%, respectively, of the adolescents had asthma. Adolescents with asthma did not score differently on the Mental Health Index than their peers without asthma. Adolescents with asthma were less likely to experience good or excellent health than their peers without asthma (aOR, 0.37; 95% CI, 0.26-0.51 for intermittent asthma and 0.33; 95% CI, 0.25-0.41 for persistent asthma). These results remain similar across the different ages. CONCLUSIONS: The mental well-being of adolescents with asthma is similar to that of their peers without asthma, although adolescents with asthma are less likely to perceive a good or excellent general health.

Effectiveness of Internet-Based Cognitive Behavior Therapy (Fatigue in Teenagers on the Internet) for Adolescents With Chronic Fatigue Syndrome in Routine Clinical Care: Observational Study.

BACKGROUND: Internet-based cognitive behavior therapy (I-CBT) for adolescents with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) has been shown to be effective in a randomized controlled trial (RCT; Fatigue in Teenagers on the Internet [FITNET]). FITNET can cause a significant reduction in fatigue and disability. OBJECTIVE: We aimed to investigate whether FITNET treatment implemented in routine clinical care (IMP-FITNET) was as effective, using the outcomes of the FITNET RCT as the benchmark. METHODS: Outcomes of CFS/ME adolescents who started IMP-FITNET between October 2012 and March 2018 as part of routine clinical care were compared to the outcomes in the FITNET RCT. The primary outcome was fatigue severity assessed posttreatment. The secondary outcomes were self-reported physical functioning, school attendance, and recovery rates. Clinically relevant deterioration was assessed posttreatment, and for this outcome, a face-to-face CBT trial was used as the benchmark. The attitude of therapists toward the usability of IMP-FITNET was assessed through semistructured interviews. The number of face-to-face consultations during IMP-FITNET was registered. RESULTS: Of the 384 referred adolescents with CFS/ME, 244 (63.5%) started IMP-FITNET, 84 (21.9%) started face-to-face CBT, and 56 (14.6%) were not eligible for CBT. Posttreatment scores for fatigue severity (mean 26.0, SD 13.8), physical functioning (mean 88.2, SD 15.0), and full school attendance (mean 84.3, SD 26.5) fell within the 95% CIs of the FITNET RCT. Deterioration of fatigue and physical functioning after IMP-FITNET was observed at rates of 1.2% (n=3) and 4.1% (n=10), respectively, which is comparable to a waiting list condition (fatigue: 1.2% vs 5.7%, χ21=3.5, P=.06; physical functioning: 4.1% vs 11.4%, χ21=3.3, P=.07). Moreover, 41 (16.8%) IMP-FITNET patients made use of face-to-face consultations. CONCLUSIONS: IMP-FITNET is an effective and safe treatment for adolescents with CFS/ME in routine clinical care.

Recruiting Adolescents With Chronic Fatigue Syndrome/Myalgic Encephalomyelitis to Internet-Delivered Therapy: Internal Pilot Within a Randomized Controlled Trial.

BACKGROUND: Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) in adolescents is common and disabling. Teenagers in the United Kingdom are more likely to recover if they access specialist care, but most do not have access to a local specialist CFS/ME service. Delivering treatment remotely via the internet could improve access to treatment. OBJECTIVE: This study aims to assess (1) the feasibility of recruitment and retention into a trial of internet-delivered specialist treatment for adolescents with CFS/ME and (2) the acceptability of trial processes and 2 web-based treatments (to inform continuation to full trial). METHODS: This study is an internal pilot for the initial 12 months of a full randomized controlled trial (RCT), with integrated qualitative methods (analysis of recruitment consultations and participant and clinician interviews). Recruitment and treatment were delivered remotely from a specialist pediatric CFS/ME treatment service within a hospital in South West United Kingdom. Adolescents (aged 11-17 years) from across the United Kingdom with a diagnosis of CFS/ME and no access to local specialist treatment were referred by their general practitioner to the treatment center. Eligibility assessment and recruitment were conducted via remote methods (telephone and on the web), and participants were randomized (via a computer-automated system) to 1 of 2 web-based treatments. The trial intervention was Fatigue in Teenagers on the InterNET in the National Health Service, a web-based modular CFS/ME-specific cognitive behavioral therapy program (designed to be used by young people and their parents or caregivers) supported by individualized clinical psychologist electronic consultations (regular, scheduled therapeutic message exchanges between participants and therapist within the platform). The comparator was Skype-delivered activity management with a CFS/ME clinician (mainly a physiotherapist or occupational therapist). Both treatments were intended to last for up to 6 months. The primary outcomes were (1) the number of participants recruited (per out-of-area referrals received between November 1, 2016, to October 31, 2017) and the proportion providing 6-month outcome data (web-based self-report questionnaire assessing functioning) and (2) the qualitative outcomes indicating the acceptability of trial processes and treatments. RESULTS: A total of 89 out of 150 (59.3% of potentially eligible referrals) young people and their parents or caregivers were recruited, with 75 out of 89 (84.2%) providing 6-month outcome data. Overall, web-based treatment was acceptable; however, participants and clinicians described both the advantages and disadvantages of remote methods. No serious adverse events were reported. CONCLUSIONS: Recruiting young people (and their parents or caregivers) into an RCT of web-based treatment via remote methods is feasible and acceptable. Delivering specialist treatment at home via the internet is feasible and acceptable, although some families prefer to travel across the United Kingdom for face-to-face treatment. TRIAL REGISTRATION: ISRCTN 18020851; http://www.isrctn.com/ISRCTN18020851. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): RR2-10.1186/s13063-018-2500-3.

Review about the impact of growing up with a chronic disease showed delays achieving psychosocial milestones.

AIM: This review aimed to provide a comprehensive overview of the psychosocial developmental trajectory of various diseases during childhood and adolescence. METHODS: Studies of Dutch young adults aged 18-35 years, who had grown up with a chronic disease, were included if the Course of Life Questionnaire had been used to assess psychosocial developmental milestones in three domains: social, autonomy and psychosexual. Differences between the disease groups and the general population were presented as Cohen's d and odds ratios. RESULTS: We included 17 studies comprising 1899 young adults, who had grown up with 18 different paediatric diseases. Psychosocial development was delayed in all three questionnaire domains. Remarkable findings with regard to specific milestones were as follows: less participation in sports clubs in the social domain, less likely to have had paid jobs in the autonomy domain and later sexual intimacy in the psychosexual domain. End-stage renal disease, galactosaemia (males), childhood cancer and orthotopic liver transplants were the most affected disease groups. CONCLUSION: Children and adolescents with chronic diseases risked delays in psychosocial development. This should be addressed by healthcare providers, along with the physical aspects of diseases, and they should focus on the optimal psychosocial development of the patient.

Non-pharmacological options for managing chronic musculoskeletal pain in children with pediatric rheumatic disease: a systematic review.

In patients with a pediatric rheumatic disease (PRD), chronic musculoskeletal pain (CMP) can have a major impact on functioning and social participation. Because CMP is not always alleviated solely by the use of pharmacological approaches, the aim was to systematically review the available evidence regarding non-pharmacological treatment options for reducing CMP in patients with PRD. PubMed, Embase, PsycINFO, and the Cochrane Library were systematically searched for (non-)randomized trials investigating non-pharmacological treatments for CMP in PRD published through October 25, 2017. The GRADE approach was used to assess the quality of evidence. The search yielded 11 studies involving 420 children 5-18 years of age. All studies were relatively small and short-term, and the quality of evidence ranged from very low to moderate. The main modalities within non-pharmacology therapy were psychological interventions and exercise-based interventions. Some studies show modest positive short-term results for psychological and exercise-based interventions. Psychological and exercise-based interventions can have a modest positive result in PRD, with no evidence of side effects. Non-pharmacological therapies are a promising option to alleviate pain in PRD and improve functioning, which can be used as an alternative for or in addition to pharmacological therapies. Because chronic pain can differ etiologically from acute pain in PRD, non-pharmacological therapies might have different effects in patients with or without active inflammation. To best determine the effect of non-pharmacological therapies, future studies should take this difference into account.

The impact of chronic fatigue syndrome on cognitive functioning in adolescents.

Chronic fatigue syndrome (CFS) is characterized by persistent fatigue and severe disability. Most adolescent patients report attention and concentration problems, with subsequent poor performance at school. This study investigated the impact of CFS on intellectual capacity by (1) assessing discrepancies between current intelligence quotient (IQ) and school level and (2) exploring differences in current IQ and pre-CFS school performance, compared with healthy individuals. Current data was cross-sectionally gathered and compared with retrospective pre-CFS school performance data. Fifty-nine CFS adolescents and 40 controls were evaluated on performance on age-appropriate intelligence tests and school level. Current IQ scores of CFS adolescents were lower than expected on the basis of their school level. Furthermore, there was a difference in intelligence performance across time when current IQ scores were compared with pre-CFS cognitive achievement. Healthy controls did not show any discrepancies. CONCLUSION: According to their pre-CFS intelligence assessments, CFS patients started with appropriate secondary school levels at the age of 12. Our data suggest that CFS may be accompanied by a decline in general cognitive functioning. Given the critical age for intellectual development, we recommend a timely diagnosis followed by appropriate treatment of CFS in adolescents. WHAT IS KNOWN: Adolescent chronic fatigue syndrome (CFS) is a debilitating condition with major impact on social and intellectual development. Most patients report concentration problems, with subsequent poor performance at school. Little is known about the influence of CFS on intellectual performances. WHAT IS NEW: IQ scores of CFS adolescents are lower than the IQ scores of healthy peers with an equivalent school level. There is a decrease in intelligence performance across time when current IQ scores are compared with pre-CFS cognitive achievement. Healthy controls do not show any discrepancies between their current IQ, school level and previous cognitive functioning. This suggest that adolescent CFS may be accompanied by a decline in general cognitive functioning.

Individual outcomes after tailored versus generic self-management strategies for persistent fatigue in youth with a fatigue syndrome or rheumatic condition: A multiple single-case study.

OBJECTIVE: To examine individual outcomes after tailored lifestyle (PROfeel) or generic dietary advice as self-management intervention for persistent fatigue in adolescents and young adults with a chronic condition, to compare participants who did and did not benefit and to explore changes to factors in the biopsychosocial model of fatigue after PROfeel. METHOD: A multiple single-case AB-phase design was embedded in a randomized crossover trial (N = 45). Intensive longitudinal data (ILD) on outcomes 'fatigue severity', 'self-efficacy' and 'quality of life' (QoL) were collected through weekly smartphone measurement for 20 weeks. ILD on biopsychosocial factors were collected through experience sampling methodology for 28 days pre-post first intervention. Baseline characteristics were compared with t-tests and chi-square tests. Permutation distancing tests were used to assess change over time in all ILD. RESULTS: Regarding weekly measurements, nineteen participants (42.22%) showed small to large positive outcomes (drange = .05 to 2.59), mostly after PROfeel. Eleven participants (24.44%) showed small to moderate negative outcomes (drange = -.02 to -2.46), mostly after dietary advice. Fatigue severity improved most, followed by self-efficacy. Participants who benefitted showed higher QoL levels and lower fatigue and pain levels compared with others at baseline (all p < .02). When positive outcomes were observed after PROfeel, typically ≥1 biopsychosocial factor had been targeted successfully. CONCLUSION: Self-management advice has more potential when tailored to individual characteristics, including the biopsychosocial model of fatigue. PROfeel appears particularly useful as fatigue intervention for individuals with relatively less severe symptoms.

Gamification in eHealth for Chronic Disease Self-Management in Youth: A Systematic Review.

This systematic review primarily aims to provide a summary of the game mechanics implemented in eHealth tools supporting young people's self-management of their chronic diseases. This review secondarily investigates the rationale for implementing game mechanics and the effects of these tools. A systematic search was conducted in Embase, Medline, PsycINFO, and Web of Science, from inception until August 30, 2022. Studies were eligible if focus was on the utilization of gamification in eHealth self-management interventions for young people (age = 10-25 years) with chronic diseases. Primary quantitative, qualitative, and mixed-method studies written in English were included. We identified 34 eHealth tools, of which 20 (59%) were gamified tools and 14 (41%) were serious games. We found that 55 unique game mechanics were implemented. The most commonly used were rewards (50%), score (44%), creative control (41%), and social interaction (32%). In comparison with gamified tools, the number and diversity of game mechanics applied were higher in serious games. For most tools (85%), a general rationale was provided for utilizing gamification, which often was to promote engaging experiences. A rationale for using specific game mechanics was less commonly provided (only for 45% of the game mechanics). The limited availability of experimental research precludes to test the effectiveness of using gamification in eHealth to support self-management in young people with chronic diseases. In this study, we highlight the importance of reporting the rationale for utilizing specific game mechanics in eHealth tools to ensure a proper alignment with evidence-based practice and the need of conducting experimental research. PROSPERO: CRD42021293037.

A dyadic perspective on parent-child dyadic coping in children with a chronic condition.

OBJECTIVE: In this study, we examined the extent to which parents and their children with a chronic condition communicate their stress to one another and whether stress communication is associated with different forms of dyadic coping. METHODS: In a sample of 239 parent-child dyads, self-reported stress communication and different forms of perceived dyadic coping (i.e., emotion-oriented, problem-oriented, and negative dyadic coping) were assessed using a cross-sectional design. RESULTS: We first found that children's stress communication was positively associated with more positive (r = 0.28, p < .001) and less negative dyadic coping responses by children (r = -0.22, p < .001). Children's stress communication was also associated with more positive (r = 0.52, r = 0.45, p's < 0.001), and less negative dyadic coping responses by parents (r = -0.19, p < .001). Using dyadic data of children with a chronic condition and their parents, we found that more stress communication of children was associated with healthier coping responses of both children (perceived emotion-oriented dyadic coping: ß = 0.23, p < .001) and parents (perceived emotion-oriented dyadic coping: ß = 0.33, p < .001; perceived problem-oriented dyadic coping: ß = 0.22, p < .001). CONCLUSION: This underscores the importance of communication and adaptive coping strategies of parents and children in the context of a child's chronic condition. These findings may help us find ways to support children and their parents to optimally communicate about and deal with their stress.

Effectiveness of internet-based cognitive behavioural treatment for adolescents with chronic fatigue syndrome (FITNET): a randomised controlled trial.

BACKGROUND: Chronic fatigue syndrome is characterised by persistent fatigue and severe disability. Cognitive behavioural therapy seems to be a promising treatment, but its availability is restricted. We developed Fatigue In Teenagers on the interNET (FITNET), the first dedicated internet-based therapeutic program for adolescents with this disorder, and compared its effectiveness with that of usual care. METHODS: Adolescents aged 12-18 years with chronic fatigue syndrome were assigned to FITNET or usual care in a 1:1 ratio at one tertiary treatment centre in the Netherlands by use of a computer-generated blocked randomisation allocation schedule. The study was open label. Primary outcomes were school attendance, fatigue severity, and physical functioning, and were assessed at 6 months with computerised questionnaires. Analysis was by intention to treat. Thereafter, all patients were offered FITNET if needed. This trial is registered, number ISRCTN59878666. FINDINGS: 68 of 135 adolescents were assigned to FITNET and 67 to usual care, and 67 and 64, respectively, were analysed. FITNET was significantly more effective than was usual care for all dichotomised primary outcomes at 6 months-full school attendance (50 [75%] vs 10 [16%], relative risk 4·8, 95% CI 2·7-8·9; p<0·0001), absence of severe fatigue (57 [85%] vs 17 [27%], 3·2, 2·1-4·9; p<0·0001), and normal physical functioning (52 [78%] vs 13 [20%], 3·8, 2·3-6·3; p<0·0001). No serious adverse events were reported. INTERPRETATION: FITNET offers a readily accessible and highly effective treatment for adolescents with chronic fatigue syndrome. The results of this study justify implementation on a broader scale. FUNDING: Netherlands Organisation for Health Research and Development.

Clinical Practice: Chronic fatigue syndrome.

The diagnosis chronic fatigue syndrome (CFS) was conceptualized in the mid-1980s. It is a clinically defined condition characterized by severe and disabling new onset fatigue with at least four additional symptoms: impaired memory or concentration, sore throat, tender cervical or axillary lymph nodes, muscle pain, multi-joint pain, new headaches, unrefreshing sleep or post-exertion malaise. Chronic fatigue syndrome in adolescents is a rare condition compared to symptomatic fatigue. The estimated prevalence of adolescent CFS ranges between 0.11 and 1.29 % in Dutch, British, and US populations. Diagnosis of the chronic fatigue syndrome is established through exclusion of other medical and psychiatric causes of chronic fatiguing illness. Taking a full clinical history and a full physical examination are therefore vital. In adolescence, CFS is associated with considerable school absence with long-term detrimental effects on academic and social development. One of the most successful potential treatments for adolescents with CFS is cognitive behavioural therapy, which has been shown to be effective after 6 months in two thirds of the adolescents with CFS. This treatment effect sustains at 2-3-year follow-up. In conclusion, the diagnosis CFS should be considered in any adolescent patient with severe disabling long-lasting fatigue. Cognitive behavioural therapy is effective in 60-70 % of the patients. Prompt diagnosis favours the prognosis.