Diagnostic pathways for children with possible autism. Which work best, for whom, when, and at what cost? Autism is a complex neuro-behaviour condition. People with autism have difficulty with social interaction and in communication with others. They may struggle with change, and repeat actions over and over. Life may be very anxious or stressful. The signs of autism can occur at any age but often appear in the first two years of life. There is no one type of autism, but many, so the condition is now called autism spectrum disorder (ASD). Autism is lifelong but this study is only about children. Caring for a child with autism can be difficult and can sometimes be tough on the whole family.

This project aims to guide the people who plan services for families and children. Different teams and services that do autism assessments will help us. The investigators will ask teams and services: What speeds up diagnosis? What delays diagnosis?

The study will be in four work packages:

1. The investigators will review research in the UK and abroad to find evidence and ideas that will help speed up diagnosis.
2. The investigators will survey professionals who work for the specialist teams who diagnose autism. The survey will be about each step in the process and ask which professionals get involved. The investigators will ask about the number of children they see and the time it usually takes to reach a diagnosis. This will give us a picture of the national situation.
3. After the national survey, the investigators will select around six or eight teams. These teams will be using different and innovative approaches. The investigators will study those approaches. The investigators will talk to clinical staff, managers, referrers, parents and young people. Parents and young people will have gone through the diagnostic process. The investigators will ask parents and young people about their experiences and views. The investigators will review the steps in the diagnosis process for about 70 children in each service. The investigators will find out how long each assessment takes, how much clinical time it takes, and how much it costs. The investigators will compare findings across teams and services.
4. The investigators will have national meetings with autism experts and patient groups. The investigators will show them our findings. These groups will agree recommendations for practice. Clinical teams, service managers, commissioners, parents' groups, and NHS England will receive recommendations.

The research team has specialist expertise in autism, health services, economics, and statistics. The team includes public and NHS England partners. This will ensure the investigators take account of the needs of families and the investigators send the findings to people who plan services.

Scientific Summary (following Rameses 2 reporting guidelines)

Study Title

A Realist Evaluation of Autism ServiCe Delivery (RE-ASCeD): Which diagnostic pathways work best, for whom, when, and at what cost?

Research Question Which autism diagnostic pathways work best, for whom, in what circumstances, and at what cost when providing timely and high quality diagnostic services?

Secondary Research Questions

• What factors underpin timely diagnosis service delivery success or failure?
• What needs to be in place to identify the needs and create individualized, holistic pathways of diagnosis for the child and their family?

Background The NHS England Long Term Plan states: "Children and young people with suspected autism wait too long before being provided with a diagnostic assessment. Over the next three years the investigators aim to test and implement the most effective ways to reduce waiting times…achieving timely diagnostic assessments in line with best practice guidelines...(and) to support children with autism and their families, throughout the diagnostic process." Set against a background of increasing demand for diagnostic assessment, and the need, as recommended by the NICE guidelines, for a multidisciplinary approach to assessment, taking at least 13 hours of professional time and £800 per child, evidence is needed to identify models of diagnostic assessment to achieve timely diagnoses, and how families can best be supported through this process. The NHSE Long Term Plan aims to reduce the wait time for diagnosis, and the aim of this project is to find ways/models to directly address that need.

Aims and Objectives This study aims to examine existing models of Autism diagnostic service delivery, and to investigate which approaches offer the most timely, cost-effective, high quality and child and family friendly solutions, and in line with realist thinking, to identify the factors that are likely to underpin their success, or failure, if rolled out more generally across providers.

Method

The study will use Realist Evaluation methodology, which enables evaluation of complex interventions in health care by building on existing research, to identify "what works, for whom and in what circumstances". Mixed methods, including Rapid Realist (literature) Review, survey of current practice, and case studies of centres delivering promising approaches, will be used to build a picture in an iterative manner, to identify underlying Contexts and Mechanisms that contribute to the desired Outcomes (CMO configurations) of improved Autism diagnostic service delivery and user experience, that in turn can provide plausible explanations of why the service models work or do not in certain circumstances. Data analysis from each phase of the study will be synthesised through a realist evaluative process comprising of four stages:

1. Articulation of programme theories and propositions from WP1 of the study (rapid realist review). Identification of candidate CMO configurations.
2. Data collection from WP4 (case studies) of the study to test and refine propositions.
3. Map the outcomes of each model across the cases; interrogate what contexts and mechanisms explain the pattern of outcomes.
4. Through stakeholder consolidation (WP5), refine explanatory CMO configurations to evaluate what works best and in what circumstances to deliver timely and cost-effective diagnostic services that meet the needs of children with possible Autism

Initial Programme Theory (Based on NICE principles) If there is an MDT assessment by a team with competencies in child neurodevelopment and mental health (context), then Autism will be recognised as a complex condition that relies on detailed history and observation across settings (mechanism) to diagnose it, leading to an accurate diagnosis, recognition of associated comorbidity such as ADHD and intellectual disability (outcome), and the ruling out of complex differential diagnoses.

This will also create - whilst not an explicit part of this project - accurate pictures of child strengths and needs to inform individualized packages of support and intervention through health, education and social care (outcome).

Work Package One (months 1-7). Rapid Realist Review. Evidence will be sought from published studies and evaluations of Autism diagnostic services, including controlled trials, uncontrolled studies, cost-effectiveness studies, process evaluations, grey literature and qualitative studies, including unpublished information about locally implemented Autism services. The purpose will be to refine or develop initial programme theories.

Work Package Two (months 1-6): National/International Survey of Current Diagnostic Practice: An electronic survey will be sent to UK CDCs, CAMHS teams, and tertiary centres, delivering Autism diagnostic services. This will ask teams to describe current challenges to, and any new models of diagnostic service delivery they are using. Analysis will use both qualitative and quantitative methods, to identify common themes, and services using novel approaches.

Work Package Three (months 7-9): Purposive Sampling Frame. The findings of WP 1 & 2 will be used to develop a matrix of the key features of service delivery, plus other contextual factors such as lower SES, rural, urban etc. This will be used as a sampling frame. It is anticipated that 6-8 service delivery models will be purposefully selected as case studies across the matrix to ensure the investigators have a full range of delivery models and population characteristics.

Work Package Four: Case studies (months 10-29): In-depth exploration of individual service models will be carried out to determine the interrelationship of context, intervention, mechanisms and outcomes as they occur in their natural setting. Data will be gathered within case studies using mixed methods at two levels: (i) service model and (ii) individual child/ family. Methods will include semi structured realist Interviews, focus groups, and document reviews to assemble a comprehensive description of the service and model. Interviewees will include commissioners, service managers, clinicians, referrers, parents and young people. Interviews will be recorded and transcribed, proceeding to qualitative analysis based on components of the programme theory. Regular team meetings will discuss coding, address inconsistencies, and refine collective understanding of coding framework. Coded data will be collated using NVivo software.

For each case study the journeys of at last 58 children completing the diagnostic pathway will be recorded from case notes to measure pathway, hours of professional input and time to diagnosis. Costs incurred by families for example travel to clinic, loss of earnings, will be collected by interview. Associations between service use costs and characteristics of the child and model will be assessed by regression analysis.

Work Package Five: Consolidation event, Months 27-28 : The investigators will run two iterative national consensus workshops, inviting key stakeholders including NHSE and parents/young people, to refine and confirm the context-mechanism-outcome configurations, and salient actions required to deliver timely, reliable and costed diagnostic services that meet the needs of children with possible Autism. This will lead to dissemination of models to inform the ongoing development of service models within the NHSE Long-Term Plan, commissioning guidelines and local service development.

Survey Inclusion Criteria:

• Lead clinicians for childhood autism diagnostic assessment teams (children age 1-18 years), or someone who they nominate.

Survey Exclusion Criteria:

• Members of staff not involved in Autism diagnostic services.

Case Studies Inclusion Criteria :

• Parents of young people who have been through the diagnostic pathway
• Young people who have been through the diagnostic pathways
• Clinicians working in centres involved in the autism diagnostic process
• Commissioners and managers of autism services; professionals who refer in to the autism pathways

Case Studies Exclusion Criteria:

• Adults on the autism pathway
• Professionals involved in the adult autism pathway

Members of staff. referrers, and users of Autism diagnostic services for children.

IPD data that is anonymised, will be released by the project steering commitee,and only then, IPD data that underlie results in publication will be shared

Starting September 2023

Anonymised IPD will be shared if the applicant writes to the project steering committee who will review the request. Access will only be allowed once the study is complete and all publications are complete.