Kate Lievesley

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.

Development and psychometric properties of the Suicidality: Treatment Occurring in Paediatrics (STOP) Suicidality Assessment Scale (STOP-SAS) in children and adolescents

BackgroundTo create a self-reported, internet-based questionnaire for the assessment of suicide risk in children and adolescents.MethodsAs part of the EU project ‘Suicidality: Treatment Occurring in Paediatrics’ (STOP project), we developed web-based Patient Reported Outcome Measures (PROMs) for children and adolescents and for proxy reports by parents and clinicians in order to assess suicidality. Based on a literature review, expert panels and focus groups of patients, we developed the items of the STOP Suicidality Assessment Scale (STOP-SAS) in Spanish and English, translated it into four more languages, and optimized it for web-based presentation using the HealthTrackerTM platform. Of the total 19 questions developed for the STOP-SAS, four questions that assess low-level suicidality were identified as screening questions (three of them for use with children, and all four for use with adolescents, parents and clinicians). A total of 395 adolescents, 110 children, 637 parents and 716 clinicians completed the questionnaire using the HealthTrackerTM, allowing us to evaluate the internal consistency and convergent validity of the STOP-SAS with the clinician-rated Columbia Suicide Severity Rating Scale (C-SSRS). Validity was also assessed with the receiver operating characteristic (ROC) area of the STOP-SAS with the C-SSRS.ResultsThe STOP-SAS comprises 19 items in its adolescent, parent, and clinician versions, and 14 items in its children’s version. Good internal consistency was found for adolescents (Cronbach’s alpha: 0.965), children (Cronbach’s alpha: 0.922), parents (Cronbach’s alpha: 0.951) and clinicians (Cronbach’s alpha: 0.955) versions. A strong correlation was found between the STOP-SAS and the C-SSRS for adolescents (r:0.670), parents (r:0.548), clinicians (r:0.863) and children (r:0.654). The ROC area was good for clinicians’ (0.917), adolescents’ (0.834) and parents’ (0.756) versions but only fair (0.683) for children’s version.ConclusionsThe STOP-SAS is a comprehensive, web-based PROM developed on the HealthTrackerTM platform, and co-designed for use by adolescents, children, parents and clinicians. It allows the evaluation of aspects of suicidality and shows good reliability and validity.

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.

Comparing the DSM-5 construct of Disruptive Mood Dysregulation Disorder and ICD-10 Mixed Disorder of Emotion and Conduct in the UK Longitudinal Assessment of Manic Symptoms (UK-LAMS) Study

It is important to understand new diagnostic entities in classifications of psychopathology such as the Diagnostic and Statistical Manual of Mental Disorders-5 (DSM-5) (code F34.8) construct of Disruptive Mood Dysregulation Disorder (DMDD) and to compare it with possible equivalent disorders in other classificatory systems such as the International Classification of Diseases-10 (ICD-10), which has a category that superficially appears similar, that is, Mixed Disorder of Emotion and Conduct (MDEC) (code F92). In this study, the United Kingdom (UK) arm (UK-LAMS) of the US National Institute of Mental Health (NIMH) supported Longitudinal Assessment of Manic Symptoms (LAMS) multi-site study was used to evaluate and retrospectively construct DMDD and MDEC diagnoses in order to compare them and understand the conditions they co-occur with, in order to improve the clinical understanding. In particular, the phenomenology of UK-LAMS participants (n = 117) was used to determine whether DMDD is a unique entity within the DSM-5. The findings showed that 24 of 68 participants with either DMDD or MDEC (35.3%) fulfilled both diagnostic criteria for DMDD and MDEC, suggesting that these entities do contain overlapping features, particularly symptoms relating to Oppositional Defiant Disorder (ODD)/Conduct Disorder (CD), Attention Deficit Hyperactivity Disorder (ADHD)/Hyperkinetic Disorder (HKD) and/or an anxiety disorder. The data also showed that most of the participants who met DMDD criteria also fulfilled the diagnostic criteria for ODD/CD, ADHD, followed by an anxiety disorder. In this context, this raises the issue whether DMDD is a unique construct or whether the symptomology for DMDD can be better explained as a specifier for ODD/CD and ADHD. Unlike DMDD, MDEC clearly specifies that the label should only be used if emotional and conduct disorders co-exist.

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).

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

Abstract 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.

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.