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

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

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

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

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

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 (β ≥ 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

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

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

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

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