Clinical and cost-effectiveness of an adapted intervention for preschoolers with moderate to severe intellectual disabilities displaying behaviours that challenge: the EPICC-ID RCT

Ondruskova T, Royston R, Absoud M, Ambler G, Qu C, Barnes J, Hunter R, Panca M, Kyriakopoulos M, Oulton K, Paliokosta E, Narain Sharma A, Slonims V, Summerson U, Sutcliffe A, Thomas M, Dhandapani B, Leonard H, Hassiotis A
Record ID 32018005582
Authors' objectives: Stepping Stones Triple P is an adapted intervention for parents of young children with developmental disabilities who display behaviours that challenge, aiming at teaching positive parenting techniques and promoting a positive parent–child relationship. To evaluate the clinical and cost-effectiveness of level 4 Stepping Stones Triple P in reducing behaviours that challenge in children with moderate to severe intellectual disabilities. Intellectual disability is a lifelong condition impairing an individual’s intellectual and adaptive functioning, affecting approximately 1.2 million children, young people and adults in England. Between 10% and 45% of children with intellectual disability display behaviours that challenge, including self-injury, aggression, destructiveness and stereotypical behaviours. These behaviours can be very distressing for both the parent and the child, and parents may find them difficult to manage. Interventions for early-onset conduct problems and disruptive behaviour in the general population are known to reduce such behaviours, improve long-term outcomes and reduce care costs. Early interventions are often delivered through group parenting programmes, which are known to increase parent efficacy through learning positive parenting techniques and contingency management strategies within a social learning framework. One such intervention, adapted for children with intellectual disability and socio-emotional disabilities, is Stepping Stones Triple P (SSTP). The SSTP programme combines psycho-educational and behavioural components, which aim to promote a positive parent–child relationship. The intervention also encourages the development of children’s skills within everyday parenting situations, for example during mealtimes, bathing or dressing. Studies outside the UK have shown that SSTP is effective, acceptable to parents, reduces behaviours that challenge and improves parenting styles. The current study (EPICC-ID) describes a randomised multicentre evaluation of level 4 group SSTP in very young children with moderate to severe intellectual disability. To our knowledge, it is the first study to test such an intervention in this population group in the UK (England). Objectives To undertake a pragmatic randomised controlled trial to evaluate level 4 group SSTP in addition to treatment as usual (TAU); To undertake an economic evaluation to assess the cost-effectiveness of the intervention compared to TAU.
Authors' results and conclusions: We found a small non-significant difference in the mean Child Behaviour Checklist scores (−4.23, 95% CI −9.98 to 1.52, p = 0.146) in the intervention arm compared to treatment as usual at 12 months. Per protocol and complier average causal effect sensitivity analyses, which took into consideration the number of sessions attended, showed the Child Behaviour Checklist mean score difference at 12 months was lower in the intervention arm by −10.77 (95% CI −19.12 to −2.42, p = 0.014) and −11.53 (95% CI −26.97 to 3.91, p = 0.143), respectively. The Child Behaviour Checklist mean score difference between participants who were recruited before and after the coronavirus disease 2019 pandemic was estimated as −7.12 (95% CI −13.44 to −0.81) and 7.61 (95% CI −5.43 to 20.64), respectively (p = 0.046), suggesting that any effect pre-pandemic may have reversed during the pandemic. There were no differences in all secondary measures. Stepping Stones Triple P is probably value for money to deliver (−£1057.88; 95% CI −£3218.6 to −£46.67), but decisions to roll this out as an alternative to existing parenting interventions or treatment as usual may be dependent on policymaker willingness to invest in early interventions to reduce behaviours that challenge. Parents reported the intervention boosted their confidence and skills, and the group format enabled them to learn from others and benefit from peer support. There were 20 serious adverse events reported during the study, but none were associated with the intervention. Level 4 Stepping Stones Triple P did not reduce early onset behaviours that challenge in very young children with moderate to severe intellectual disabilities. However, there was an effect on child behaviours for those who received a sufficient dose of the intervention. There is a high probability of Stepping Stones Triple P being at least cost neutral and therefore worth considering as an early therapeutic option given the long-term consequences of behaviours that challenge on people and their social networks. Clinical effectiveness Our primary analysis was based on intention-to-treat in which we adjusted for baseline CBCL total score, centre, level of intellectual disability and therapist clustering, showed a mean difference between arms of −4.23 [95% confidence interval (CI) −9.98 to 1.52, p = 0.146]. We found that SSTP, as delivered in this trial, did not reduce behaviours that challenge compared to TAU at 12 months post-randomisation. Our initial sample size estimation was predicated on a minimal clinically significant difference of eight points between the two study arms. Of the 155 patients who were randomised to the SSTP arm, 50 participants were adherent to the SSTP intervention, meaning they attended at least 4 (out of 6) group sessions and 2 (out of 3) individual sessions. We carried out a per-protocol analysis which excluded non-adherent participants; we found that the intervention effect at 12 months was −10.77 (95% CI −19.12 to −2.42, p = 0.014). We also carried out a complier-average causal effect (CACE) analysis to measure the effect of the intervention on CBCL total scores at 12 months. We found a reduction of −11.53 (95% CI −26.97 to 3.91, p = 0.143) compared to TAU. We further performed a subgroup analysis to investigate whether the effect of SSTP differed depending on whether recruitment was before or after 16 March 2020. In this model, the mean difference of the effect of SSTP on CBCL total scores at 12 months was estimated as −7.12 (95% CI −13.44 to −0.81) and 7.61 (95% CI −5.43 to 20.64), respectively, with a p = 0.046. This suggests that the effect of SSTP was different before and during the pandemic. The point estimates suggest the direction of effect may have reversed during the pandemic. There were no statistically significant differences between arms in any of the secondary outcome measures. However, we noted a reduction in negative child behaviours as shown in observations of parent–child interaction. A total of 20 serious adverse events were reported, with 12 in the SSTP and 8 in the TAU arms. Of these, 13 were reported for children and 7 for parents. None of these were determined to be related to the intervention. The main statistical analysis did not reveal any statistical differences in mean CBCL scores between the intervention arms, suggesting that SSTP at 12 months is not effective compared with TAU. However, the sensitivity analyses showed that those receiving the intervention experienced a positive, albeit non-statistically significant change in the child’s behaviours of concern (reduction). Parents reported that the intervention boosted their confidence and skills, and the group format enabled them to learn from others and receive peer support. Overall, the findings suggest the intervention has clinical utility and should be available to underserved children who are more likely to have long-term adverse consequences due to the early onset of behaviours that challenge. Further, SSTP appears to be cost-effective and well within the NICE threshold for cost-effectiveness at £20,000–30,000 and at the lower cost of £13,000. Therefore, there are indications the intervention may be beneficial under certain conditions and can be delivered within NHS care. Further research is needed to explore and find solutions to the implementation of parenting groups for behaviours that challenge in this underserved population, as well as the optimal mode of delivery to maximise engagement and outcomes.
Authors' methods: A parallel two-arm pragmatic multisite single-blind randomised controlled trial recruited a total of 261 dyads (parent and child). The children were aged 30–59 months and had moderate to severe intellectual disabilities. Participants were randomised, using a 3 : 2 allocation ratio, into the intervention arm (Stepping Stones Triple P; n = 155) or treatment as usual arm (n = 106). Participants were recruited from four study sites in Blackpool, North and South London and Newcastle. There were low attendance rates in the Stepping Stones Triple P arm, as well as the coronavirus disease 2019-related challenges with recruitment and delivery of the intervention. The current study was a pragmatic parallel two-armed multisite single-blind randomised control trial with a 3 : 2 randomisation ratio (SSTP vs. TAU). The chief investigator, researchers and the lead statistician were blinded to participant allocation. Altogether, 261 dyads (parent with index child) were enrolled in this trial, of whom 155 were allocated to the SSTP and TAU arm and 106 were allocated to the TAU arm alone. The inclusion criteria were (1) to be a parent aged 18 years or over, (2) consenting to take part, (3) having a child with moderate to severe intellectual disability, (4) the child to be aged 30–59 months at identification and (5) the child to display behaviours that challenge as reported by a parent over a 6-month period prior to the study. The participant was excluded if the child had mild, profound or no intellectual disability, if a sibling was participating in the study, or if the parent had insufficient English language skills to complete or understand the study questionnaires. Participants were recruited from various community services including Participant Identification Centres in four main areas in England: North West of England (Blackpool, Site 1), North London (Site 2), South London (Site 3) and North East of England (Newcastle, Site 4). The primary outcome measure was the parent-reported Child Behaviour Checklist (CBCL). We also assessed secondary outcomes using parent–child observations, other caregiver/teacher reports, questionnaires of parents’ mental health, stress, sense of competence and parent and child health-related quality of life. We further conducted a process evaluation using a mixed methods approach to assess intervention delivery (fidelity, dose, adaptations, reach) and to capture the views of the participants, therapists and service managers. The study was ethically reviewed and approved by the London – Camden and Kings Cross Research Ethics Committee (reference: 17/LO/0659). The last 18 months of the trial took place during the coronavirus disease 2019 (COVID-19) pandemic. Fifty-one out of 261 families were randomised after 16 March 2020 (i.e. the beginning of the pandemic) with 219 baseline and follow-up assessments carried out from that date to the end of the study (the last participant follow-up was completed in December 2021). We made changes to the study to comply with the public health measures implemented by the UK government. This ensured participant and researcher safety and allowed us to safeguard the study validity and quantify, where possible, the impact of this event. After the start of the pandemic, all study procedures, for example obtaining consent, carrying out assessments and delivering the intervention, were carried out remotely. We also adjusted the a priori statistical and health economic analysis plans to account for these changes. We were unable to continue carrying out behavioural observations and completing cognitive assessments with the children, as techniques for doing so remotely were unavailable at the time. Stepping Stones Triple P In the EPICC-ID study, we delivered a manualised level 4 SSTP intervention composed of six group sessions and three individual telephone or face-to-face contacts with the parent over a period of 9 weeks. Each group session lasted approximately 2.5 hours. Individual sessions took around 30 minutes. SSTP has the most evidence for efficacy, and while available in the UK via Triple P UK, it has not been formally tested for its clinical and cost-effectiveness and is not rolled out in the National Health Service or a part of the local offer (resources available from Local Authorities for children with disabilities). The group sessions were delivered in person until March 2020 and on the online platforms zoom and Microsoft Teams thereafter. Parents allocated to both arms also received a list of national resources and the Contact (a Family) charity guide for managing behaviours that challenge, which included signposting to social and health care support.
Project Status: Completed
Year Published: 2024
URL for additional information: English
English language abstract: An English language summary is available
Publication Type: Full HTA
Country: England, United Kingdom
MeSH Terms
  • Intellectual Disability
  • Developmental Disabilities
  • Parenting
  • Child, Preschool
  • Child
  • Child Behavior Disorders
  • Cost-Effectiveness Analysis
Organisation Name: NIHR Health Technology Assessment programme
Contact Address: NIHR Journals Library, National Institute for Health and Care Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK
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