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.

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.

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.

Targeting persistent fatigue with tailored versus generic self-management strategies in adolescents and young adults with a fatigue syndrome or rheumatic condition: A randomized crossover trial.

OBJECTIVES: To evaluate the use of two self-management intervention strategies for persistent fatigue in adolescents and young adults with a fatigue syndrome or rheumatic condition. DESIGN: A randomized crossover trial administering tailored lifestyle advice and generic dietary advice, each 12 weeks, with a four-week washout period between. METHODS: Sixty participants (aged 12-29) were included. Tailoring was achieved through the PROfeel method. Dietary guidelines were conceptualized by the Netherlands Nutrition Centre. Questionnaires were used pre-post-interventions to measure primary outcome 'fatigue severity' (Checklist Individual Strength-8) and secondary outcomes 'self-efficacy' (Self-Efficacy Scale-28) and 'quality of life' (QoL) (Paediatric Quality of Life Inventory 4.0). Feasibility and adherence were self-rated on a scale of 1 to 10 (low to high). Linear mixed modelling was used to assess change over time, compare strategy effectiveness and study the impact of intervention order. RESULTS: Fatigue severity, self-efficacy and QoL regarding 'physical' and 'emotional' functioning improved significantly over time (all p < .015). The average improvement of the two QoL subscales was clinically relevant, as was the fatigue improvement in 20 out of 46 participants who completed the trial and 5 dropouts. The interventions were equally effective, and intervention order did not impact the improvement level (prange = .242-.984). The self-management strategies received similar feasibility (M = 6.45, SD = 1.91) and adherence (M = 7.67, SD = 1.67) ratings. CONCLUSIONS: As small to clinically relevant improvements were observed, self-management strategies might be particularly useful to bridge waiting time for guided treatments such as Cognitive Behavioural Therapy.

How to support children to develop and express their coping preferences around minor invasive medical procedures: children's and parents' perspectives.

Invasive medical procedures in hospitals are major sources of stress in children, causing pain and fear. Non-pharmacological interventions are indispensable in effective pain and fear management. However, these interventions must be personalized to be effective. This qualitative study aims to gain insight into children's and parents' experiences, needs, and wishes related to supporting children to develop and express their coping preferences for dealing with pain and fear during minor invasive medical procedures in order to decrease pain and fear. A qualitative study using thematic analysis was performed. Data were collected through semi-structured interviews with children and parents who had undergone at least five minor invasive medical procedures in the last year. Nineteen children (8-18 years) and fourteen parents were interviewed individually. The experiences, needs, and wishes expressed in the interviews could be classified into one overarching theme, that of the personal process, and two content-related sub-themes: feeling trust and gaining control. The personal process was divided into two different phases, that of developing and of expressing coping preferences. Children and parents both reported it as a continuous process, different for every child, with their own unique needs. Children and parents expected personalized attention and tailored support from professionals.     Conclusion: Professionals must combine clinical skills with child-tailored care. In the process of searching for and communicating about coping preferences, children's unique needs and personal boundaries will thereby be respected. This gives children and parents increased trust and control during invasive medical procedures. What is Known: • Untreated pain and stress caused by medical procedures can have severe and important short- and long-term consequences for children. Personalized non-pharmacological interventions are an essential element of procedural pain management. What is New: • A personalized coping strategy is important for children when undergoing medical procedures. Each individual child has a personal way of expressing their own coping strategy. Children and their parents need information and the space to develop and express their individual coping preferences. • Children and parents expect to receive child-tailored care from professionals including respect for their own, unique needs and boundaries. Professionals should build trustful relationships and provide appropriately tailored autonomy around medical procedures.

Towards a more comprehensive understanding of PTSD and parenting.

BACKGROUND: The impact of post-traumatic stress disorder (PTSD) on parenting and the parent-child relationship has been well-documented in the scientific literature. However, some conceptual and methodological challenges within this research field remain. PROCEDURE: We reflect on a number of challenges that we identified while examining the literature in preparation of an individual participant data meta-analysis on the relationships between PTSD and parenting. FINDINGS: We address 1) the presence of 'trauma-islands'; 2) the need for transdiagnostic theoretical frameworks for mechanisms between PTSD and parenting; 3) the lack of developmental perspectives; 4) the overuse of self-reported retrospective measures; 5) the need to study more diverse samples and cultural contexts; and 6) the lack of research on resilience and post-traumatic growth in parenting. Based on these reflections, we offer suggestions on strategies for responding to these challenges through: 1) welcoming open science; 2) working towards shared theoretical frameworks; 3) doing more longitudinal research 4) expanding the methodological palette; 5) centering lived experience; and 6) taking systemic inequality into account. CONCLUSION: With this commentary, we aim to open a discussion on next steps towards a more comprehensive understanding of the association between PTSD and parenting, and inspire collaborative research.

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.

Dynamic modeling of experience sampling methodology data reveals large heterogeneity in biopsychosocial factors associated with persistent fatigue in young people living with a chronic condition.

OBJECTIVE: To evaluate associations between self-reported biopsychosocial factors and persistent fatigue with dynamic single-case networks. METHODS: 31 persistently fatigued adolescents and young adults with various chronic conditions (aged 12 to 29 years) completed 28 days of Experience Sampling Methodology (ESM) with five prompts per day. ESM surveys consisted of eight generic and up to seven personalized biopsychosocial factors. Residual Dynamic Structural Equation Modeling (RDSEM) was used to analyze the data and derive dynamic single-case networks, controlling for circadian cycle effects, weekend effects, and low-frequency trends. Networks included contemporaneous and cross-lagged associations between biopsychosocial factors and fatigue. Network associations were selected for evaluation if both significant (α < 0.025) and relevant (ß ≥ 0.20). RESULTS: Participants chose 42 different biopsychosocial factors as personalized ESM items. In total, 154 fatigue associations with biopsychosocial factors were found. Most associations were contemporaneous (67.5%). Between chronic condition groups, no significant differences were observed in the associations. There were large inter-individual differences in which biopsychosocial factors were associated with fatigue. Contemporaneous and cross-lagged associations with fatigue varied widely in direction and strength. CONCLUSIONS: The heterogeneity found in biopsychosocial factors associated with fatigue underlines that persistent fatigue stems from a complex interplay between biopsychosocial factors. The present findings support the need for personalized treatment of persistent fatigue. Discussing the dynamic networks with the participant can be a promising step towards tailored treatment. TRIAL REGISTRATION: No. NL8789 (http://www.trialregister.nl).

Paediatric short fatigue questionnaire, a 4-item fatigue questionnaire for children.

OBJECTIVE: To investigate whether a paediatric Short Fatigue Questionnaire (pSFQ) assesses a valid construct for subjective fatigue, to assess its psychometric properties and provide a cut-off score for severe fatigue in children. METHODS: The pSFQ consists of 4 items from the Checklist Individual Strength-8 (CIS-8). Data of previous studies using the CIS-8 were used to assess the pSFQ in healthy children (n = 316), children with chronic fatigue syndrome (n = 173), and children with a chronic disease (n = 442). All children were 12-18 years old. Confirmatory factor analysis (CFA) was performed, followed by Cronbach alpha's to investigate internal consistency, and Spearman's correlations to assess construct validity. With ROC analysis, we determined a cut-off score for severe fatigue and provide normative data on the pSFQ for children with and without a chronic disease. RESULTS: CFA confirmed a one-factor model in the pSFQ representing subjective fatigue. Cronbach's alpha ranged from good to excellent (0.84-0.94), as did construct validity (-0.76 and - 0.87 for correlation with two other fatigue measurements). ROC analysis delivered a cut-off score of ≥21, with a sensitivity of 93.9% and specificity of 96.2% for severe fatigue. Normative data for children with and without a chronic disease showed similar patterns compared to other fatigue questionnaires. CONCLUSION: The pSFQ is a practical and reliable screening instrument for severe fatigue in children with and without a chronic disease, and effectively reduces the questionnaire length with 50% compared to the conventional CIS-8.

Development of the My Positive Health dialogue tool for children: a qualitative study on children's views of health.

BACKGROUND: Children's views of health were explored in order to develop a health dialogue tool for children. METHODS: A qualitative research design was used as part of a codesign process. Based on semi-structured interviews with both healthy children and children with a chronic condition (aged 8-18). Two approaches were applied. The first was an open exploration of children's views on health, which was then thematically analysed. Subsequently, a framework was used, based on the six-dimensional My Positive Health (MPH) dialogue tool for adults, to guide the second part of the interviews, focusing on reviewing the children's view on health within the context of the framework. For the final draft of the dialogue tool, a framework analysis was conducted and then validated by members of the 'children's council' of the Wilhelmina Children's Hospital. RESULTS: We interviewed 65 children, 45 of whom had a chronic condition and 20 were healthy. The children described a broad concept of health with the central themes of 'feeling good about yourself' and 'being able to participate'. Based on the subsequent framework analysis, the wording of two of the six dimensions of the MPH dialogue tool was adjusted and the related aspects were adapted for better alignment with the children's concept of health. After these modifications, the tool fully matched the children's concept of health. CONCLUSION: The MPH dialogue tool for children was developed for children with and without a chronic condition, to help them open up about what they consider important for their health and well-being, and to improve directorship over decisions and actions that would affect their health. The MPH dialogue tool aims to support healthcare professionals in providing the type of care and treatment that is in line with the needs of their young patients/clients.

Identifying disrupted biological factors and patient-tailored interventions for chronic fatigue in adolescents and young adults with Q-Fever Fatigue Syndrome, Chronic Fatigue Syndrome and Juvenile Idiopathic Arthritis (QFS-study): study protocol for a randomized controlled trial with single-subject experimental case series design.

BACKGROUND: Chronic fatigue with a debilitating effect on daily life is a frequently reported symptom among adolescents and young adults with a history of Q-fever infection (QFS). Persisting fatigue after infection may have a biological origin with psychological and social factors contributing to the disease phenotype. This is consistent with the biopsychosocial framework, which considers fatigue to be the result of a complex interaction between biological, psychological, and social factors. In line, similar manifestations of chronic fatigue are observed in chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) and juvenile idiopathic arthritis (JIA). Cognitive behavioral therapy is often recommended as treatment for chronic fatigue, considering its effectiveness on the group level. However, not everybody benefits on the individual level. More treatment success at the individual level might be achieved with patient-tailored treatments that incorporate the biopsychosocial framework. METHODS: In addition to biological assessments of blood, stool, saliva, and hair, the QFS-study consists of a randomized controlled trial (RCT) in which a single-subject experimental case series (N=1) design will be implemented using Experience Sampling Methodology in fatigued adolescents and young adults with QFS, CFS/ME, and JIA (aged 12-29). With the RCT design, the effectiveness of patient-tailored PROfeel lifestyle advices will be compared against generic dietary advices in reducing fatigue severity at the group level. Pre-post analyses will be conducted to determine relevance of intervention order. By means of the N=1 design, effectiveness of both advices will be measured at the individual level. DISCUSSION: The QFS-study is a comprehensive study exploring disrupted biological factors and patient-tailored lifestyle advices as intervention in adolescent and young adults with QFS and similar manifestations of chronic fatigue. Practical or operational issues are expected during the study, but can be overcome through innovative study design, statistical approaches, and recruitment strategies. Ultimately, the study aims to contribute to biological research and (personalized) treatment in QFS and similar manifestations of chronic fatigue. TRIAL REGISTRATION: Trial NL8789 . Registered July 21, 2020.

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.

The Impact of the COVID-19 Outbreak on Mental Wellbeing in Children with a Chronic Condition Compared to Healthy Peers.

The aim of this study was to assess the impact of the COVID-19 pandemic on the mental wellbeing of children 8-18 years old with chronic conditions, by comparing pandemic data with pre-pandemic data and with healthy peers. Data were obtained from two ongoing longitudinal cohorts: the PROactive cohort study following children with a chronic condition, and the WHISTLER population cohort. Mental wellbeing was assessed by three indicators: life satisfaction, internalising symptoms, and psychosomatic health. The stringency of the COVID-19-related lockdown was considered a moderating factor. Data on chronic patients were recorded before (n = 934, 65% girls) and during (n = 503, 61% girls) the pandemic, and compared to healthy peers during the pandemic (n = 166, 61% girls). Children with a chronic condition reported lower life satisfaction, but no clinically relevant changes in internalising symptoms or psychosomatic health, during the pandemic compared to before. In comparison to healthy peers, children with a chronic condition experienced decreased life satisfaction and psychosomatic health, but internalising symptoms did not differ between groups during the COVID-19 pandemic. The lockdown stringency was negatively associated with all indicators of mental wellbeing-worse life satisfaction, more internalising symptoms, and more psychosomatic symptoms.

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.

Exploring key elements of approaches that support childrens' preferences during painful and stressful medical procedures: A scoping review.

PROBLEM: Children undergoing medical procedures can experience pain and distress. While numerous interventions exist to mitigate pain and distress, the ability to individualize the intervention to suit the needs and preferences of individual children is emerging as an important aspect of providing family-centered care and shared decision making. To date, the approaches for supporting children to express their preferences have not been systematically identified and described. A scoping review was conducted to identify such approaches and to describe the elements that are included in them. ELIGIBILITY CRITERIA: Studies that (a) described approaches with the aim to support children to express their coping preferences during medical procedures; (b) included the option for children to choose coping interventions; (c) included a child (1--18 years). SAMPLE: Searches were conducted in December 2019 and November 2020 in the following databases: Cinahl, Embase, PubMed and Psycinfo. RESULTS: Thirteen studies were identified that included six distinct approaches. Four important key elements were identified: 1) Aid to express preferences or choice, 2) Information Provision, 3) Assessment of feelings/emotions, 4) Feedback/Reflection and Reward. CONCLUSIONS: Identified approaches incorporate components of shared decision-making to support children in expressing their preferences during medical procedures and treatments. IMPLICATIONS: Children undergoing medical procedures can be supported in expressing their coping needs and preferences by using components of shared decision-making.

Parent-Child Dyadic Coping and Quality of Life in Chronically Diseased Children.

Different forms of dyadic coping are associated with positive outcomes in partner relationships, yet little is known about dyadic coping in parent-child relationships. The current research explored the association between parent-child dyadic coping and children's quality of life in 12-18-year old children with a chronic disease (i.e., cystic fibrosis, autoimmune diseases, and children post-cancer treatment). In a sample of 105 parent-child dyads, self-reported forms of dyadic coping (i.e., stress communication, problem-oriented, emotion-oriented, and negative dyadic coping) and children's quality of life were assessed. Children reported more stress communication and negative dyadic coping than their parents, while parents reported more problem-oriented dyadic coping and emotion-oriented dyadic coping than their children. More stress communication of the child was associated with more emotion-oriented dyadic coping and less negative dyadic coping of the parent. More negative dyadic coping of the child was associated with less stress communication, problem-oriented dyadic coping and emotion-oriented dyadic coping of the parent. Additionally, both children's and parents' negative dyadic coping were associated with lower self-reported pediatric quality of life and parents' emotion-oriented dyadic coping was associated with higher pediatric quality of life. These findings emphasize that children and their parents mutually influence each other and that dyadic coping is associated with children's quality of life. Theoretical and practical implications are discussed.

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.

Role of parents in fatigue of children with a chronic disease: a cross-sectional study.

Objective: As parents majorly impact their child's well-being, and as fatigue is a highly prevalent threat to the well-being of children with a chronic disease, we aimed to explore the association between parental factors and fatigue in children with a chronic disease. Design: Cross-sectional study. Setting: Two Dutch children's hospitals. Population: Children 2-18 years of age with either an autoimmune disease, cystic fibrosis or post-cancer treatment, and one of their parents. Main outcome measures: Paediatric fatigue was measured using the PedsQL Multidimensional Fatigue Scale. Parental factors included parental pain, fatigue and physical symptoms, parental distress, catastrophising thoughts about their child's pain and family empowerment. Multiple linear regressions were used to study associations with paediatric fatigue. A multivariable regression model was used to assess the effect of the different parental factors on paediatric fatigue. All analyses were adjusted for the age and sex of the child. Results: 204 families participated (mean age 11.0±4.3 and 43.5±6.3 years for children and parents, respectively; 69% participation rate). More parental pain, fatigue and physical symptoms, and more parental distress and pain catastrophising were associated with more paediatric fatigue. More parental empowerment was associated with less paediatric fatigue on both subscales. In the multivariable model, only paediatric age remained significantly associated with fatigue. In a separate multivariable model for children 8-18 years old, more parental distress (ß=-1.9, 95% CI -3.7 to -0.1) was also significantly associated with more paediatric fatigue. Conclusions: In a population of children with a chronic disease, parental factors, both physical and psychosocial, were associated with paediatric fatigue. Our study provides evidence that more family empowerment is associated with less paediatric fatigue. This exploratory study adds to our knowledge of associated factors with fatigue in paediatric chronic disease, providing starting points for targeted interventions.

Daily life participation in childhood chronic disease: a qualitative study on the child's and parent's perspective.

Objective: To understand how a child with a stable chronic disease and his/her parents shape his/her daily life participation, we assessed: (1) the parents' goals regarding the child's daily life participation, (2) parental strategies regarding the child's participation and () how children and their parents interrelate when their goals regarding participation are not aligned. Methods: This was a qualitative study design using a general inductive approach. Families of children 8-19 years with a stable chronic disease (cystic fibrosis, autoimmune disease or postcancer treatment) were recruited from the PROactive study. Simultaneous in-depth interviews were conducted separately with the child and parent(s). Analyses included constant comparison, coding and categorisation. Results: Thirty-one of the 57 invited families (54%) participated. We found that parents predominantly focus on securing their child's well-being, using participation as a means to achieve well-being. Moreover, parents used different strategies to either support participation consistent with the child's healthy peers or support participation with a focus on physical well-being. The degree of friction between parents and their child was based on the level of agreement on who takes the lead regarding the child's participation. Conclusions: Interestingly, parents described participation as primarily a means to achieve the child's well-being, whereas children described participation as more of a goal in itself. Understanding the child's and parent's perspective can help children, parents and healthcare professionals start a dialogue on participation and establish mutual goals. This may help parents and children find ways to interrelate while allowing the child to develop his/her autonomy.

Internet and smartphone-based ecological momentary assessment and personalized advice (PROfeel) in adolescents with chronic conditions: A feasibility study.

OBJECTIVE: Growing up with a chronic disease comes with challenges, such as coping with fatigue. Many adolescents are severely fatigued, though its associated factors exhibit considerable interpersonal and longitudinal variation. We assessed whether PROfeel, a combination of a smartphone-based ecological momentary assessment (EMA) method using the internet, followed by a face-to-face dialogue and personalized advice for improvement of symptoms or tailor treatment based on a dynamic network analysis report, was feasible and useful. STUDY DESIGN: Feasibility study in fatigued outpatient adolescents 12-18 years of age with cystic fibrosis, autoimmune disease, post-cancer treatment, or with medically unexplained fatigue. Participants were assessed at baseline to personalize EMA questions. EMA was conducted via smartphone notifications five times per day for approximately six weeks. Hereby, data was collected via the internet. The EMA results were translated into a personalized report, discussed with the participant, and subsequently translated into a personalized advice. Afterwards, semi-structured interviews on feasibility and usefulness were held. RESULTS: Fifty-seven adolescents were assessed (mean age 16.2 y ± 1.6, 16% male). Adolescents deemed the smartphone-based EMA feasible, with the app being used for an average of 49 days. Forty-two percent of the notifications were answered and 85% of the participants would recommend the app to other adolescents. The personalized report was deemed useful and comprehensible and 95% recognized themselves in the personalized report, with 64% rating improved insight in their symptoms and subsequent steps towards an approach to reduce one's fatigue as good or very good. CONCLUSIONS: PROfeel was found to be highly feasible and useful for fatigued adolescents with a chronic condition. This innovative method has clinical relevance through bringing a patient's daily life into the clinical conversation.