Helen McConachie1, Jeremy R Parr2, Magdalena Glod1, Jennifer Hanratty3, Nuala Livingstone3, Inalegwu P Oono1, Shannon Robalino1, Gillian Baird4, Bryony Beresford5, Tony Charman6, Deborah Garland7, Jonathan Green8, Paul Gringras4, Glenys Jones9, James Law1, Ann S Le Couteur1, Geraldine Macdonald3, Elaine M McColl1, Christopher Morris10, Jacqueline Rodgers2, Emily Simonoff6, Caroline B Terwee11, Katrina Williams12. 1. Institute of Health and Society, Newcastle University, Newcastle upon Tyne, UK. 2. Institute of Neuroscience, Newcastle University, Newcastle upon Tyne, UK. 3. School of Sociology, Social Policy and Social Work, Queen's University Belfast, Belfast, Northern Ireland, UK. 4. Guy's and St Thomas' NHS Foundation Trust, London, UK. 5. Social Policy Research Unit, University of York, York, UK. 6. Institute of Psychiatry, Psychology & Neuroscience, King's College London, London, UK. 7. National Autistic Society North East Autism Resource Centre, Newcastle upon Tyne, UK. 8. Institute of Brain, Behaviour and Mental Health, University of Manchester, Manchester, UK. 9. School of Education, University of Birmingham, Birmingham, UK. 10. PenCRU, Child Health Group, University of Exeter Medical School, University of Exeter, Exeter, UK. 11. Department of Epidemiology and Biostatistics, VU University Medical Center, Amsterdam, The Netherlands. 12. University of Melbourne, Royal Children's Hospital and Murdoch Childrens Research Institute, Melbourne, Australia.
Abstract
BACKGROUND: The needs of children with autism spectrum disorder (ASD) are complex and this is reflected in the number and diversity of outcomes assessed and measurement tools used to collect evidence about children's progress. Relevant outcomes include improvement in core ASD impairments, such as communication, social awareness, sensory sensitivities and repetitiveness; skills such as social functioning and play; participation outcomes such as social inclusion; and parent and family impact. OBJECTIVES: To examine the measurement properties of tools used to measure progress and outcomes in children with ASD up to the age of 6 years. To identify outcome areas regarded as important by people with ASD and parents. METHODS: The MeASURe (Measurement in Autism Spectrum disorder Under Review) research collaboration included ASD experts and review methodologists. We undertook systematic review of tools used in ASD early intervention and observational studies from 1992 to 2013; systematic review, using the COSMIN checklist (Consensus-based Standards for the selection of health Measurement Instruments) of papers addressing the measurement properties of identified tools in children with ASD; and synthesis of evidence and gaps. The review design and process was informed throughout by consultation with stakeholders including parents, young people with ASD, clinicians and researchers. RESULTS: The conceptual framework developed for the review was drawn from the International Classification of Functioning, Disability and Health, including the domains 'Impairments', 'Activity Level Indicators', 'Participation', and 'Family Measures'. In review 1, 10,154 papers were sifted - 3091 by full text - and data extracted from 184; in total, 131 tools were identified, excluding observational coding, study-specific measures and those not in English. In review 2, 2665 papers were sifted and data concerning measurement properties of 57 (43%) tools were extracted from 128 papers. Evidence for the measurement properties of the reviewed tools was combined with information about their accessibility and presentation. Twelve tools were identified as having the strongest supporting evidence, the majority measuring autism characteristics and problem behaviour. The patchy evidence and limited scope of outcomes measured mean these tools do not constitute a 'recommended battery' for use. In particular, there is little evidence that the identified tools would be good at detecting change in intervention studies. The obvious gaps in available outcome measurement include well-being and participation outcomes for children, and family quality-of-life outcomes, domains particularly valued by our informants (young people with ASD and parents). CONCLUSIONS: This is the first systematic review of the quality and appropriateness of tools designed to monitor progress and outcomes of young children with ASD. Although it was not possible to recommend fully robust tools at this stage, the review consolidates what is known about the field and will act as a benchmark for future developments. With input from parents and other stakeholders, recommendations are made about priority targets for research. FUTURE WORK: Priorities include development of a tool to measure child quality of life in ASD, and validation of a potential primary outcome tool for trials of early social communication intervention. STUDY REGISTRATION: This study is registered as PROSPERO CRD42012002223. FUNDING: The National Institute for Health Research Health Technology Assessment programme.
BACKGROUND: The needs of children with autism spectrum disorder (ASD) are complex and this is reflected in the number and diversity of outcomes assessed and measurement tools used to collect evidence about children's progress. Relevant outcomes include improvement in core ASD impairments, such as communication, social awareness, sensory sensitivities and repetitiveness; skills such as social functioning and play; participation outcomes such as social inclusion; and parent and family impact. OBJECTIVES: To examine the measurement properties of tools used to measure progress and outcomes in children with ASD up to the age of 6 years. To identify outcome areas regarded as important by people with ASD and parents. METHODS: The MeASURe (Measurement in Autism Spectrum disorder Under Review) research collaboration included ASD experts and review methodologists. We undertook systematic review of tools used in ASD early intervention and observational studies from 1992 to 2013; systematic review, using the COSMIN checklist (Consensus-based Standards for the selection of health Measurement Instruments) of papers addressing the measurement properties of identified tools in children with ASD; and synthesis of evidence and gaps. The review design and process was informed throughout by consultation with stakeholders including parents, young people with ASD, clinicians and researchers. RESULTS: The conceptual framework developed for the review was drawn from the International Classification of Functioning, Disability and Health, including the domains 'Impairments', 'Activity Level Indicators', 'Participation', and 'Family Measures'. In review 1, 10,154 papers were sifted - 3091 by full text - and data extracted from 184; in total, 131 tools were identified, excluding observational coding, study-specific measures and those not in English. In review 2, 2665 papers were sifted and data concerning measurement properties of 57 (43%) tools were extracted from 128 papers. Evidence for the measurement properties of the reviewed tools was combined with information about their accessibility and presentation. Twelve tools were identified as having the strongest supporting evidence, the majority measuring autism characteristics and problem behaviour. The patchy evidence and limited scope of outcomes measured mean these tools do not constitute a 'recommended battery' for use. In particular, there is little evidence that the identified tools would be good at detecting change in intervention studies. The obvious gaps in available outcome measurement include well-being and participation outcomes for children, and family quality-of-life outcomes, domains particularly valued by our informants (young people with ASD and parents). CONCLUSIONS: This is the first systematic review of the quality and appropriateness of tools designed to monitor progress and outcomes of young children with ASD. Although it was not possible to recommend fully robust tools at this stage, the review consolidates what is known about the field and will act as a benchmark for future developments. With input from parents and other stakeholders, recommendations are made about priority targets for research. FUTURE WORK: Priorities include development of a tool to measure child quality of life in ASD, and validation of a potential primary outcome tool for trials of early social communication intervention. STUDY REGISTRATION: This study is registered as PROSPERO CRD42012002223. FUNDING: The National Institute for Health Research Health Technology Assessment programme.
Authors: C H Chatham; K I Taylor; T Charman; X Liogier D'ardhuy; E Eule; A Fedele; A Y Hardan; E Loth; L Murtagh; M Del Valle Rubido; A San Jose Caceres; J Sevigny; L Sikich; L Snyder; J E Tillmann; P E Ventola; K L Walton-Bowen; P P Wang; T Willgoss; F Bolognani Journal: Autism Res Date: 2017-09-21 Impact factor: 5.216
Authors: Oliver D Howes; Maria Rogdaki; James L Findon; Robert H Wichers; Tony Charman; Bryan H King; Eva Loth; Gráinne M McAlonan; James T McCracken; Jeremy R Parr; Carol Povey; Paramala Santosh; Simon Wallace; Emily Simonoff; Declan G Murphy Journal: J Psychopharmacol Date: 2017-12-14 Impact factor: 4.153
Authors: B Di Rezze; E Duku; P Szatmari; J Volden; S Georgiades; L Zwaigenbaum; I M Smith; T Vaillancourt; T A Bennett; M Elsabbagh; A Thompson; W J Ungar; C Waddell Journal: J Autism Dev Disord Date: 2019-11
Authors: Rebecca Grzadzinski; Themba Carr; Costanza Colombi; Kelly McGuire; Sarah Dufek; Andrew Pickles; Catherine Lord Journal: J Autism Dev Disord Date: 2016-07