An observational study of individual child journeys through autism diagnostic pathways, and associated costs, in the UK National Health Service.

Background: Demand for diagnostic assessment in children with possible autism has recently increased significantly. Services are under pressure to deliver timely and high-quality diagnosis, following National Institute and Care Excellence multidisciplinary assessment guidelines. This UK National Health Service study aimed to answer: how many hours of health professional time are required to deliver autism diagnostic assessment, and how much does this cost?. Method: Case notes of 20 children (1-16 yrs.) from 27 NHS trusts, assessed through an autism diagnostic pathway in the previous year, were examined retrospectively. Data included: hours of professional time, diagnostic outcome. Assessment costs calculated using standardised NHS tariffs. Results: 488 children (aged 21-195 months, mean 82.9 months, SD 39.36) from 22 Child Development Services (CDS), four Child and Adolescent Mental Health Services (CAMHS) and one tertiary centre; 87% were either under 5 (36%) or 5 to 11 years (51%). Children seen by CDS were younger than CAMHS (mean (SD) 6.10 (2.72) vs. 10.39 (2.97) years, p < 0.001). Mean days to diagnosis were 375 (SD 235), with large variation (range 41-1553 days). Mean hours of professional time per child was 11.50 (SD 7.03) and varied substantially between services and individuals. Mean cost of assessment was £846.00 (SD 536.31). 339 (70.0%) children received autism diagnosis with or without comorbidity; 54 (11%) received no neurodevelopmental diagnosis; 91 (19%) received alternative neurodevelopmental diagnoses. Children with one or more coexisting conditions took longer to diagnose, and assessment was more costly, on average 117 days longer, costing £180 more than a child with no neurodevelopmental diagnosis. Age did not predict days to diagnosis or assessment costs. Conclusion: Typical assessment took 11 h of professional time and over 12-months to complete, costing GB£850 per child. Variation between centres and children reflect differences in practice and complexity of diagnostic presentation. These results give information to those delivering/planning autism assessments using multi-disciplinary team approach, in publicly funded health systems. Planning of future diagnostic services needs to consider growing demand, the need for streamlining, enabling context appropriate services, and child/family complexity.

A clinical and cost-effectiveness trial of a parent group intervention to manage challenging restricted and repetitive behaviours in young children with autism spectrum disorder: study protocol for a randomised controlled trial.

BACKGROUND: Restricted and repetitive behaviours vary greatly across the autism spectrum, and although not all are problematic some can cause distress and interfere with learning and social opportunities. We have, alongside parents, developed a parent group based intervention for families of young children with autism, which aims to offer support to parents and carers; helping them to recognise, understand and learn how to respond to their child's challenging restricted repetitive behaviours. METHODS: The study is a clinical and cost-effectiveness, multi-site randomised controlled trial of the Managing Repetitive Behaviours (MRB) parent group intervention versus a psychoeducation parent group Learning About Autism (LAA) (n = 250; 125 intervention/125 psychoeducation; ~ 83/site) for parents of young children aged 3-9 years 11 months with a diagnosis of autism. All analyses will be done under intention-to-treat principle. The primary outcome at 24 weeks will use generalised estimating equation (GEE) to compare proportion of children with improved RRB between the MRB group and the LAA group. The GEE model will account for the clustering of children by parent groups using exchangeable working correlation. All secondary outcomes will be analysed in a similar way using appropriate distribution and link function. The economic evaluation will be conducted from the perspective of both NHS costs and family access to local community services. A 'within trial' cost-effectiveness analysis with results reported as the incremental cost per additional child achieving at least the target improvement in CGI-I scale at 24 weeks. DISCUSSION: This is an efficacy trial to investigate the clinical and cost-effectiveness of a parent group based intervention designed to help parents understand and manage their child's challenging RRB. If found to be effective, this intervention has the potential to improve the well-being of children and their families, reduce parental stress, greatly enhance community participation and potential for learning, and improve longer-term outcomes. TRIAL REGISTRATION: Trial ID: ISRCTN15550611 Date registered: 07/08/2018. Sponsor and Monitor: Cumbria, Northumberland, Tyne and Wear NHS Foundation Trust R&D Manager Lyndsey Dixon, Address: St Nicholas Hospital, Jubliee Road, Gosforth, Newcastle upon Tyne NE3 3XT, lyndsey.dixon@cntw.nhs.uk , Tel: 0191 246 7222.

Response: Anxiety and behaviour in and beyond ASD; does the idea of 'PDA' really help? - a response to Green (2020).

We acknowledge Green's viewpoint and agree that currently the literature does not support the validity of PDA as an independent syndrome. However, the controversy is real and exists for a reason. We therefore argue that it is important to move beyond labelling and the potentially unhelpful implications of discussion around whether or not it is a condition, diagnosis or a co-morbid condition, to a point of recognition of the phenomenon. We argue that what we need is more accurate description of these behaviours and better measurement to allow us to answer key questions about this phenomenon, whatever we name it. In our paper (Stuart et al, Child and Adolescent Mental Health, 2019) and response, we are trying to move the debate on from opinion and anecdote to be more data-driven, which hopefully leads to increased identification of what research needs to be done. We build on this argument by outlining our second proposed and yet to be published paper exploring the role that IU has in the relationship between ASD, PDA (however we label this) and anxiety.

Intolerance of uncertainty and anxiety as explanatory frameworks for extreme demand avoidance in children and adolescents.

BACKGROUND: Pathological demand avoidance (PDA) is a proposed subtype of autism spectrum disorder (ASD), characterised by extreme avoidance of demands. Demand avoidant behaviour has been proposed to be driven by an anxious need to be in control, although has never been explicitly studied. Emerging evidence suggests intolerance of uncertainty (IU) and anxiety may explain the behaviours seen in ASD. We propose these concepts may be useful starting points for furthering understanding of PDA. METHODS: In Study 1, quantitative methods examined the relationship between PDA, IU and anxiety using data collected in an online survey (N = 214). The sample included cases with clinically diagnosed PDA (n = 69) and those with no clinical diagnosis but parent-identified features of PDA (n = 151). 'Children with a diagnosis of PDA scored significantly higher on the IUS-P (t(212) = 2.45, p < .05) compared to those without a diagnosis of PDA. PDA diagnosis did not impact on scores on any other measure.' In Study 2, a selection of Study 1 participants (n = 11) were followed up with a telephone interview to gain descriptive data relating to PDA and its association with IU and anxiety. RESULTS: Regression analyses indicate that demand avoidant behaviour can be conceptualised in part as a possible attempt to increase certainty and predictability to alleviate increasing anxiety. Children and young people with PDA employed varying strategies to manage IU depending on the level of demand presented and degree of anxiety generated. These strategies can be represented by different features of the behaviour profile seen in PDA (control behaviour, withdrawal to fantasy, and meltdown). These behavioural features of PDA showed differential relationships with IU and anxiety, although all were predicted by IU, only meltdown demonstrated a mediation effect by anxiety. CONCLUSIONS: This study represents one of the first attempts to conceptualise and understand the behavioural features of the PDA profile in children and young people. It builds upon emerging evidence from the ASD literature that IU is a relevant construct for conceptualising demand avoidant behaviour in children who show PDA behaviour. This has potential clinical implications for the assessment and management of PDA in children and young people.

Response: Demand Avoidance Phenomena: a manifold issue? Intolerance of uncertainty and anxiety as explanatory frameworks for extreme demand avoidance in children and adolescents - a response to Woods (2020).

This paper is in response to the commentary written by Richard Woods in which he attempts to provide support for his Monotropism autism theory and the research indicating that Demand Avoidance Phenomena may not be developmentally persistent (Woods, 2019). We acknowledge the continuing controversy around the proposed construct of PDA and the clinical dilemma faced by professionals, within the United Kingdom, following increased demand from families seeking assessment and support. We appreciate that research on this topic is scarce and understanding of PDA behaviours remains limited and that methodological improvements are required. However, it is important to remember that anxiety, which often has an onset in middle childhood and adolescence, is a major risk factor for mental health difficulties. Therefore, treatments targeting underlying and potentially modifiable mechanisms rather than anxiety symptoms may be more likely to be effective.

Teachers' perceptions of Restricted and Repetitive Behaviours (RRBs) in children with ASD: Attributions, confidence and emotional response.

BACKGROUND: Restricted and Repetitive Behaviours (RRBs) are some of the most difficult behaviours to manage in children with Autism Spectrum Disorders (ASD). Although RRBs frequently occur in educational settings, we know little about the way in which teachers understand these behaviours. AIMS: The study aimed to explore the attributions, emotional response and feelings of confidence held by teachers working in different educational settings when faced with RRBs. METHODS AND PROCEDURES: A single group survey design using behavioural vignettes was adopted in order to elicit teacher beliefs and ratings. OUTCOMES AND RESULTS: Analysis indicated that there were differences in the attributions and confidence ratings held for different types of RRBs. Significant differences were also observed between teachers working in mainstream and specialist educational settings. Emotional response and confidence scores were often predictive of one another alongside factors related to teaching experience. CONCLUSION AND IMPLICATIONS: The findings indicate that teachers from mainstream schools potentially hold less helpful beliefs in response to RRBs and therefore are a professional group who may benefit the most from additional support and training. Further research could consider conducting a qualitative exploration of why teachers hold certain beliefs about RRBs and/or sampling those who are less experienced in working with children with ASD.

A Randomised Controlled Feasibility Trial of Immersive Virtual Reality Treatment with Cognitive Behaviour Therapy for Specific Phobias in Young People with Autism Spectrum Disorder.

We examined the feasibility and acceptability of using an immersive virtual reality environment (VRE) alongside cognitive behaviour therapy (CBT) for young people with autism experiencing specific phobia. Thirty-two participants were randomised to treatment or control. Treatment involved one session introducing CBT techniques and four VRE sessions, delivered by local clinical therapists. Change in target behaviour was independently rated. Two weeks after treatment, four treatment participants (25%) and no control participants were responders; at 6 months after treatment, six (38%) treatment and no control participants were responders. At 6 months post-treatment, symptoms had worsened for one treatment and five control (untreated) participants. Brief VRE exposure with CBT is feasible and acceptable to deliver through child clinical services and is effective for some participants.

Mental Wellbeing of Family Members of Autistic Adults.

Family members are often the primary caregiver for autistic adults and this responsibility may impact on the carer's wellbeing and quality of life. 109 family members of autistic adults completed an online survey assessing their wellbeing relating to their caring role for their autistic relative. Family members who were supporting an autistic relative with co-occurring mental health difficulties and who they reported as unprepared for the future, self-reported higher levels of worry, depression, anxiety and stress, and poorer quality of life. These findings emphasise the importance of support for family members of autistic adults, whether through external services to support their relative or individual mental health support for the carer.

Brief Report: What Happens When I Can No Longer Support My Autistic Relative? Worries About the Future for Family Members of Autistic Adults.

Very little is known about autism and adulthood. Family members are often the primary support for autistic adults and frequently express concerns about what the future will hold and what support will be available for their relative. 120 family members of autistic adults completed an online survey exploring concerns about the future for their relative. The most endorsed concerns were "their needs won't be met" (77% worried weekly), "whether they will be happy" (72% worried weekly) and "who will care for them" (58% worried weekly). The results highlight the importance of implementing structured and timely support through collaboration with governmental policy, local commissioning and communication with charities to help prepare family members and their autistic relative for the future.

Anxiety, Intolerance of Uncertainty and Restricted and Repetitive Behaviour: Insights Directly from Young People with ASD.

In order to investigate the experience of anxiety and restricted and repetitive behaviours (RRB) in young people with ASD, 19 families with young people with ASD aged between 13 and 20 years completed questionnaire measures of RRB, anxiety, and intolerance of uncertainty. Ten young people also completed a novel semi-structured interview exploring an individualised example of an RRB. Findings demonstrated that young people with ASD can self-report and show insight in to their RRB, and replicated previous findings based on parent report showing a significant positive relationship between RRB and anxiety. This is the first evidence of young person self-report using both quantitative and qualitative data and indicates a range of reasons why young people may engage in RRB.

Sensory atypicalities in dyads of children with autism spectrum disorder (ASD) and their parents.

Sensory atypicalities are a common feature of autism spectrum disorder (ASD). To date, the relationship between sensory atypicalities in dyads of children with ASD and their parents has not been investigated. Exploring these relationships can contribute to an understanding of how phenotypic profiles may be inherited, and the extent to which familial factors might contribute towards children's sensory profiles and constitute an aspect of the broader autism phenotype (BAP). Parents of 44 children with ASD and 30 typically developing (TD) children, aged between 3 and 14 years, participated. Information about children's sensory experiences was collected through parent report using the Sensory Profile questionnaire. Information about parental sensory experiences was collected via self-report using the Adolescent/Adult Sensory Profile. Parents of children with ASD had significantly higher scores than parents of TD children in relation to low registration, over responsivity, and taste/smell sensory processing. Similar levels of agreement were obtained within ASD and TD parent-child dyads on a number of sensory atypicalities; nevertheless significant correlations were found between parents and children in ASD families but not TD dyads for sensation avoiding and auditory, visual, and vestibular sensory processing. The findings suggest that there are similarities in sensory processing profiles between parents and their children in both ASD and TD dyads. Familial sensory processing factors are likely to contribute towards the BAP. Further work is needed to explore genetic and environmental influences on the developmental pathways of the sensory atypicalities in ASD. Autism Res 2017, 10: 531-538. © 2016 International Society for Autism Research, Wiley Periodicals, Inc.

Facing the Unknown: Intolerance of Uncertainty in Children with Autism Spectrum Disorder.

BACKGROUND: Anxiety is a common problem for children with autism spectrum disorder (ASD). Recent research indicates that intolerance of uncertainty (IU) may be an important aspect of anxiety for this population. IU is the belief that uncertainty is upsetting, and not knowing what is going to happen is negative. There is little known about the phenomenology of IU in children with ASD. We therefore present data from parent focus groups exploring this concept in children with ASD. METHODS: Participants were asked to differentiate IU from dislike of change and fear, and to discuss examples of IU and the strategies they use to manage it. Their experiences of IU and strategies are presented. RESULTS: IU was evident across novel and familiar situations and parents used a variety of strategies to manage IU. Their experiences of IU and strategies used are presented. CONCLUSIONS: Participants were able to identify and provide examples of IU suggesting that it is a recognizable construct among children with ASD.

Towards a Treatment for Intolerance of Uncertainty in Young People with Autism Spectrum Disorder: Development of the Coping with Uncertainty in Everyday Situations (CUES©) Programme.

Intolerance of uncertainty (IU) is indicated as an important transdiagnostic process variable in a range of anxiety disorders. Anxiety is very common in children with autism spectrum disorders (ASD). This study aimed to develop a parent group based manualised treatment programme for young people with ASD, which focused on IU. An eight session programme was developed and then delivered to 11 parents across three treatment groups, two recruited via a research data base and one via clinical services. Data regarding retention, acceptability and feasibility indicate that the parents valued the programme. Effect size analyses of outcome measures for potential use in larger trial indicate that the programme has promise as a treatment option of your people with ASD and IU.

Development of the anxiety scale for children with autism spectrum disorder (ASC-ASD).

Many children with autism spectrum disorder (ASD) experience high levels of anxiety. A widely used measure for typically developing children is the Revised Child Anxiety and Depression Scale (RCADS). However, such anxiety measures may require adaptation to accommodate characteristics of those with ASD. An adapted version of the RCADS was created based on empirical evidence of anxiety phenomenology in ASD, which included additional items related to sensory anxiety, intolerance of uncertainty, and phobias. Content validity was refined during focus groups with parents. Polychoric factor analysis was undertaken on data from 170 children with ASD, aged 8-16, and their parents. This process resulted in the creation of a new 24 item scale (self and parent report) each with four subscales: Performance Anxiety, Uncertainty, Anxious Arousal, and Separation Anxiety, with evidence of good reliability and validity. The freely available Anxiety Scale for Children - ASD, Parent and Child versions (ASC-ASD) has promising psychometric properties including good internal consistency, validity, and 1 month test-retest reliability. Autism Res 2016, 9: 1205-1215. © 2016 International Society for Autism Research, Wiley Periodicals, Inc.

Group therapy for anxiety in children with autism spectrum disorder.

AIM: To investigate the acceptability and feasibility of adapted group therapy for anxiety in children with autism spectrum disorder in a pilot randomised controlled trial. METHOD: A total of 32 children aged 9-13 years were randomised to immediate or delayed therapy using the 'Exploring Feelings' manual (Attwood, 2004). Child and parent groups were run in parallel, for seven weekly sessions, under the supervision of experienced psychologists. The primary blinded outcome measures addressed change in overall functioning and in severity of the primary anxiety diagnosis after 3 months. RESULTS: Children met diagnostic criteria for 1-6 anxiety disorders (median 3). At end point, both parents and children in the immediate therapy group were more likely to report a reduction in anxiety symptoms. Fidelity of delivery of the group therapy was high, and attendance was 91%. CONCLUSIONS: This pilot trial established that children and families were willing to be recruited and randomised, the outcome measures were acceptable, the format and content of the groups were feasible within UK child and adolescent mental health services, the intervention was appreciated by families and attrition was very small.

Parent-mediated early intervention for young children with autism spectrum disorders (ASD).

BACKGROUND: Young children with autism spectrum disorders (ASD) have impairments in the areas of communication and social interaction and often display repetitive or non-compliant behaviour. This early pattern of difficulties is a challenge for parents. Therefore, approaches that help parents develop strategies for interaction and management of behaviour are an obvious route for early intervention in ASD. This review updates a Cochrane review first published in 2002 but is based on a new protocol. OBJECTIVES: To assess the effectiveness of parent-mediated early interventions in terms of the benefits for both children with ASD and their parents and to explore some potential moderators of treatment effect. SEARCH METHODS: We searched a range of psychological, educational and biomedical databases including CENTRAL, MEDLINE, Embase, PsycINFO and ERIC in August 2012. As this is an update of a previous review, we limited the search to the period following the original searches in 2002. Bibliographies and reference lists of key articles were searched, field experts were contacted and key journals were handsearched. SELECTION CRITERIA: We included only randomised controlled trials of early intervention for children with ASD. The interventions in the experimental condition were mediated by parents; the control conditions included no treatment, treatment as usual, waiting list, alternative child-centred intervention not mediated by parents, or alternative parent-mediated intervention of hypothesised lesser effect than the experimental condition. DATA COLLECTION AND ANALYSIS: Two review authors (HM and IPO) independently screened articles identified in the search and decided which articles should be retrieved in full. For each included study, two review authors (IPO and EH) extracted and recorded data, using a piloted data collection form. Two review authors (IPO and HM) assessed the risk of bias in each study. We performed data synthesis and analysis using The Cochrane Collaboration's Review Manager 5.1 software. MAIN RESULTS: The review includes 17 studies from six countries (USA, UK, Australia, Canada, Thailand and China), which recruited 919 children with ASD. Not all 17 studies could be compared directly or combined in meta-analyses due to differences in the theoretical basis underpinning interventions, the duration and intensity of interventions, and the outcome measurement tools used. Data from subsets of 10 studies that evaluated interventions to enhance parent interaction style and thereby facilitate children's communication were included in meta-analyses. The largest meta-analysis combined data from 316 participants in six studies and the smallest combined data from 55 participants in two studies. Findings from the remaining seven studies were reported narratively.High risk of bias was evident in the studies in relation to allocation concealment and incomplete outcome data; blinding of participants was not possible.Overall, we did not find statistical evidence of gains from parent-mediated approaches in most of the primary outcomes assessed (most aspects of language and communication - whether directly assessed or reported; frequency of child initiations in observed parent-child interaction; child adaptive behaviour; parents' stress), with findings largely inconclusive and inconsistent across studies. However, the evidence for positive change in patterns of parent-child interaction was strong and statistically significant (shared attention: standardised mean difference (SMD) 0.41; 95% confidence interval (CI) 0.14 to 0.68, P value < 0.05; parent synchrony: SMD 0.90; 95% CI 0.56 to 1.23, P value < 0.05). Furthermore, there is some evidence suggestive of improvement in child language comprehension, reported by parents (vocabulary comprehension: mean difference (MD 36.26; 95% CI 1.31 to 71.20, P value < 0.05). In addition, there was evidence suggesting a reduction in the severity of children's autism characteristics (SMD -0.30, 95% CI -0.52 to -0.08, P value < 0.05). However, this evidence of change in children's skills and difficulties as a consequence of parent-mediated intervention is uncertain, with small effect sizes and wide CIs, and the conclusions are likely to change with future publication of high-quality RCTs. AUTHORS' CONCLUSIONS: The review finds some evidence for the effectiveness of parent-mediated interventions, most particularly in proximal indicators within parent-child interaction, but also in more distal indicators of child language comprehension and reduction in autism severity. Evidence of whether such interventions may reduce parent stress is inconclusive. The review reinforces the need for attention to be given to early intervention service models that enable parents to contribute skilfully to the treatment of their child with autism. However, practitioners supporting parent-mediated intervention require to monitor levels of parent stress. The ability to draw conclusions from studies would be improved by researchers adopting a common set of outcome measures as the quality of the current evidence is low.

One-year change in repetitive behaviours in young children with communication disorders including autism.

Repetitive behaviours are a relatively neglected area of study in autism. Previous research has concluded that repetitive behaviour is inversely related to ability and that it tends to increase over the preschool years. One-hundred and four children ages 24-48 months, with autism, autism spectrum disorder (ASD) or other disorders, were followed for 13 months. Twelve items from the Autism Diagnostic Interview (ADI-R) were analysed, as well as diagnostic algorithm scores. Ability was related to degree of repetitive behaviours, except for one cluster of relatively able children. ADI-R repetitive behaviour algorithm scores increased over time; however, when all 12 behaviours were considered, there was a general decrease in impact upon the child's and family's activities. Reasons for this decrease are discussed.

Repetitive behaviour and play in typically developing children and children with autism spectrum disorders.

The view of a triad of impairments [(Wing and Gould (1979). Journal of Autism and Developmental Disorders, 9, 11-30] in which impaired imagination is linked with repetitive behaviour is widely accepted. However this categorisation differs from the international classification systems, which link imagination to communication impairments rather than to repetitive behaviours. To investigate this relationship, the Activities and Play Questionnaire-Revised was completed by 196 parents of 2-8-year-old children with autism spectrum disorders (ASD) and typical development. Results showed that repetitive behaviours were associated with play in ASD but not in typical development, supporting Wing and Gould's triad. However there was also an association between play, repetitive behaviour and language, confirming the international classification systems description of imagination as a component of language and communication difficulties.

Use of the questionnaire on resources and stress (QRS-F) with parents of young children with autism.

The Questionnaire on Resources and Stress (Friedrich, short form: QRS-F) has been used widely with parents of children with disabilities. However, its psychometric properties in parents of young children with autism have not been established. Here, 174 mothers and 43 fathers of children under 6 years with autism spectrum disorder were studied by two independent research teams. Each parent completed a 31-item version of the QRS-F. Factor analysis of the mothers' scores on these items failed to identify an expected two- or three-factor structure. Thus, the properties of a total stress score were explored. Analyses revealed evidence of good reliability, and expected associations with social support, coping and autism severity. These analyses lend preliminary support to the convergent validity of the scale. Overall, the data support the use of a total stress score from the 31-item version of the QRS-F in research with parents of young children with autism.

Can a diagnosis of Asperger syndrome be made in very young children with suspected autism spectrum disorder?

Of a cohort of 104 children with Autism, PDD-NOS or specific language disorder, recruited at age 2-3 years of age, only three appeared to meet diagnostic assessment criteria for Asperger syndrome (AS). The children were followed up at 4-5 years, and assessments at both time points included the Autism Diagnostic Interview (ADI-R), the Autism Diagnostic Observation Schedule (ADOS) and the Mullen Scales of Early Learning. The paper explores the reasons why so few children with possible AS were identified early, including problems inherent in the assessment tools and the range of normal variation within characteristics required for a diagnosis. Only 10 children altogether had first words by 24 months, and abilities in the average range, and 9 were followed up. All of these able children had varied repetitive behaviours, and these increased in terms of ADI-R algorithm score over a 13 month interval. However, items concerning resistance to change and liking of routines tended to decrease in terms of reported impact on the child and family. Repetitive behaviours seem significant in the early referral of able children for a PDD diagnosis, but identification of children with AS is more likely to occur reliably once children are older and enter school.