Autism OSSDX Study
Research type
Research Study
Full title
Global Open Access Screening and Diagnostic Tools for ASD – Prototype Development and Feasibility Evaluation
IRAS ID
289325
Contact name
Melissa Gladstone
Contact email
Sponsor organisation
University of Liverpool
Duration of Study in the UK
0 years, 10 months, 31 days
Research summary
Summary of Research
This study has been developed to design and pilot a screening and a diagnosis tool for autism spectrum disorder (ASD) that is culturally appropriate and open access, so that it has global application.
Children in Low and Middle Income Countries (LMICs) at a higher risk of developing neurodevelopment disorders (NDD's) and disabilities, particularly due to more exposure to causative factors such as infections and brain trauma. There is also anecdotal evidence that indicates that children also receive a diagnosis later in their development. Despite the burden of these conditions, very little data exists on how common NDD's including ASD is in different settings globally. This is due in part to the absence of culturally-appropriate measures and assessment procedures, to enable the identification and diagnosis of NDD's including ASD. The majority of screening and diagnostic tools available, have been developed by and for use in high income country settings.
Therefore, this study will aim to develop and begin testing a suite of screening and diagnostic instruments that will be accessible, appropriate, acceptable, reliable and valid for global use.
Summary of Results
The Global Open Access Screening and Diagnostic Tools for ASD – Prototype Development and Feasibility Evaluation (Autism OSSDx) Study was conducted in Liverpool at Alder Hey Children’s Hospital NHS Foundation Trust from June 2022 to October 2024. The study was funded by Autism Speaks and sponsored by the University of Liverpool. We would like to thank everyone who took part.
This study was developed to design and pilot a screening and diagnosis tool for autism spectrum disorder (ASD) that is culturally appropriate and open access, so that it has global application.
Children in Low and Middle Income Countries (LMICs) are at a higher risk of developing neurodevelopmental disorders (NDDs), particularly due to more exposure to causative factors such as infections and brain trauma. There is also anecdotal evidence that indicates that children receive a diagnosis later in their development. Despite the impact of these conditions, very little data exists on how common NDDs, including ASD, is in different settings globally. This is due in part to the absence of culturally-appropriate measures and assessment procedures, to enable the identification and diagnosis of NDDs including ASD. The majority of screening and diagnostic tools available, have been developed by and for use in high income country settings.
To develop the prototype tools, 11 experts from across the world provided opinions on relevance of items found in academic literature in comparison with the DSM-V (the standard classification of mental disorders used by mental health and other health professionals and used for both diagnostic and research purposes). A further expert evaluation with 70 experts from across the world was used to create the study prototype screening and diagnosis tool.
Before the pre-pilot testing, a small study was conducted in Liverpool to test the tolerability of the interview format and diagnostic assessment prototype tool. The screening tool is based on caregiver report and covers all the core features of ASD. The diagnosis tool consists of 4 parts: developmental history, caregiver report on ASD symptoms, child observational section on ASD symptoms, and assessment of the impact on day-to-day functioning.
The study took place in three countries: the UK (in Liverpool), Kenya (in Kilifi and Nairobi) and South Africa (in Cape Town). Participants were recruited in the UK and in Kenya for the pre-pilot of the screening and diagnosis tools.
In the UK, pre-pilot of the screening and diagnosis tools and interviews about the feasibility and acceptability of the tools were completed with 12 pairs of children and their caregivers from August 2022 to January 2023. Children included in the pre-pilot testing were aged 2-9 years and had a confirmed ASD diagnosis using the DSM-V criteria by a team with expertise in ASD diagnosis at Alder Hey Children’s Hospital NHS Foundation Trust in Liverpool.
Families were provided with information about the study at their appointment in which their diagnosis of ASD was given. Families interested in taking part were contacted by the study researchers to give further information and arrange a study appointment if they chose to take part. Informed consent was given by all caregivers who took part in the study and from children, where relevant.
Families were given the option of attending online or in-person for the pre-pilot assessment. They were also given the option to complete the interview and observation either as a single appointment or two separate appointments, lasting a maximum of 120 minutes in total. Information from the interviews and pre-pilot assessment in Liverpool and Kenya was combined and the screening and diagnosis tools were adapted based on feedback from families and researchers. The adapted version of the screening and diagnosis tools were used for the pilot testing stage in the UK.
In the UK, pilot testing of the diagnosis tool was completed with 19 children and their caregivers from March to May 2023. Children included in the pilot testing were aged 2-9 years and had a confirmed ASD diagnosis using the DSM-V criteria by a team with expertise in ASD diagnosis at Alder Hey Children’s Hospital NHS Foundation Trust in Liverpool. The pilot assessment was completed in-person with pairs of children and their caregivers in a single appointment, lasting between 60-90 minutes.
In Liverpool, the diagnosis tool was tested with 14 boys and 5 girls; 4 were aged 2-3 years, 6 4-5 years, 5 6-7 years, and 4 8-9 years. Most children were in mainstream primary or nursery school (17 of 19) and had specific difficulties with learning or thinking compared to their peers (15 of 19). Most families were from areas of high levels of deprivation (18 of 19); with a mother who was educated to college or university level (9 and 7 of 19); and a father who was educated to college level (9 of 19). In addition to testing the diagnosis tool, caregivers completed a validated questionnaire for symptoms of ASD and assessment of their development if under 5 years or learning if over 5 years.
Total scores on the ASD specific items of the questionnaire ranged from 16-44 with an average of 32 from a maximum possible score of 50 (indicating that the child would met all DSM-V diagnostic criteria for ASD) and minimum of 0 (indicating that the child does not meet any of the DSM-V diagnostic criteria for ASD). Scores on the observation section ranged from 6-33 with an average of 16 from a maximum possible score of 46 (indicating that the child would met all DSM-V diagnostic criteria for ASD) and minimum of 0 (indicating that the child does not meet any of the DSM-V diagnostic criteria for ASD).
Based on our collective findings across countries, the screening and diagnosis tools have been adapted and will be available for further research. More information on the study findings will be available through the funding charity Autism Speaks and through the study team.
REC name
North West - Greater Manchester East Research Ethics Committee
REC reference
21/NW/0289
Date of REC Opinion
16 Dec 2021
REC opinion
Further Information Favourable Opinion