DIADS - Diagnostic Instruments for Autism in Deaf Children Study

Up to 4% of people who are deaf may have autism or another autism spectrum condition (ASC). However, this can be hard to spot because many of the tools used to diagnose autism were never designed with deaf people in mind. For example, some questions on the Autism Diagnostic Interview - Revised (ADI-R), a tool used by clinicians and researchers, will ask about how a child responds when someone calls their name and the autism diagnostic observation schedule uses a toy telephone to examine make believe play.

The aim of this project is to develop and test a set of tools that will help us to identify and diagnose autism in deaf children. We plan to do this by adapting some of the existing measures of autism, so that they are fair tests that are accessible to deaf and hearing parents and can be used effectively with deaf children. We are going to do this with the help of deaf and hearing families, experts and researchers in the fields of deafness and autism, and organisations such as the National Autistic Society (NAS) and the National Deaf Children's Society (NDCS).

The objectives are split into stages.

In the first stage of the project we will interview a range of families about deafness and autism. This will involve deaf and hearing parents of deaf autistic children. The deaf children with autism will also have a play-based assessment called the ADOS 2. This will be compared to hearing children's symptoms of autism.

We will then use this information and a review of research to design a questionnaire for parents and professionals. All this information will go to an expert panel to review look at the information from the interviews and start to inspect whether current items in the questionnaires are appropriate. A formal scoring questionnaire (called a Delphi Consensus) will then be used with deaf autism international experts to identify modifications. They will suggest items that can be included without change, ones that needs adapting (and suggest changes), and ones that should be rejected. Revisions will be re-circulated with further scoring until an agreement can be reached. We will use agreement levels as our first milestone.

In the 2nd stage, the newly adapted questionnaires will then be translated into British Sign Language (BSL) using a strict translation/back translation methodology. A group of translators will video the items in BSL, and then another group of translators who have not seen the original questionnaire will translate them back into English. This process will repeat as necessary. Then focus groups of deaf people will check the translations and may suggest more changes. Once there is agreement, the original authors will check final versions.

We will check that the new items and questionnaires work well in the 3rd stage when we will use them with

65 deaf children with autism
65 deaf children without autism (+ 65 more for screening)
65 hearing children with autism
65 hearing children without autism

We will collect a range of other important information about epidemiology language, intelligence etc.

After the first 20 autistic children have been recruited, we will undertake a futility analysis. This lets us see if there is benefit in continuing the research by checking with statistics if the results are looking promising (milestone 2).

We will also look at whether it is good at picking up autism in children that do have autism (sensitivity), and good at ruling autism out in children that don't have autism (specificity).

This will help us identify and diagnose autism better in young people who are deaf. It will also make it more accessible by being available in many deaf people's first language. This will mean earlier diagnosis and earlier treatment. We will develop training for National Deaf Child and Adolescent Mental Health Services and other clinicians in using the new measures for screening and assessing for autism spectrum disorders.

Design: Modification of screening and assessment instruments for autism spectrum disorders, followed by translation, and field testing to establish robust validity and reliability, compared to instruments used with hearing children.

Setting: National Deaf Child and Adolescent Mental Health Services (four main centres and six outreach centres) with strong academic, clinical expert and user partnership involvement.

Target Population: Children and young people aged 2-18 who have a permanent bilateral hearing loss of 40 dB or more, who have been referred for an assessment for an autism spectrum disorder, and selected assessment groups.

Outcomes: Fully validated and reliable screening and assessment instruments for autism in deaf children. This will be acceptable to users and we will evidence key psychometric properties of these instruments.

Sample Size: 195 children and young people for assessment instruments (260 for screening)

Milestones
Milestone 1: agreement on Delphi Consensus of 80% (75% acceptable). If not met will lead to cessation of grant with further field research being pursued.
Milestone 2: futility analysis after 20 deaf children with and 20 without autism recruited. 0.8 under ROC curve (0.75 acceptable). If the area under ROC curve were 0.8, this would correspond to a difference in mean score of 1.2 standard deviations. To detect the existence of a difference between means with power 0.95 if the true difference were 1.2 would require 19 in each group. If we do not detect a significant difference the test will be deemed unsuitable for diagnostic purposes. This will lead to a cessation of the grant with further research into autism in deaf children and a new approach to screening with further grant applications and discussions with MRC.
Milestone 3: 60% of target population recruited by 30 months (55% acceptable). Failure to meet this will result in meeting with MRC and steering group and urgent recruitment strategies.

Who will benefit from this research?
Children and Families
Deaf children and families will benefit from this research since it will provide new and more accurate tools for screening, assessment and diagnosis.

This will lead to earlier diagnosis since many children are not being assessed because services believe that they do not have the skills to assess for autism in deaf children, as evidenced by Social Policy Unit Research (2008/9/10).

This in turn will lead to earlier interventions and intervention planning and may lead to more appropriate and earlier support. This in turn will lead to potentially better outcomes for deaf children with autism. Research suggests that early intervention in autism is very helpful as it does for deaf children. Early intervention in deaf children WITH autism is therefore likely to be effective. We plan to research this.

Child and Adolescent Mental Health (CAMH) clinicians who assess for autism will benefit since it will generate much better guidance on screening and assessment for autism in deaf children.

Clinical beneficiaries will include wider groups than child psychiatrists and clinical psychologists and there has already been interest from cochlear implant centres and child mental health staff in generic child and adolescent mental health (CAMH) services and specialist National Deaf CAMH ten centres in England. There is also international interest. Recent conferences in Austria (June 2012) and York (October 2011) have highlighted this. Other disciplines such as paediatricians and speech and language therapists in Health services and Educational Psychologists in Local Authority services will also benefit.

We believe that at the end of this research the new tools will be immediately available for use, and we will make time available to provide training materials (DVD, manual) for their use.

Staff working on this project will develop a range of skills and experience of assessing multiple children that will greatly help in improving services and expertise across the country.

The results will provide policy makers with much better estimates of autism prevalence within the deaf child population. This will inform education and social care provision and we will be working closely with stakeholders such as National Deaf Children's Society and the Social Research for Deaf People (SORD) unit at Manchester University.

Public Health and National Strategic Importance
Benefits may also accrue more widely to society. For example, we have come across deaf teenagers with severe behaviour problems with autism where the diagnosis was not made in early life and where the child has gone from school to school because of expulsions eventually going to deaf residential school. Such young men will have ongoing behaviour problems in adulthood and need long term care. It is our belief that early assessment, diagnosis and intervention planning may have led to much improved understanding of difficulties and better outcomes.

The research may benefit society by leading to more cost-effective interventions which help young people learn skills that can be productive for them and society. It has great potential to lead to enhancement in the quality of life of young people and their families, with improved health outcomes and greater creative output. We plan to research this.

The research will also help to publicise the importance of Deaf culture leading to greater deaf awareness in schools and society.

This research will make the UK leaders in this field. England is the only country that has a National Deaf Child Mental Health Service with good geographical spread nationally, and is a flagship in this area.