Does fatigue and distress in a clinical cohort of adolescents with chronic fatigue syndrome correlate with fatigue and distress in their parents?

OBJECTIVES: Previous studies have found that parents of children with chronic fatigue syndrome (CFS) are more fatigued, and mothers are more distressed than healthy controls. Managing the disabling symptoms of CFS can result in disruption and burden for the family. Most research has focused on mothers. This study sought to further explore the associations between adolescent fatigue and distress and parental fatigue and distress, as well as family functioning, including both mothers and fathers. DESIGN: Cross-sectional study of a clinical cohort of consecutive attenders at a specialist chronic fatigue unit. METHODS: Questionnaires were completed by adolescents (N = 115, age 11-18) with a confirmed diagnosis of CFS and their mothers (N = 100) and fathers (N = 65). RESULTS: Maternal fatigue was significantly correlated with maternal distress, but not with adolescent fatigue, depression, anxiety, or functioning. This pattern held true for paternal fatigue. Maternal and paternal anxiety and depression were significantly correlated with family functioning. Paternal and maternal distress were correlated with each other. Mothers and fathers tended to have a consistent view of family functioning. Family functioning, specifically being overwhelmed by difficulties and scoring lower on strengths and adaptability, was positively associated with adolescent depression. Unexpectedly, higher levels of adolescent fatigue and poorer physical functioning were associated with better family functioning as rated by the mother. CONCLUSIONS: Parents of adolescents with fatigue scored near to or within normative range for non-clinical samples on distress, fatigue, and family functioning. Parental distress may contribute to or result from poorer family functioning. Family functioning, particularly building strengths and adaptability, may be clinically important in CFS, as well as attending to parental (particularly paternal) distress in families where adolescents are low in mood.

A process evaluation of the first year of Leading Change, Adding Value.

BACKGROUND: Leading Change, Adding Value (LCAV) is a national framework to support transformational change across health and social care. DESIGN: a qualitative approach of semi-structured interviews was used to capture information on how LCAV has been disseminated in its early stages from the perspective of key stakeholders and partners. This also included looking at how it might be embedded into everyday practice. METHODS: data collection took place over a 3-month period between January and March 2017. Twenty semi-structured telephone interviews were conducted with key LCAV partners and stakeholders from across health and social care. Perceptions were sought as to how LCAV has been, and may be, used by frontline staff following initial dissemination and any potential barriers and enablers to taking the framework forward. RESULTS: a thematic framework analysis of data identified a three-theme paradigm to evaluate LCAV: past-where has this come from? Present-where is it now? Future-where is this going?. CONCLUSION: a programme of dissemination events and examples of good practice in the form of case studies have been valuable tools to engage nursing, midwifery and care staff across health and social care. Continuing to establish networks of frontline staff engaging with LCAV and supporting each other will help facilitate best practice sharing, and multi-professional and cross-boundary working.

Stress vulnerability in adolescents with chronic fatigue syndrome: experimental study investigating heart rate variability and skin conductance responses.

BACKGROUND: Stress vulnerability has been implicated in adolescent chronic fatigue syndrome (CFS), but has rarely been investigated directly. This study compared psychological and physiological responses to a laboratory social performance task in adolescents with CFS with chronic illness (asthma) and healthy control groups. METHODS: Adolescents with CFS (n = 60), adolescents with asthma (n = 31) and healthy adolescents (n = 78) completed questionnaires before and after a social performance task. Skin conductance responses (SCR; mean SCR and Max-Min) and heart rate variability (low frequency/high frequency; LF/HF and root mean square difference of successive RR intervals; RMSSD) was measured before, during and after the task. RESULTS: Baseline heart rate variability (HRV) (RMSSD) was significantly lower in the CFS and Asthma groups than the HC. During the speech, the CFS and Asthma groups had higher HRV (LF/HF) than the HC, adjusting for baseline LF/HF. Although the asthma group showed a subsequent reduction in HRV during recovery, the CFS group did not. Similarly, during recovery after the task, the CFS group showed a continued increase in skin conductance (Min-Max), unlike the Asthma and HC groups. Compared to control groups, adolescents with CFS expected to find the task more difficult, were more anxious beforehand and afterwards, rated it as more difficult, evaluated their performance more negatively and had lower observer ratings of performance. Parents of adolescents with CFS expected that their child would perform less well in the task than parents of control participants. CONCLUSIONS: Adolescents with CFS showed autonomic nervous system responses that are consistent with chronic stress vulnerability, difficulty coping with acute stress and slower recovery after acute stress. Self-report measures also indicated greater trait, pre- and posttask anxiety in the CFS group.

Suicidality Treatment Occurring in Paediatrics (STOP) Medication Suicidality Side Effects Scale in young people in two cohorts across Europe.

OBJECTIVES: As part of the 'Suicidality: Treatment Occurring in Paediatrics (STOP)' study, we developed and performed psychometric validation of an electronic-clinical-outcome-assessment (eCOA), which included a patient-reported-outcome (ePRO), an observer-rated-outcome (eObsRO) for parents/carers and a clinician-reported-outcome (eClinRO) that allows identification and monitoring of medication-related suicidality (MRS) in adolescents. DESIGN: STOP: Prospective study: A two phase validation study to assess the impact of medication on suicidal ideations. SETTING: Six participating countries: Netherlands, UK, Germany, France, Spain and Italy that were part of the Community's Seventh Framework Programme (FP7/2007-2013) under grant agreement no. 261411. PARTICIPANTS: Cohort 1 consisted of 41 adolescent-completions, 50 parent-completions and 56 clinician-completions. Cohort 2 consisted of 244 adolescent-completions, 198 parent-completions and 240 clinician-completions from across the six countries. The scale was administered only to participants who have screened positive for the STOP-Suicidality Assessment Scale (STOP-SAS). RESULTS: A total of 24 items for the development of the STOP-Medication Suicidality Side Effects Scale (STOP-MS3) were identified and three versions (for patients, parents and clinicians) of the STOP-MS3 were developed and validated in two separate study cohorts comprising of adolescents, their parents and clinicians. Cronbach's α coefficients were above 0.85 for all domains. The inter-rater reliability of the STOP-MS3 was good and significant for the adolescent (ePRO), clinician (eClinRO) (r=0.613), parent (eObsRO) versions of the scale (r=0.394) and parent and clinician (r=0.347). Exploratory factor analysis identified a 3-factor model across 24 items for the adolescent and parent version of the scale: (1) Emotional Dysregulation, (2) Somatic Dysregulation and (3) Behavioural Dysregulation. For the clinician version, a 4-factor model defined the scale structure: (1) Somatic Dysregulation, (2) Emotional Dysregulation, (3) Behavioural Dysregulation and (4) Mood Dysregulation. CONCLUSION: These findings suggest that the STOP-MS3 scale, a web-based eCOA, allows identification and monitoring of MRS in the adolescent population and shows good reliability and validity.

Cohort profile: demographic and clinical characteristics of the MILESTONE longitudinal cohort of young people approaching the upper age limit of their child mental health care service in Europe.

PURPOSE: The presence of distinct child and adolescent mental health services (CAMHS) and adult mental health services (AMHS) impacts continuity of mental health treatment for young people. However, we do not know the extent of discontinuity of care in Europe nor the effects of discontinuity on the mental health of young people. Current research is limited, as the majority of existing studies are retrospective, based on small samples or used non-standardised information from medical records. The MILESTONE prospective cohort study aims to examine associations between service use, mental health and other outcomes over 24 months, using information from self, parent and clinician reports. PARTICIPANTS: Seven hundred sixty-three young people from 39 CAMHS in 8 European countries, their parents and CAMHS clinicians who completed interviews and online questionnaires and were followed up for 2 years after reaching the upper age limit of the CAMHS they receive treatment at. FINDINGS TO DATE: This cohort profile describes the baseline characteristics of the MILESTONE cohort. The mental health of young people reaching the upper age limit of their CAMHS varied greatly in type and severity: 32.8% of young people reported clinical levels of self-reported problems and 18.6% were rated to be 'markedly ill', 'severely ill' or 'among the most extremely ill' by their clinician. Fifty-seven per cent of young people reported psychotropic medication use in the previous half year. FUTURE PLANS: Analysis of longitudinal data from the MILESTONE cohort will be used to assess relationships between the demographic and clinical characteristics of young people reaching the upper age limit of their CAMHS and the type of care the young person uses over the next 2 years, such as whether the young person transitions to AMHS. At 2 years follow-up, the mental health outcomes of young people following different care pathways will be compared. TRIAL REGISTRATION NUMBER: NCT03013595.

Validation of the Transition Readiness and Appropriateness Measure (TRAM) for the Managing the Link and Strengthening Transition from Child to Adult Mental Healthcare in Europe (MILESTONE) study.

OBJECTIVE: Young people moving from child and adolescent mental health services (CAMHS) to adult mental health services (AMHS) are faced with significant challenges. To improve this state of affairs, there needs to be a recognition of the problem and initiatives and an urgent requirement for appropriate tools for measuring readiness and outcomes at the transfer boundary (16-18 years of age in Europe). The objective of this study was to develop and validate the Transition Readiness and Appropriateness Measure (TRAM) for assessing a young person's readiness for transition, and their outcomes at the transfer boundary. DESIGN: MILESTONE prospective study. SETTING: Eight European Union (EU) countries participating in the EU-funded MILESTONE study. PARTICIPANTS: The first phase (MILESTONE validation study) involved 100 adolescents (pre-transition), young adults (post-transition), parents/carers and both CAMHS and AMHS clinicians. The second phase (MILESTONE cohort study and nested cluster randomised trial) involved over 1000 young people. RESULTS: The development of the TRAM began with a literature review on transitioning and a review of important items regarding transition by a panel of 34 mental health experts. A list of 64 items of potential importance were identified, which together comprised the TRAM. The psychometric properties of the different versions of the TRAM were evaluated and showed that the TRAM had good reliability for all versions and low-to-moderate correlations when compared with other established instruments and a well-defined factor structure. The main results of the cohort study with the nested cluster randomised trial are not reported. CONCLUSION: The TRAM is a reliable instrument for assessing transition readiness and appropriateness. It highlighted the barriers to a successful transition and informed clinicians, identifying areas which clinicians on both sides of the transfer boundary can work on to ease the transition for the young person. TRIAL REGISTRATION NUMBER: ISRCTN83240263 (Registered 23 July 2015), NCT03013595 (Registered 6 January 2017); Pre-results.

Home-based family focused rehabilitation for adolescents with severe Chronic Fatigue Syndrome.

AIMS:: The purpose of this article is to describe and evaluate a home based, family focused rehabilitative approach for severely affected housebound adolescents with Chronic Fatigue Syndrome (CFS). The main aims were to facilitate a return to school, improve physical functioning, reduce fatigue and assess any adverse effects of the intervention. METHODS:: Six housebound adolescents aged 11-18, diagnosed with CFS by a paediatrician, were assessed and treated at home by an experienced cognitive behaviour therapist. Outcomes were assessed 12 months after discharge from treatment. RESULTS:: At 12 months follow-up all patients had returned to either school or college, and physical functioning had improved in most of the patients. Fatigue had reduced in some. No adverse effects of the intervention were reported. CONCLUSION:: Severely affected adolescents with CFS showed improved physical functioning and social adjustment after a home-based rehabilitative approach. Although several patients showed improvements in physical functioning, they did not all show substantial improvements in fatigue. At this crucial stage of development, it is important to offer young people and their parents hope by stating that improvement is possible.

Cognitive and behavioural responses to symptoms in adolescents with chronic fatigue syndrome: A case-control study nested within a cohort.

BACKGROUND: What adolescents think about symptoms and what they do in response could contribute to fatigue maintenance. We compared the cognitive and behavioural responses of adolescents and their parents with chronic fatigue syndrome (CFS; N = 121) and asthma (N = 27) and explored the predictive value of these variables on fatigue and functioning in CFS. METHOD: Consecutively referred adolescents with CFS were recruited. Questionnaires, completed by adolescents and parents, assessed fatigue, functioning, mood and cognitive and behavioural responses to symptoms. Age-matched adolescents with asthma completed the same questionnaires. Adolescents with CFS completed questionnaires again approximately 3 months later. RESULTS: Adolescents with CFS scored higher on all unhelpful cognitive and behavioural subscales than adolescents with asthma. Parents' cognitions about their child's symptoms were associated with adolescent's own cognitions. Unhelpful cognitive and behavioural responses, particularly, damage beliefs, predicted subsequent fatigue in CFS, and all-or-nothing behaviour, catastrophising and damage beliefs predicted subsequent physical functioning. CONCLUSION: Unhelpful cognitive and behavioural responses to symptoms appear to be particularly prominent in adolescents with CFS. There is some consistency but not a perfect match between cognitive and behavioural responses to symptoms reported by adolescents and their parents. These responses could be contributing to fatigue maintenance and disability.

Perfectionism and beliefs about emotions in adolescents with chronic fatigue syndrome and their parents: a preliminary investigation in a case control study nested within a cohort.

Objectives: To investigate perfectionism and beliefs about emotions in adolescents with chronic fatigue syndrome (CFS) and their parents. Design: Case-control comparing adolescents (age 11-18) with CFS (N = 121), asthma (N = 27) and healthy controls (N = 78) with a 3-month follow up for CFS participants. Main outcome measures: Adolescents: Chalder Fatigue Questionnaire, physical functioning, Beliefs about Emotions scale (BES), Child and Adolescent Perfectionism Scale, Frost Multidimensional Perfectionism Scale (FMPS). Parents: BES, FMPS, Self-sacrificing scale, Affective styles questionnaire. Results: Adolescents with CFS did not consistently report higher levels of perfectionism and unhelpful beliefs about emotions than adolescents with asthma or healthy adolescents. Mothers' and adolescents' beliefs about emotions and unhelpful perfectionism were significantly associated (p = .007). Linear regression found that neither adolescent perfectionism nor beliefs about emotions accounted for variance in subsequent fatigue or physical functioning. Conclusion: Parental perfectionism and emotion regulation style may contribute to perfectionism in adolescents with CFS. Parental representations could contribute to fatigue maintenance.

Propranolol for treating emotional, behavioural, autonomic dysregulation in children and adolescents with autism spectrum disorders.

OBJECTIVES: To date, there is no single medication prescribed to alleviate all the core symptoms of Autism Spectrum Disorder (ASD; National Institute of Health and Care Excellence, 2016). Both serotonin reuptake inhibitors and drugs for psychosis possess therapeutic drawbacks when managing anxiety and aggression in ASD. This review sought to appraise the use of propranolol as a pharmacological alternative when managing emotional, behavioural and autonomic dysregulation (EBAD) and other symptoms. MATERIALS AND METHODS: Sixteen reports examined the administration of propranolol in the context of ASD. RESULTS: Sixteen reports broadly covered cognitive domains, neural correlates, and behavioural domains. From the eight single-dose clinical trials, propranolol led to significant improvements in cognitive performance - verbal problem solving, social skills, mouth fixation, and conversation reciprocity; and changes in neural correlates - improvement in semantic networks and functional connectivity. The remaining eight case series and single case reports showed improvements in EBAD, anxiety, aggressive, self-injurious and hypersexual behaviours. Additionally, propranolol significantly improved similar behavioural domains (aggression and self-injury) for those with acquired brain injury. CONCLUSION: This review indicates that propranolol holds promise for EBAD and cognitive performance in ASD. Given the lack of good quality clinical trials, randomised controlled trials are warranted to explore the efficacy of propranolol in managing EBAD in ASD.

Illness beliefs of adolescents with CFS and their parents: the perceived causes of illness and beliefs about recovery.

The objective here was to explore beliefs about the causes of chronic fatigue syndrome (CFS) in a cohort of adolescents with CFS and their parents, and to explore the adolescent's beliefs about recovery. Questionnaires were administered to a clinical cohort of adolescents (n = 104) and their parents (n = 102 mothers and 63 fathers), presenting to a specialist CFS unit. These included a question about the causes of their illness. Adolescents were also asked about the projected timeline of their recovery from CFS. The most commonly endorsed causes of CFS by adolescents and their parents were a virus and/or contextual factors and stress. Adolescents and their parents were in close agreement about the causes of CFS. Most adolescents said they did not know how long it would take them to recover from CFS. Informing adolescents about the prognosis for CFS is an important aspect of treatment.

The presence of co-morbid mental health problems in a cohort of adolescents with chronic fatigue syndrome.

OBJECTIVE: To report on the prevalence of mental health disorders in adolescents with chronic fatigue syndrome (CFS) and to compare the diagnoses identified by a brief clinician-administered psychiatric interview with self-report screening questionnaires. DESIGN: Cross-sectional study. SETTING: Consecutive attenders to specialist CFS clinics in the United Kingdom. PATIENTS: N = 52 adolescents, age 12-18 years with CFS. MEASURES: Self-report questionnaires and a brief structured psychiatric diagnostic interview, administered by a researcher. RESULTS: On the psychiatric interview, 34.6% met a diagnosis of major depressive disorder and 28.8% had an anxiety disorder. Of these, 15% had co-morbid anxiety and depression. Those with a depression diagnosis reported significantly greater interference on the school and social adjustment scale. They also scored significantly higher on trait anxiety, but not on state anxiety. There were no differences between those who had an anxiety disorder and those who did not on fatigue, disability or depressive symptoms. Children's Depression Inventory (CDI) score was associated with a depression diagnosis on the psychiatric interview. However, neither the state nor the trait subscale of the State-Trait Anxiety Inventory (STAI) was associated with an anxiety diagnosis. CONCLUSION: Clinicians should assess for the presence of anxiety and depressive disorders in adolescents with CFS using a validated psychiatric interview. Treatment should be flexible enough to accommodate fatigue, depression and anxiety. Transdiagnostic approaches may suit this purpose. Goals should include pleasurable activities particularly for those who are depressed.

Development of the Tailored Rett Intervention and Assessment Longitudinal (TRIAL) database and the Rett Evaluation of Symptoms and Treatments (REST) Questionnaire.

INTRODUCTION: Rett syndrome (RTT) is a pervasive neurodevelopmental disorder that presents with deficits in brain functioning leading to language and learning regression, characteristic hand stereotypies and developmental delay. Different mutations in the gene implicated in RTT-methyl-CpG-binding protein 2 (MECP2) establishes RTT as a disorder with divergent symptomatology ranging from individuals with severe to milder phenotypes. A reliable and single multidimensional questionnaire is needed that can embrace all symptoms, and the relationships between them, and can map clinically meaningful data to symptomatology across the lifespan in patients with RTT. As part of the HealthTracker-based Tailored Rett Intervention and Assessment Longitudinal (TRIAL) database, the Rett Evaluation of Symptoms and Treatments (REST) Questionnaire will be able to marry with the physiological aspects of the disease obtained using wearable sensor technology, along with genetic and psychosocial data to stratify patients. Taken together, the web-based TRIAL database will empower clinicians and researchers with the confidence to delineate between different aspects of disorder symptomatology to streamline care pathways for individuals or for those patients entering clinical trials. This protocol describes the anticipated development of the REST questionnaire and the TRIAL database which links with the outcomes of the wearable sensor technology, and will serve as a barometer for longitudinal patient monitoring in patients with RTT. METHODS AND ANALYSIS: The US Food and Drug Administration Guidance for Patient-Reported Outcome Measures will be used as a template to inform the methodology of the study. It will follow an iterative framework that will include item/concept identification, item/concept elicitation in parent/carer-mediated focus groups, expert clinician feedback, web-based presentation of questionnaires, initial scale development, instrument refinement and instrument validation. ETHICS AND DISSEMINATION: The study has received favourable opinion from the National Health Service (NHS) Research Ethics Committee (REC): NHS Research Ethics Committee (REC)-London, Bromley Research Ethics Committee (reference: 15/LO/1772).

A review of the predisposing, precipitating and perpetuating factors in Chronic Fatigue Syndrome in children and adolescents.

Chronic Fatigue Syndrome (CFS) is a condition characterised by severe mental and physical fatigue coupled with profound disability. The purpose of this review was to investigate psychological, social and physiological factors associated with fatigue and disability in CFS in children and adolescents. The review aimed to gain an overview of the strength of evidence for the relationship between these different factors and CFS in young people. Seventy-nine studies met the inclusion criteria and were included in the review. A narrative synthesis of these studies was conducted. The strongest and most consistent finding was that rates of psychiatric co-morbidity, predominantly anxiety and depressive disorders, were higher in young people with CFS compared to healthy controls or illness control groups. Studies suggested that many children and adolescents with CFS reported that their illness began with an infection and there was some objective and prospective evidence to support this. Preliminary evidence suggested a link between CFS and a family history of CFS, high expectations from both the parent and child, personality traits such as conscientiousness and physical illness attributions. The evidence was limited by methodological problems. Few studies were prospective in nature and future research should address this. Clinical implications of the findings are discussed and a hypothesised model of the factors associated with CFS in children and adolescents is presented.