A clinical and cost-effectiveness trial of a parent group intervention to manage challenging restricted and repetitive behaviours in young children with autism spectrum disorder: study protocol for a randomised controlled trial

Victoria Grahame,Magdalena Glod,Ehsan Kharatikoopaei,Emma Honey,Saoirse Heron,Ann Le Couteur,Christopher Weetman,Faye Wolstenhulme,Priyanka Rob,Adetayo Kasim ,Rebecca Iversen,Linda Dixon,Deborah M Riby ,Sarah L Thompson ,Jane Goodwin,Elspeth Imogen Webb,Ruth Wood,Eamonn Walls,Sue Fletcher‐Watson ,Ashleigh Kernohan,Leanne Rogan,Leila Mackie,Ayesha Mathias,Deborah Garland,Zoe Grayson,Luke Vale,Helen Probert,Jacqui Rodgers

doi:10.1186/s13063-021-05175-y

Abstract

BackgroundRestricted and repetitive behaviours vary greatly across the autism spectrum, and although not all are problematic some can cause distress and interfere with learning and social opportunities. We have, alongside parents, developed a parent group based intervention for families of young children with autism, which aims to offer support to parents and carers; helping them to recognise, understand and learn how to respond to their child’s challenging restricted repetitive behaviours.MethodsThe study is a clinical and cost-effectiveness, multi-site randomised controlled trial of the Managing Repetitive Behaviours (MRB) parent group intervention versus a psychoeducation parent group Learning About Autism (LAA) (n = 250; 125 intervention/125 psychoeducation; ~ 83/site) for parents of young children aged 3–9 years 11 months with a diagnosis of autism.All analyses will be done under intention-to-treat principle. The primary outcome at 24 weeks will use generalised estimating equation (GEE) to compare proportion of children with improved RRB between the MRB group and the LAA group. The GEE model will account for the clustering of children by parent groups using exchangeable working correlation. All secondary outcomes will be analysed in a similar way using appropriate distribution and link function.The economic evaluation will be conducted from the perspective of both NHS costs and family access to local community services. A ‘within trial’ cost-effectiveness analysis with results reported as the incremental cost per additional child achieving at least the target improvement in CGI-I scale at 24 weeks.DiscussionThis is an efficacy trial to investigate the clinical and cost-effectiveness of a parent group based intervention designed to help parents understand and manage their child’s challenging RRB. If found to be effective, this intervention has the potential to improve the well-being of children and their families, reduce parental stress, greatly enhance community participation and potential for learning, and improve longer-term outcomes.Trial registrationTrial ID: ISRCTN15550611 Date registered: 07/08/2018. Sponsor and Monitor: Cumbria, Northumberland, Tyne and Wear NHS Foundation Trust R&D Manager Lyndsey Dixon, Address: St Nicholas Hospital, Jubliee Road, Gosforth, Newcastle upon Tyne NE3 3XT, lyndsey.dixon@cntw.nhs.uk, Tel: 0191 246 7222

Highlights

Autism spectrum disorder is a lifelong neurodevelopmental condition affecting 1–2% of the population, with profound impact on diagnosed individuals, families, and society [1, 2]
Study aims The aim of this study is to evaluate the clinical and costeffectiveness of the Managing Repetitive Behaviours (MRB) parent group intervention for young children with autism compared with a parent autism psychoeducation group, at 10, 24 and 52 weeks follow-up
This study will evaluate the clinical and costeffectiveness of the Managing Repetitive Behaviours (MRB) parent group intervention compared with the Learning About Autism psychoeducation parent group

Summary

Introduction

Background Autism spectrum disorder (hereafter, ‘autism’) is a lifelong neurodevelopmental condition affecting 1–2% of the population, with profound impact on diagnosed individuals, families, and society [1, 2]. Restricted and repetitive behaviours (RRB) are one of two key behavioural domains required for a diagnosis of autism [3]. They include repetitive motor mannerisms, rigid adherence to specific routines, highly circumscribed interests and extreme responses to everyday sensory experiences. Some RRBs are reported by autistic adults to be enjoyable, functional and helpful They may provide a basis for friendship and can build areas of strength, supporting skill development and yielding employment opportunities [4, 5]. Alongside parents, developed a parent group based intervention for families of young children with autism, which aims to offer support to parents and carers; helping them to recognise, understand and learn how to respond to their child’s challenging restricted repetitive behaviours

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