Some parts of this manuscript have been reproduced from Grahame et al.1 This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) license, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.
Terminology used in the report
In this report (and our published trial protocol2) we use identity-first language to describe autistic people, in line not only with the preferences of many English-speaking autistic people, but also argued by many to be less stigmatising than the alternative, person-first language.3,4 In addition, we use a novel term ‘functionally impactful RRB’ to describe a subset of behaviours under the umbrella of the Restricted, Repetitive and Stereotyped Patterns of Interests, Behaviours and Activities [restricted and repetitive behaviours (RRBs)] diagnostic domain which was targeted in the intervention being evaluated. This comes from a desire to refer to a group of RRB that have detrimental consequences for the child themselves, where those consequences are defined as far as we can with inclusion and autistic flourishing in mind. Previous reports from our team (e.g. the trial protocol2) used the term ‘challenging RRB’ for the same purpose, but we have changed this to reflect concerns that this terminology centres the parent/carer/practitioner perspective (the person facing the challenge) more than the child perspective.4 A shift to the concept of functional impact focuses more precisely on why these RRB can be considered valid intervention targets.
Restricted and repetitive behaviours such as repetitive movements, rigid routines, unusual preoccupations, circumscribed interests, resistance to change and sensory sensitivities form one of two key symptom domains required for a diagnosis of autism spectrum disorder (ASD), hereafter referred to as autism.5 While not exclusive to autism and occurring in typical development, as well as in children with learning disabilities, research consistently indicates increased frequency, duration, range and intensity of RRB in autistic children.6 Frequently, RRBs are reported by autistic adults to be enjoyable, functional and helpful. RRBs can be a source of great pleasure, both in the physical domain (i.e. the positive experience of repetitive movement or ‘stimming’) and as an expression of passionate interest (e.g. collections, hobbies). They may provide a basis for friendship, for example, when meeting people with a shared interest, or within a fandom. They can also build areas of strength, supporting skill development and yielding employment opportunities.7,8 For these reasons, and in line with the non-normalising agenda of neurodiversity, the default pathologisation of RRBs should be resisted.
However, RRB may also be an outward sign of anxiety or distress, deserving of attention and care from others.9 We know that families can find understanding this repertoire of behaviours particularly difficult, with parents spontaneously identifying RRB as the most difficult aspect of parenting their autistic child.10 High rates of RRB can also be stigmatising11 and lead to agitation or aggression, if interrupted12 RRB can interfere with family functioning, and can provoke negative parenting styles.8,12 In the last 5–10 years, there is emerging evidence for the immediate and longer-term benefit of interventions to support parents’ early interactions with their young autistic child, for example, promoting their responses to their autistic child’s social and communication development – the other key behavioural domain required for a diagnosis.13 However, at the time this study started, there are no evidence-based interventions available that focus on supporting parents to understand and respond to their child’s RRB. ‘At home’ activities are set each week for parents/carers and children to complete between sessions.
Restricted and repetitive behaviours are a core symptom domain for a diagnosis of ASD and thus RRBs are present in all autistic individuals. RRB can take a variety of forms and vary in the kind and degree of impact. Narrowness of focus, inflexibility, perseveration of interest in activities and insistence on sameness reflect the restricted aspect of this domain, while repetition is demonstrated in repetitive speech, routines, rituals and rhythmic stereotypies.14 For parents, RRB can be extremely difficult to understand and as a result it can be difficult to know how to support their child. For example, a family may avoid taking their 9-year-old to the shops if every time they go into the supermarket the child responds to the sensory experiences of the lights and the presence of other people with disruptive spinning and screaming. The reduction in autonomy consequent on this kind of daily limitation can negatively impact the child directly, via a reduction of their opportunities to experience the world, and indirectly via parent stress and parenting choices.
We have on reflection, and taking into account feedback from clinicians who delivered the programme and members of our dissemination advisory group, changed the name of our programme to Understanding Repetitive Behaviours (URB). This better reflects the neurodiversity affirming approach of the programme and the research and clinical team.
Developmental origins of restricted and repetitive behaviour
Restricted and repetitive behaviours are not exclusive to autism; they are also seen within typical and delayed development. A range of adaptive functions of RRB are widely recognised.15 Indeed, Thelen16 identified that stereotyped behaviours in the early years contribute to neuromuscular development and the acquisition of skilled motor actions. Repeating certain actions with toys and uttering the same sounds or words over and over again are considered vital for developmental progress.16 For example, seemingly obsessional interests such as dinosaurs can help children develop attention to detail and language skills. In typically developing children, we often see a reduction in RRB at around 3–4 years of age.17 However, although these early behaviours are similar for autistic children, the frequency and types of RRB they display vary according to age and cognitive ability and do not necessarily decline over time as children mature.
A lack of typical decline in frequency of RRB as autistic children grow up is not, in and of itself, a problem. However, RRB can spiral into harmful and extreme patterns of behaviour, sometimes also triggering a continuous cycle of action and response between parent/carer and child. For example, a widening gap between the autistic child’s behaviour and that of their non-autistic peers or siblings might mean parents/carers or other adults in the child’s life try to limit the child’s access to their preferred RRB. This inadvertently can lead to more effortful control from the child on their environment with the risk that the RRB becomes entrenched and the child even less willing, for example, to try new foods or access new experiences. In addition, as children grow older increasing social, academic and sensory demands in their environment, together with the inevitable transitions from home to nursery or nursery to school, expose the child and family to new experiences likely to disrupt existing routines. Alongside these developmental shifts, a range of sensory reactivities can be associated with repetitive and restricted patterns of behaviour, as when children self-restrict their diet to food with specific colours or textures. All these different situations require people around the autistic child – especially their parents/carers – to understand and respond to their child’s RRB in ways that promote understanding and facilitate autonomy and development.
In young children (aged approximately 3–9 years) irrespective of whether their neurodevelopmental progress is typical or atypical, there is a period of rapid brain maturational growth (during a time of plasticity/rapid learning and change).18 This period therefore presents an ideal window of opportunity to assist parents/carers to learn how to understand, anticipate and redirect their child’s emerging functionally impactful RRB, before they become entrenched and limiting. For all these reasons, it is important to support parents/carers of young autistic children to understand how to manage functionally impactful RRB and so help their child develop a more flexible range of behaviours and constructive ways to deal with the demands of their world.
The role of the family system
There is emerging evidence for the benefit of parent/carer-based interventions in young autistic children, although to date this research has focused on social communication interventions.19–21 However, parent/carer mediated programmes may be an especially appropriate intervention modality for RRB. Parents/carers need to know that many RRB might be ignored, or cultivated, as a mechanism of self-regulation and a source of enjoyment. In the case of RRB with serious immediate or long-term negative consequences, carefully implemented limit-setting, interruption or redirection may be appropriate. Functional analysis training (a core component of Understanding Repetitive Behaviours; URB) can support parents/families to begin to understand the environmental triggers and purposes of their child’s RRB. They can make use of this understanding to consider if, when, where and how to help their child develop alternative strategies and techniques to manage negative experiences across a range of everyday contexts.
We propose that there are three key stages/steps that need to be incorporated into an intervention to help parents/carers recognise, understand and manage their child’s functionally impactful RRB. These steps are included in the URB intervention.
- Help parents/carers understand why their child may engage in a particular behaviour and, in particular, to identify which of those behaviours are outward signs of distress and/or are most likely to have a functional impact.
- Enable parents/carers to recognise and understand the way in which their child’s RRB may be an outward expression of distress in response to situational stressors, such as anxiety related to change, or sensory reactivity. These relationships will be mediated by the child’s social communication difficulties (the other key diagnostic domain for ASD) and cognitive ability.
- Support parents/carers to identify and apply developmentally appropriate strategies for responding to selected, functionally impactful RRB, including by adjusting or limiting situational stressors. Given the important function that even functionally impactful RRB may have for autistic children, as a way to manage acute stress and distress, it is critical that any intervention which aims to reduce the consequences for the child of these functionally impactful RRB provides opportunities for the development of an extended repertoire of positive coping strategies to deal with the stressors that may trigger a particular RRB.
The URB intervention aims to work with and alongside parents/carers rather than directly with the young child, as parents/carers have unique in-depth knowledge of their child’s development and behaviours across a range of settings, and how the child’s everyday environment and experiences might be adapted. For example, by providing parents with information and understanding about RRB they may, with practice, be able to reduce background anxiety or support their child to extend their behavioural repertoire in different contexts. Furthermore, we anticipate that providing parents/carers with the skills to understand their child’s functionally impactful RRB will have a beneficial effect on parental well-being and family resilience by increasing their sense of competence, reducing stress and improving family cohesion. This in turn will have benefits for the child.
To our knowledge, this study is the first large-scale randomised controlled trial (RCT) of a parent/carer group intervention focused on functionally impactful RRB in autistic young children.22 To date, the majority of the research interventions for RRB come from single case behavioural studies and while these small-scale studies offer some early emerging evidence of success, limitations include implementation at specialised centres, and highly individualised delivery with expert clinicians.6
The URB intervention, if found to be effective, has the potential to extend the range of early interventions available to meet the needs of young autistic children and their families, ensuring the best use of therapeutic resources and reducing the risk that functionally impactful RRB may cause serious harm. Effective early interventions for young autistic children will reduce the longer-term requirement for more specialist services, increasing families’ independence from services and thus lowering (NHS) costs.23 There is increasing impetus among families, professionals, NHS priorities and commissioning documents for research findings on effective and cost-effective early interventions to inform practice, and for effective interventions to be incorporated into the range of available services for young autistic children and their families.
The Autistica/James Lind Alliance24 identified support for parents/carers to care for and understand their autistic child as amongst the top 10 research priorities for autism research in the future. The National Institute for Health and Care Excellence (NICE)25 acknowledged the encouraging emerging, but limited evidence base, for young autistic children targeting social communication, but highlighted the absence of specific treatments for young children with functionally impactful RRB that can be so disruptive of individual development and family life. Parent-mediated interventions have the unique advantage of enabling parents/carers to capitalise on opportunities in everyday situations to provide learning experiences. Building on these real-world opportunities may also facilitate generalisation of the child’s learning across contexts. Achieving functional change in the context of the child’s life and everyday experiences is a priority for the autism community and a key challenge for autism intervention research.
Aims and objectives
The aim of this study is to build on our successfully completed feasibility and acceptability trial22,26 by evaluating the clinical and cost-effectiveness of the URB parent/carer group intervention compared with a psychoeducation parent/carer group (equivalent to current best practice), in a fully powered two-arm, assessor-blinded, multicentre, individually RCT. Our feasibility and acceptability study showed that parents/carers were willing to be recruited and randomised, and that the format and content of the URB group and the research procedures were feasible and acceptable. Recruitment and retention rates were encouraging: 89% retention (from baseline to final 6-month follow-up assessment), with an attendance rate of 90% at the parent/carer group sessions. The pilot study also examined variability in the proposed outcome measures and provided preliminary evidence of treatment effect.22 However, given that the feasibility and acceptability study was a single-centre study, we have adopted conservative assumptions throughout as we are mindful that success in a single-centre study may be hard to replicate in a multicentre study.
We predict that supporting parents/carers to recognise, understand and learn to respond to their child’s functionally impactful RRBs will result in a reduction in functionally impactful RRBs, and a downstream increase in children’s social participation and learning. Furthermore, working with parents/carers will have a beneficial effect on parent/carer well-being by increasing their sense of competence, reducing stress and improving family cohesion.
The trial objectives are to:
- Compare the clinical effectiveness of the URB intervention for NHS community clinical practice with psychoeducation for the management of functionally impactful RRB in autistic children at 24 and 52 weeks follow-up.
- Assess the cost-effectiveness and cost consequences of the URB intervention compared with an autism parent/carer psychoeducation group at 52 weeks follow-up.