Interventions based on early intensive applied behaviour analysis for autistic children: a systematic review and cost-effectiveness analysis

Abstract

Background Early intensive applied behaviour analysis-based interventions are intensive interventions for autistic children that are often delivered on a one-to-one basis for 20–50 hours per week. Objectives To evaluate the clinical effectiveness and cost-effectiveness of early intensive applied behaviour analysis-based interventions for autistic children, based on current evidence. Methods A systematic review and individual participant data meta-analysis were conducted to evaluate the clinical effectiveness of an early intensive applied behaviour analysis-based intervention for autistic children. An economic analysis included a review of existing analyses and the development of a new model. Results Twenty studies were included in the clinical review. Individual participant data were retrieved from 15 of these studies. Results favoured the interventions when assessing adaptive behaviour after 2 years compared with treatment as usual/eclectic interventions (mean difference 7.00, 95% confidence interval 1.95 to 12.06). In analyses of cognitive ability (intelligence quotient), results favoured the interventions by approximately 10 points after 1 year (mean difference 9.16, 95% confidence interval 4.38 to 13.93) and 2 years (mean difference 14.13, 95% confidence interval 9.16 to 19.10). Evidence for other outcomes was limited and meta-analyses were generally inconclusive. There was no evidence that the effect of the interventions varied with characteristics of the children, but data were limited. Adopting a £30,000 per quality-adjusted life-year threshold, the results of the cost-effectiveness analysis indicate that early intensive applied behaviour analysis-based interventions would need to generate larger benefits or cost savings to be cost-effective. Adopting a public sector perspective and making pessimistic assumptions about long-term effects, the incremental cost-effectiveness ratio for early intensive applied behaviour analysis-based therapy compared with treatment as usual is £189,122 per quality-adjusted life-year. When optimistic assumptions are made, the incremental cost-effectiveness ratio is £46,768 per quality-adjusted life-year. Scenario analyses indicated that these interventions can potentially be cost-effective if long-term improvements persist into adulthood, or if they have significant impact on educational placement. Care should be taken when interpreting these scenarios owing to the limited data. Limitations All included studies were at risk of bias, there was substantial heterogeneity and effects varied considerably across studies. The effect of intervention on autism symptom severity, language development and school placement remains uncertain because of the limited data. The long-term effects are unclear owing to a lack of follow-up data. Conclusions This review found limited evidence that early intensive applied behaviour analysis-based interventions may improve cognitive ability and adaptive behaviour, but the long-term impact of the interventions remains unknown. The economic analysis is constrained by the limited effectiveness evidence, but suggests that these interventions are unlikely to be cost-effective unless clear long-term benefits, or a substantial change in which schools children attend, can be identified. Future work Further studies into the effectiveness of early intensive applied behaviour analysis-based interventions may be warranted if they include well-defined, alternative interventions as comparators and collect relevant outcomes. Consideration should be given to future studies that not only address whether or not early intensive applied behaviour analysis-based interventions are clinically effective, but also aim to identify which components of early intensive applied behaviour analysis-based interventions might drive effectiveness. Study registration This study is registered as PROSPERO CRD42017068303. Funding This project was funded by the National Institute for Health Research (NIHR) Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 24, No. 35. See the NIHR Journals Library website for further project information.

Association of Child and Family Attributes With Outcomes in Children With Autism

“…Recent longitudinal follow-up studies in childhood and adolescence have taken a person-centered approach to focus on individual differences in development in people with ASD 8 , 9 or else have used intervention studies to measure relatively short-term outcomes. 10 Indeed, much of the literature in this area takes a deficit-based measurement approach of good, best, or optimal outcomes that focuses on an absence of intellectual disability and no longer meeting criteria for ASD. 11 , 12 While this approach has some face validity and has proved enlightening, a more nuanced and holistic approach might provide a more complete picture of outcomes generally (as argued by Mason et al 7 ).…”

Early Diagnostics and Early Intervention in Neurodevelopmental Disorders—Age-Dependent Challenges and Opportunities

“…The large majority of studies on the effect of early intervention has been performed in children diagnosed with ASD, implying that the children’s average age was at least 2.5 years of age. Recent systematic reviews on children of at least 2.5 years concluded that most studies suffered from a high risk of bias [ 169 , 170 , 171 , 172 , 173 ]. This was particularly true for the studies evaluating behavioral approaches.…”

“…This was particularly true for the studies evaluating behavioral approaches. Consequentially, their reports of a beneficial intervention effect on the child’s adaptive behavior and cognition should be interpreted with caution [ 170 , 171 , 173 ]. The limited evidence available on the effect of early intervention in ASD indicates that interventions using a developmental or a combined developmental and behavioral approach may have a positive effect on the child’s play and social communication [ 170 , 172 ].…”

“…Overall, this means—as the recent systematic reviews concluded [ 169 , 170 , 171 , 172 , 173 ]—that studies on the effect of early intervention in ASD with good methodological quality are urgently needed. For the time being, interventions with a developmental approach with or without behavioral components are most promising to have a beneficial effect on social communication.…”

Commentary: What conflicts of interest tell us about autism intervention research—a commentary on Bottema‐Beutel et al. (2020)

“…This omission contrasts with the vast amounts of public and private resources invested in early behavioral and developmental autism interventions. These high‐profile interventions have high costs, whose benefits to providers have attracted equity funds (See https://www.blackstone.com/the-firm/press-releases/article/blackstone-to-acquire-center-for-autism-and-related-disorders-(card)), yet their benefits and harms to autistics remain uncertain at best due to the poverty of existing evidence (Green & Garg, 2018; Rodgers et al, 2020). A recent systematic review of early autism interventions applied at least some basic standards in evaluating evidence and found, in the entire literature, only six (12.5% of the total) RCTs at low risk of bias (French & Kennedy, 2018).…”