Title: Development and validation of a new instrument and assessment protocol for assessing autism in young children with severe visual impairment
Funder: Department of Health and Social Care

Abstract: Background to research Currently there is no Visual Impairment specific assessment protocol which has been scientifically tested for its reliability in assessing autism in young children with VI, preventing early reliable and replicable diagnosis. As part of developing a new comprehensive assessment protocol, a novel instrument, the Visual Impairment Social Communication Observational Schedule (VISCOS) has been tested for feasibility with a national cohort of infants with VI. This study sets out to test the VISCOS and other adjusted standardised parent rating interviews for reaching a reliable diagnosis of autism in young children with VI. Aims of the research including questions to be addressed This study aims to develop and validate a scientifically robust diagnostic assessment protocol for detecting and reliably diagnosing autism in children with VI aged 4-6 years. The questions to be addressed include Does the VISCOS observational coding tool have internal coherence and reliability when used to code behaviour that is elicited by standard behaviour presses? Does the VISCOS show criterion reliability, including concurrent and discriminant validity, in detecting autism versus non-autism when compared to clinician formulation and other adjusted standardised parent rating measures? Does the VISCOS show consistency between ratings at 2, 3 and 4-6 years on the same cohort and how reliably can the VISCOS be used to predict clinician rating of autism at 4-6 years? Do adjusted parent rating scales, SCQ4, Social Responsiveness Scale3 and 3Di2 (with vision related items removed), have discriminant validity in detecting autism versus non-autism according to expert clinician formulation? Plan of investigation A national representative cohort of young children with severe VI who are already recruited on the OPTIMUM project (n=79) will be invited to join the project when they reach 4-6 years of age. The children have heterogeneous visual disorders of either simple or complex congenital disorders of the peripheral visual system. Additional children will be recruited cross-sectionally to ensure a total sample size (n= 100). Children will be invited to a one day assessment in the research centre to undertake assessment by a trained psychologist including cognition, language and standardised behavioural presses to elicit social interaction, communication, play and other behaviour which will be video d. Parents will be asked to rate a number of autism screening and adaptive behavioural questionnaires. The videotaped episodes will be coded according to the VISCOS by the psychologist assessor and also separately by an independent expert clinician who is masked to the VISCOS ratings. Statistical analyses will determine the psychometric properties of the VISCOS and parent rating interview measures in discriminating autism versus non-autism cases as judged by clinician formulation. Predictive validity of earlier use of the VISCOS at 2 and 3 years with the same cohort will be undertaken to establish earliest reliability of diagnostic screening. Summary of potential benefits to patients and the NHS There is a high clinical need to reliably diagnose autism in this population as early as possible to ensure parents and practitioners understand the child s needs and to enable early preventative and interventionist practices to be put into place. The findings of the project will demonstrate the efficacy and reliability of the VISCOS and adjusted parent rating interview measures in diagnosing autism in young children with VI and how early the diagnostic procedure and early screening can be reliably undertaken. The study will lead to a new scientifically validated comprehensive assessment protocol and procedures for diagnosing autism in children with VI, including a new instrument as part of the diagnostic process (VISCOS), which can then be rolled out across the NHS through dissemination of findings and training to appropriate clinicians to assist in their clinical diagnostic practice.

Lay abstract: Young children with profound or severe visual impairment have a high risk of developing difficulties in social communication and interacting with others and behaviour problems. If left untreated, these early difficulties may lead to long term autistic problems, causing significant child problems as well as a high challenge and burden for their parents. There is an urgent need for a valid way of identifying these difficulties so that the appropriate support and intervention can be put in place. Current assessment methods and tools are not suitable as they are designed for sighted children. This project will develop a novel scientific designed clinical assessment procedure including a new assessment measurement tool, the Visual Impairment Social Communication Observation Schedule (VISCOS). 100 children with profound or severe visual impairment, aged 4-6 years, will be assessed in their cognition, language and then video d undertaking novel standard tasks in social communication, play and behaviour. The videos of the social communication, play and behaviour will be coded by an expert rater using the VISCOS and independently by experienced clinicians. The clinician ratings will be used to check that the VISCOS is a reliable new assessment tool and is fit for purpose . A second part of the study compares the children s scores on the VISCOS with their earlier performance on similar play based activities at 2 and 3 years of age (which has been previously collected). This will lead to establishing the earliest age in which significant social communication difficulties or risk for autism can be accurately identified. The findings of the project will provide the new assessment procedure and tool for enabling us to identify social communicative difficulties and safe and accurate diagnosis of autism in young children with visual impairment, thereby ensuring that they have access to the earliest and most effective support and intervention.

Research activity codes: 4.1 DISCOVERY AND PRECLINICAL TESTING OF MARKERS AND TECHNOLOGIES
Health categories: MENTAL HEALTH;EYE