Improving outcomes in autism spectrum disorder through effective service provision: diagnostic assessment pathways and parent focussed interventions

Abstract

Introduction: This thesis on the topic of autism spectrum disorder presents six publications focussed on research about improving outcomes through effective service provision, together with a critical appraisal, which adds depth and breadth to the reasoning and decision making involved in this work. The work follows an iterative process and is positioned from a pragmatist philosophical standpoint, using mixed methods to clarify shared language, concepts and meanings and to ensure translation of research findings into real world practice. The thesis provides evidence of the urgent need for research to inform services as to how they might address the issue of delays in ASD assessment and diagnosis, alongside better understanding of which interventions improve wellbeing outcomes. Aims: This portfolio of published research has arisen from three related research programmes, with the unifying objectives of: 1. Adding to clinical and research knowledge of current ASD service provision across the lifespan and how that might be improved through adherence to ASD clinical guidelines; 2. Reducing family stress associated with ASD by reducing delays in diagnosis through identification of factors which influence efficiency and quality in a diagnostic pathway, and; 3. Developing understanding of the effectiveness of parent focused interventions to inform future research and practice. The critical appraisal aims to: (a) Explore key areas for debate that have arisen in the work, which transcend the individual publications; (b) Position this debate within the context of international literature, research evidence and theory in relation to autism, implementation science and pragmatist epistemology underpinning the work; (c) Highlight the contribution of this research to the advancement of clinical practice and research knowledge, and the potential for further clinical reach and informing evidence based practice through diffusion of innovation. Methods: Research aims, methods and outcomes are presented within a series of publications, using mixed methods to seek to address these. Findings: Through this research, it was identified that the wait for diagnosis in child and adult services, from referral to diagnosis shared, exceeds the recommended 119 day time standard in 74% of child and 59% of adult services. There was a significant difference in mean age of referral and diagnosis for girls compared to boys and this delay occurred prior to referral through delayed recognition rather than through delays in the assessment process. Findings provide strong evidence of the need to address the way we collectively deliver ASD services. This research identified factors which influence waiting times in child and adult services. These included the availability of relevant pre-referral information at first appointment, consideration of the number of contacts or appointments used to reach a conclusion and complexity of the case. Mixed methods were used to further identify a broader range of factors affecting wait times in each service and to develop child and adult action plans as proposed solutions that could be applied by local service providers. Plans developed were then successfully applied in a 12 month service improvement intervention with 11 adult services, resulting in a statistically significant reduction in duration of assessment (b=-0.25, t(136) = -2.88, p=0.005), taking the duration to within the recommended timescale. In child services, this model also led to a statistically significant reduction in waiting times for diagnostic assessment and increased identification of girls with ASD. Systematic review and meta analysis of parent focused intervention for older children and adults found that a) parent training and education and b) mindfulness interventions provide measureable improvements in family wellbeing, which in turn are known to have a reciprocal effect on the individuals with ASD. A number of recommendations for future research arise from this work. These include the aspiration for an ASD specific wellbeing measure which can be applied with greater consistency across ASD intervention studies; and the need for consensus on theoretical models to underpin evaluation of complex interventions in ASD, which in turn may lead to deeper understanding of which elements of interventions are most effective in which circumstances. Conclusion: In a field where research evidence has been lacking, this body of work applies a range of research methods, in order to add to the evidence base and provide practical steps, which clinical service providers could apply, to reduce the delays in diagnosis. Earlier access to ASD specific interventions, as a result of earlier diagnosis, has the potential to improve wellbeing for individuals with ASD and their families. Despite limitations of the research on ASD parent focused interventions for older children and adults, findings reported here add further support to the ASD clinical guideline recommendations to use such interventions.