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

Study design

A parallel two-armed pragmatic multisite single-blind randomised control trial (RCT) was conducted with blinding of outcome assessors. A process evaluation was also included utilising parent and service manager qualitative interviews to enhance understanding of the appropriateness and feasibility of the intervention. The study was planned and implemented following the Consolidated Standards of Reporting Trials (CONSORT) extension to compare the cost-effectiveness of the combination of SSTP plus TAU, versus TAU alone in reducing behaviours that challenge at 12 months post randomisation. The trial design is summarised in Figure 1.

FIGURE 1.

The CONSORT flow diagram for the EPICC-ID RCT.

Randomisation

Participants were randomised into the intervention arm using the 3 : 2 ratio (SSTP vs. TAU) using randomly permuted blocks of different block sizes and stratification by site and the level of intellectual disability (moderate and severe). Eligible participants were allocated online to the next available treatment code in the relevant randomisation list. All randomisation and data management were provided by an internet-based site called Sealed Envelope.

Allocation concealment and implementation

At the end of the baseline assessment, the researchers entered the results on a web-based case report form (CRF) form. Parents and therapists were informed about the allocation status and arranged the commencing of the group sessions. Researchers were based in a different location from the staff involved in the delivery of the level 4 SSTP. Therapists were not involved in any treatment of the families that were allocated to TAU.

Blinding

Since parents and therapists were aware of the treatment allocation, it was not possible to ensure a completely blind trial. All research assistants who were collecting the data remained blinded to treatment allocation throughout the study. Parents were reminded not to disclose any information about their treatment allocation to the research assistant during assessments.

The researcher entered the results of all study assessments on web-based electronic case report forms (eCRFs). They did not have access to eCRFs that may pose a risk of unblinding them [e.g. Child and Adolescent Service Use Schedule (CA-SUS) at 4 and 12 months, Client Satisfaction Questionnaire, serious adverse event (SAE) form]. During coronavirus disease 2019 (COVID-19), CA-SUS forms were collected remotely, and the question relating to parenting groups was omitted and collected later.

The lead study statistician remained blinded to arm allocation throughout and the subsidiary trial statistician attended the Data Safety and Monitoring Board (DSMB) meetings. The lead statistician attended the Trial Management Group (TMG) and Trial Steering Committee (TSC) meetings during the study.

Primary outcome

Secondary outcomes

Other measures

Sample size

The original sample size calculation required a sample of 258 children in an allocation ratio of 3 : 2 (155 vs. 103) to detect a low to a moderate (standardised) effect size of 0.40 for the primary outcome CBCL at 12 months at the 5% significance level with 90% power; the equivalent to detecting a clinically important difference of 8 points, assuming a standard deviation (SD) of 20. This was calculated as follows: A standard calculation based on analysis of covariance (ANCOVA) leads to a sample size of 99 children per arm, assuming a correlation of 0.5 between baseline and follow-up measurements. An equivalent calculation (same power) based on an allocation ratio of 1.15 : 1 gives group sizes of 107 and 93. Increasing the SSTP arm (only) to allow for therapist clustering leads to 139 children in the SSTP arm, assuming an intraclass correlation of 0.05 and an average therapist group size of 7 (design effect = 1.3). An adjustment for the anticipated dropout of 10% leads to 155 children in the SSTP group and 103 in the control group.

Eligibility criteria

The inclusion criteria for the study were as follows:

1. parents aged 18 years or over;

2. child aged 30–59 months at identification;

3. child had a moderate to severe intellectual disability, measured by parent-reported Adaptive Behaviour Assessment Schedule (ABAS) General Adaptive Functioning score between 40 and 69;

4. reported child’s behaviours that challenge over a 6-month period prior to the study, but no less than 2 months;

5. obtain a written consent by parent/caregiver.

Exclusion criteria

Participants were not included in the study if:

1. child had a mild, profound or no intellectual disability on parent-reported ABAS form;

2. parent/carer had insufficient English language skills to complete the study questionnaires;

3. another sibling was taking part in the study.

Recruitment

Participants were recruited from various community services including Participant Identification Centres (PIC) in four main areas in England: Blackpool (and surrounding areas; Site 1), North London (Site 2), South London (Site 3) and Newcastle (and surrounding areas; Site 4). The services included National Health Services (NHS) settings (e.g. Child Development Teams), Child and Adolescent Mental Health Services (CAMHS), education (e.g. nursery, preschool) and third sector (e.g. caregiver groups). Health or social care professionals identified eligible participants through new referrals or existing cases. Children were assessed for developmental delay through the Healthy Child Programme, which aims to identify families that require further support with their children, and children aged 0–5 years who are at risk of ‘poor outcomes’. Flyers were put up at local parent groups, nurseries, special schools and general practitioners (GPs) to promote the study among families. We followed a multisource referral strategy facilitated by the clinical research networks, our national, clinical and third-sector contacts and social media. We opened five PIC adjacent to Site 4, three PIC sites adjacent to Site 2 and two PIC sites adjacent to Site 3 to reach our planned recruitment target at the sites. Several recruitment difficulties were addressed throughout the study. This included parents declining or not being eligible to take part in the study due to various reasons, such as time commitments, child-care issues, health problems, language barriers or parents not thinking that child has an intellectual disability or behaviours that challenge. Table 3 presents the overview of recruitment at each study site.

Intervention arm

SSTP is part of Triple P (Positive Parenting Program), a psycho-educational parenting and family intervention developed for families who have children with behavioural or emotional problems. 58 SSTP was adapted specifically for parents of children with intellectual disabilities. This trial provided the SSTP level 4 manualised course, which offers 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 and individual sessions took about 30 minutes. The combination of group and individual sessions allowed parents to share experiences and build on skills gained while providing personalised support. The group sessions aimed to educate and actively train parents in skills and behaviour management. The individual consultations aimed to facilitate independent problem-solving and allowed parents to pick strategies most relevant to their child’s difficulties. All parents also received a course book that covered topics from each session. Those who missed a session were contacted by the therapist to discuss their progress and were encouraged to attend the next sessions. The group sessions were delivered in person until March 2020. Afterwards, all group sessions were delivered online via Zoom or Microsoft Teams. Table 4 provides the outline of the SSTP sessions.

Treatment as usual arm

Treatment as usual was available to participants in both arms of the trial. Parents allocated to both arms also received a list of national resources and the Contact charity’s guide for managing behaviours that challenge with recommendations about social and health care support. We identified a variety of services and support offered to families with children with intellectual disabilities and autism in each of the study’s local areas. This included individual help from speech and language therapists, occupational therapists, child psychotherapists, clinical psychologists, child psychiatrists and family therapists. There were numerous children’s centres offering support with parenting advice, local child-care options and access to specialist services for families and their children with intellectual disabilities and parenting classes (e.g. Camden MOSAIC centre in Site 2; SLAM Child and Family Service in Site 3, etc.). Parenting programmes were available in each of the study local areas (N = 3 in Site1; N = 8 in Site 2; N = 9 in Site 3; N = 9 in Site 4). For example, the Early Bird parenting programme was available in all four study areas. This is a 3-month programme for parents providing general information about autism and advice about looking after children with autism. It combines group training sessions for parents with individual home visits where video feedback is used to help parents apply what they have learnt with their child. Parenting programmes that focus on conduct problems in children without an intellectual disability were also available, such as The Incredible Years (Site 2, Site 3 and Site 4), The Family Links Course (Site 2) and Triple P (Site 3). Other support included third-sector organisations, which provided wide-ranging help through workshops, home visits, child care, information and advice and time-limited interventions. Most of the available programmes focused on autism awareness and general information for parents about autism or intellectual disabilities, providing limited advice on parenting strategies and techniques for addressing behaviours that challenge displayed by disabled children.

Participant identification

The study population was parents of preschool children with moderate to severe intellectual disability, who were concerned about their child’s behaviour living in the community in the four study sites. We excluded children with mild intellectual disabilities, as they are likely to access other interventions more suitable for their needs. Similarly, children with profound intellectual disabilities were excluded as they were unlikely to be able to follow the observation tasks and the psychometric assessments. Eligible participants were identified by local community paediatric or CAMHS teams through new referrals or existing cases. Members of the clinical teams screened and reviewed identifiable personal information of potential participants. Parents who were approached by a member of clinical staff or a clinical study officer were given a description of the study, the study Patient Information Sheet and an Expression of Interest form to complete. Those who were eligible and interested in taking part completed the ‘Expression of Interest’ form, which was passed to the researchers, who then contacted the parents for further screening.

Screening process

The eligibility of each participant was confirmed during the screening assessment with a member of the research team. During the screening, the parent rated the child’s level of functional abilities on the ABAS form. Children who had a General Adaptive Functioning score between 40 and 69 were considered to have moderate to severe intellectual disability, and thus were eligible to participate. Parents also had to confirm that their child displayed behaviours that challenge continually over the 2 months prior to the screening assessment. If the result of the screening assessment indicated the child’s adaptive functioning to be outside of our inclusion criteria, parents were given the reason for non-inclusion, were thanked for their cooperation and time, and no further contact was made with the family. Those who were eligible after the screening assessment were contacted to schedule the baseline assessment. Following this, the participants were randomised into the intervention arm (SSTP plus TAU) or TAU.

Informed consent procedure

Informed consent was taken in two stages. First, the person who had parental responsibility for the child completed the consent form at the beginning of the screening assessment prior to completing the ABAS form. Second, if the child was eligible, the family was invited to enter the study and complete the consent form prior to their randomisation. Parents were given a minimum of 24 hours to decide whether they wanted to enter the study and were given contact details of the research assistant to ask further questions. For both stages, parents provided written or audio-recorded verbal informed consent. The right to withdraw at any time without giving a reason or affecting further treatment was explained to each participant. Each participant was given a copy of their informed consent form, and the signed original was retained at the study site.

The researchers were inducted into the study procedures about obtaining informed consent and completed the online Good Clinical Practice (GCP) course.

For the process evaluation, parents signed a consent form and were provided with a copy to keep if interviews were done in person. For telephone interviews, the consent process was audio-recorded and stored securely in Data Safe Haven (DSH), a secure online platform to store confidential data at University College London (UCL).

Data collection methods and handling

Data management in this study complied with the UK Data Protection Act 1998, PRIMENT Standard Operating Procedures (SOPs) and GCP. Researchers uploaded and stored all data into DSH, which is a secure system for storing sensitive information. Audio and video recordings were uploaded to the system, and researchers deleted all other versions of the data from digital machines where they were originally recorded.

The investigator was responsible for ensuring the accuracy of all data entered in the CRFs. Data verification checks were completed on 100% of CBCLs (primary outcome measure), and 10% of all secondary outcome measures. The original application stipulated that 5% of all secondary outcome measures would be checked, but due to researcher turnover, it was deemed necessary to carry out a higher percentage of checks for quality assurance in three study sites (Site 2, 3 and 4), where 10% checks were carried out. All study staff were also reminded of the importance of careful data entry to minimise errors.

Confidentiality

All personal data collected about the participant was managed following the PRIMENT SOP for Managing Personal Data. Each participant was given a unique identification number at randomisation. The participant’s initials, date of birth and unique randomisation number were used for identification. All other personal or identifiable information about the participants was stored separately and securely in the UCL DSH system. The CRFs did not contain identifiable information. We used a delegation log to track personnel responsible for data management.

Trial database

All data from the participants were entered into an online clinical data management system called Sealed Envelope. PRIMENT approved the quality management, software development and security of this system. The original paper forms of outcome measures were stored in secured locked cabinets in the office at each trial site. PRIMENT SOP Database Lock, Unlock and Closure was followed at the end of the trial.

Data entry

Baseline data for 261 randomised participants from all four sites were entered into the Sealed Envelope database. The primary outcome measure (CBCL) was available in the database for 261 participants and the CRFs for Source Data Verification (SDV) checking were available for all participants.

Four-month follow-up data for 229 participants from four sites have been entered into the database. The primary outcome measure was available for 223 participants.

Approval

Approval for the study was given by the London – Camden and Kings Cross REC (reference: 17/LO/0659).

Substantial amendments

Ethics and Health Research Authority (HRA) approval for the study was given on 19 May 2017. Overall, there were six substantial amendments approved by the REC as well as nine non-substantial amendments to the study protocol.

• Substantial amendment 1: added participant recruitment via third-sector organisations, such as ‘Contact’ (co-applicant) and added PIC sites to the study.

• Substantial amendment 2: extended the screening and baseline visit window from 2 to 4 weeks, approved giving the brochure and list of resources to all participants in both arms of the trial.

• Substantial amendment 3: included minor updates to the participant information sheets and the study poster, created treatment allocation letters for the participants and created a letter for clinicians, teachers or third-sector organisations to send the study information sheet and expression of interest form to potential participants.

• Substantial amendment 4: added an independent reviewer to code the video interviews, included the possibility of verbal consent and the possibility of conducting qualitative interviews for process evaluation over the phone, and added sections of the transparency wording from the HRA website in line with the new General Data Protection Regulation (GDPR), added intervention survey, created a letter for parents from the intervention group that did not attend any of the sessions, provided minor amendments to information sheets, consent forms and topic guide for interviewing service managers.

• Substantial amendment 5: approval to invite parents not taking part in the study to attend the SSTP groups to ensure viable group sizes, created a ‘Thank you’ card for parents to boost attendance at follow-up assessments, added a minor update to the information sheet.

• Substantial amendment 6: approved use of external transcription service to transcribe recorded audio files and to include some participants from the TAU group in qualitative interviews, including information regarding the 10-month costed extension approved by the study funders.

Deviations from the study protocol

There were 37 protocol deviations at 4 sites. Most of these deviations concerned baseline or follow-up assessments being completed outside of the assessment window as specified in the protocol. The assessment window between screening and baseline was extended to 4 weeks as part of substantial amendment 2. Other deviations included issues with the consent forms (e.g. a participant signed a consent form with the wrong site logo). These consent forms were corrected, or the correct forms were completed retrospectively. Protocol deviations relating to the COVID-19 pandemic are described later in the report under the heading ‘COVID-19 Adaptations’.

Recruitment during the COVID-19 pandemic

In total, 51 out of 261 families in the trial were randomised on or after 16 March 2020. The non-substantial amendments also simplified the process for Clinical Study Officers to pass on expressions of interest to the study team and updated the study protocol to reflect all changes. Study recruitment was halted between 18 March 2020 and 18 May 2020, and the timescale for re-opening was determined independently by each site. Sites reopened gradually between May and August 2020. Study follow-ups continued throughout this period, although child−parent observations and cognitive neuropsychological assessments (i.e. Mullen Scales of Early Learning)57 that required face-to-face contact were omitted from the study, as we were unable to find alternative ways to conduct the assessments (max n = 51). Wet signatures for logs were replaced with online signatures, and verbal recorded consent was obtained rather than written consent.

Observing government guidance on the spread of COVID-19 led to the study team working remotely. Before the pandemic, the CA-SUS forms at 4 and 12 months were completed by parents during visits and then handed to the Trial Manager for data entry. As this was no longer possible, research assistants completed the CA-SUS assessment over the phone but did not ask questions about attendance at parenting groups that would unblind them. These data were collected once the participant had completed the final follow-up. Moreover, the issues associated with the pandemic caused further difficulties with completing the study assessments with participants. For example, some parents found it challenging to find the time to complete all questionnaires over the phone, therefore many only completed the primary outcome measure (CBCL). Since all work, appointments, therapies and school responsibilities became remote, many assessments were re-scheduled and this increased the number of missing assessments and those completed outside of the assessment window.

The restrictions from the COVID-19 pandemic caused difficulties with obtaining the secondary outcome measure (C-TRF), which was meant to be completed by the child’s teacher. Most of the schools were closed and many parents did not have other relatives or people who would know their child’s behaviours well enough to complete this measure.

Parents from the PAG reflected on their own experiences during the pandemic, including big routine changes, which could affect the availability of parents to complete assessments. In particular, single parents may not have had the time to dedicate to the study. One parent reflected that during the pandemic situation, it was difficult to make phone calls without interruptions, but as the situation progressed, the family started to settle into a routine and this increased their capacity to take phone calls and allocate time to more responsibilities. They said this may also be similar for other parents. Another parent from the PAG noted that many of these families may not have education health care plans yet, which was allowing children to continue attending school during lockdowns. For those without a plan, routines are being disrupted, which may have been extremely difficult for both children and their families. Overall, the COVID-19 pandemic brought challenges not only to the delivery of SSTP and other services in TAU but also to the daily lives of every individual. This likely resulted in a deterioration in children’s behaviour patterns, increased parental stress and poorer parental ability to manage behaviours. These disruptions likely challenged many parents’ ability to comply with study directives.

Treatment as usual during the COVID-19 pandemic

Treatment as usual could not have been delivered in the conventional way as most of the services available in local areas were interrupted or completely suspended from March 2020. As such, families that were recruited in this study from this time had little to no help or support available to them.

During the qualitative interviews conducted for the process evaluation, parents shared their views about the COVID-19 pandemic and the national lockdowns. They described this time as extremely challenging as the children’s challenging behaviours worsened due to lack of routine and limited access to school. Parents were trying to balance working from home and caring for their child with no additional support from services or school. As such, parents felt even more abandoned during the times when they felt the most need to be supported.

The behaviour during the three months of lockdown took its toll. It escalated to where it was, like, unmanageable most days. Um, but there wasn’t a lot we could do about that.

One of the biggest challenges I’ve ever had to face is being in three months lockdown with a child with needs … It was really, really unfair to do I think, what they done originally, was said children who have EHCP plans would be taken care of. We’d be taken into consideration, the needs, and it wasn’t.

Stepping Stones Triple P delivery during the COVID-19 pandemic

From March 2020, the intervention was adapted to follow the COVID-19 safety precautions and regulations. At the start of the pandemic, no online version of SSTP was available from the Triple P group. The therapists continued with the same session plans and content that was delivered before the COVID-19 pandemic and delivered this remotely. The final group combined participants from all sites to ensure a sufficient group size. The Triple P held a panel discussion on remote delivery in practice on 21 July 2020. This was attended by the Trial Manager. The Triple P developers granted permission for us to share the advice provided in this session, including:

• Using video conferencing software, which allows for better participation than the telephone as parents can see the therapist when skills are being demonstrated.

• Allowing space for discussing progress and being able to problem-solve technical challenges.

• Acknowledging the stressors parents are facing and normalising the feelings they are experiencing. Offer hope that with the plans and strategies to be discussed, they will feel more confident in how to tackle this new normal, reduce stress and enjoy their parent–child interaction.

• Maintain contact with fellow Triple P practitioners, including Peer-Assisted Supervision and Support (PASS) sessions, to support each other in learning this new format and maintain fidelity.

• For parents with children at home, consider proposing that parents use a Planned Activities Routine to occupy the children so parents can participate fully in the session.

• Consider sending parents text reminders of their upcoming session.

• Resources can be available to pick up from an office (if open) or mailed to families.

• Consider planning how to use behavioural rehearsal based on your chosen delivery format (on videoconference platforms, it can be done as usual).

• When parents or practitioners are stressed or anxious, it may be more difficult to come up with ideas. Practitioners may need to think about giving more examples or offering more specific prompts sooner if they realise that parents are struggling with idea generation.

• Provide an opportunity for informal communication between parents before or after the sessions.

• Show DVDs by sharing your screen or demonstrate live the strategies. The DVD could also be played in the background, sharing the audio only (additional commentary may be required), or therapists should read the relevant section of the Positive Parenting Booklet.

• Consider if the parent has safe activities for the children while they are taking part in the intervention.

• Content can be fully delivered remotely, and the process/strategies should not change.

Data on attendance at parenting groups were collected once the participant has completed the study. Forms were sent to the Trial Manager for data entry.

Assessment of baseline characteristics

Summary measures for the baseline characteristics of each group are presented as mean and SDs for continuous, symmetric variables, medians and interquartile ranges (IQR) for continuous, skewed variables and frequencies and percentages for categorical variables. We compared baseline characteristics visually to assess whether the balance had been achieved. Any notable imbalances prompted additional adjusted analyses (see later).

Adherence to allocated programme and attrition

Some loss to follow-up was expected over 12 months. The proportion of participants missing was summarised for each outcome measure, in each arm, and at each time point. Potential bias due to missing data was initially investigated by comparing the baseline characteristics of the trial participants who have (analysable) primary outcome data to those who do not, using descriptive comparisons, t-tests, chi-squared tests and Fisher’s exact tests as appropriate. Reasons for withdrawal from the programme were summarised. Participants’ adherence with SSTP is defined as attendance to most of the planned group and individual sessions, that is participation in at least 4 (out of 6) group sessions and 2 (out of 3) individual sessions. Participants who engaged with SSTP were compared descriptively with those who did not in terms of their baseline characteristics.

Analysis of primary outcome

The primary outcome is the CBCL total score at 12 months. The primary analysis used mixed models to perform an individual-level analysis and followed Roberts and Roberts60 in adjusting for therapist clustering in the intervention arm only (random coefficient model). This model also adjusted for baseline total CBCL score and randomisation stratification factors (centre, level of intellectual disability) using fixed effects. This was a complete case analysis. The only ‘imputation’ performed was done by following guidance from the CBCL scoring manual. That is, missing values for CBCL were scored as zero unless more than eight items were missing, in which case the participant was excluded. The presentation of all findings is in accordance with the latest CONSORT statement. The model assumes that the residuals are normally distributed and homoscedastic, which was checked using residual plots (e.g. normal Q−Q plots). Where substantial departures from normality occurred, a transformation of the outcome variable was considered.

Analysis of the secondary and other outcomes

In addition to the analysis of the total CBCL score, we analysed the internal and external scores separately using the same approach. In addition, analyses were performed for each of the secondary outcomes. Continuous outcomes were analysed using the same approach as that described for the primary outcome. For binary outcomes, we used analogous logistic mixed models,61 although without adjustment for baseline outcome scores. All analyses of secondary and other outcomes should be considered supportive analyses. Missing values in the outcomes were handled, where possible, using guidance from the corresponding manual.

Sensitivity analysis

The following additional sensitivity analyses were performed. We repeated the primary analysis with additional adjustments to see if any notable baseline imbalances were encountered (due to chance or missing data). We used a mixed model to analyse both the 4- and 12-month CBCL outcomes simultaneously. We repeated the primary analysis after imputing outcome data using multiple imputations using chained equations. Specifically, we imputed component items of the CBCL score (i.e. not the total score) using item information from the baseline, 4- and 12-month CBCL scores and other variables (as appropriate). Missing baseline data were imputed using single imputation methods where multiple imputation was not successful. Per-protocol and complier-average causal effect (CACE) analyses were performed as participant adherence was relatively low.

Coronavirus disease 2019 considerations and additional analyses

The following analyses were carried out to explore the effect of the COVID-19 pandemic on the trial findings. The baseline characteristics of children and parents were summarised by randomised group before and after 16 March 2020. In addition, adherence to the programme, attrition and adverse events were summarised before and after this time point. We performed a subgroup analysis to investigate whether the intervention effect differed depending on whether recruitment was before or after 16 March 2020. This was achieved using the primary analysis model with additional indicator and interaction terms. Finally, we considered whether the effect of the intervention depended on the size of session groups. Separate analyses considered: (1) the overall group size and (2) the average group size in sessions attended. These analyses were achieved using the primary analysis model with additional indicator and interaction terms. Similar health economics analyses were performed to assess whether the cost of the intervention was affected when intervention sessions were moved online. We also assessed whether the COVID-19 pandemic had an impact on children and carers’ mental health and HRQoL during this period.

Valuation of economic outcomes

The primary economic outcome measure was the QALYs derived from utility scores obtained using the PedsQL™ General Core Scales (GCS). Mapped EQ-5D-Y utility scores algorithm62 provided an empirical basis for estimating health utilities. For parents/caregivers utility scores were derived from responses to the EuroQol-5 Dimensions, five-level version (EQ-5D-5L) using valuations obtained from an English population. 63 These were used to form QALYs over the 12-month period, adjusting for any imbalances in baseline scores. 64 Measurements have been recorded at baseline, 4 and 12 months.

Valuation of resource use

Study records of the number of therapists attending training sessions were used to track resources used in the delivery of the training programmes including trainee and trainer time (and preparation time), travel costs, attendance incentives and course materials to calculate the fixed cost of training. For the intervention delivery, we recorded the number of sessions delivered, the time each therapist spent with a family, and also any materials provided to parents/caregivers.

For the NHS and PSS analysis, data were collected on the use of health services in primary and community care, investigations and prescribed medication, hospital admission and outpatient attendance, ambulance use and social care. Data on health and social care resource use were collected at baseline (for the past 6 months), 4 months (for the past 4 months) and 12 months (for the past 6 months) post intervention.

For the analysis from the parent/caregiver perspective, we additionally collected data on out-of-pocket expenses. Expenditure on private use of treatments and therapies was captured in the CA-SUS.

Health and social care resource use was costed using unit costs from the most recent Unit Costs of Health and Social Care published by the Personal Social Services Research Unit (PSSRU)65 and NHS reference costs. The costs of medications were estimated from the British National Formulary. The cost of each resource item was calculated by multiplying the number of resource units used by the unit cost. The total cost for each participant was estimated as the sum of the cost of resource use items consumed. The primary analysis included only health and social care data collected as part of the trial and hence covered only 10 months of the trial (missing months 4–6). We have projected costs from 4- and 12-month follow-ups to estimate the 12-month health and social care resource use as part of sensitivity analyses. All costs are reported in 2019–20 Great British pounds. No discounts were applied, as trial follow-up did not exceed 12 months.

The overall economic evaluation

All analyses were conducted using intention-to-treat (ITT) principles, comparing the two groups as randomised and including all participants in the analysis. Analyses were compliant with the accepted economic evaluation methods. 66

• Cost-effectiveness analysis: mean incremental cost from the NHS and PSS perspective per change in CBCL. Incremental cost-effectiveness ratios (ICER) were reported, and uncertainty was explored using cost-effectiveness acceptability curves (CEACs). 67,68

• Exploratory analysis of quality of life using PedsQL™ to predict utility scores. The mean cost per participant for the intervention and TAU was reported by type of service use. We calculated the mean cost per QALY using the mapped EQ-5D-Y. Mean QALY per participant was calculated as the area under the curve for the duration of the trial, adjusting for baseline values.

Cost−benefit analysis of the impact on the parents/caregivers: Responses to EQ-5D-5L were used to calculate QALYs in a standard format and valued as willingness to pay (WTP) for a QALY gained. We calculated the mean cost (including out-of-pocket expenditure) per QALYs. Mean QALY per participant was calculated as the area under the curve for the duration of the trial, adjusting for baseline values.

Missing data

Missing data were explored to determine any patterns, extent and association with participant characteristics. The primary analysis included all participants using multiple imputation to predict missing costs and outcomes. 69

Analysis of relative costs and outcomes

Cost and QALY data were combined to calculate an ICER. Uncertainty in the point estimate of the cost per QALY was quantified using bootstrapping methods to calculate confidence intervals around the ICER.

The results of the non-parametric bootstrap are presented on a cost-effectiveness plane (CEP). CEACs, showing the percentage of cases where the intervention is cost-effective, over a range of values of WTP for a QALY gained, were constructed using the bootstrap data from a range of values of WTP for a QALY gained for each different costing perspective and for the different methods of calculating QALYs. The probability that the intervention is cost-effective compared to TAU at a WTP for a QALY gained of £20,000 and £30,000, and £13,000 as a measure of opportunity cost were reported. 70

Sensitivity analyses

One-way sensitivity analyses were used to judge the potential impact of sources of uncertainty:

• complete case analysis;

• given that training costs may differ between the trial and implementation of the intervention due to learning or being delivered to a larger patient group, we tested the impact of varying training costs (particularly because of larger patient numbers per staff member trained) on the mean incremental cost per QALY gained.

Parent 1

As a parent of two children with additional needs that have very challenging behaviour, I was so excited to be part of this project to give the voices of parents who had genuine lived experience. I also had the benefit of being part of a large network, had opportunity to ask direct questions and got feedback from others. We were included in every part of the project. The information we gave as parents was always respected and included and we felt very much a part of the team. The professionals on the trial always listened to our opinions and genuinely wanted the support that we gave on the parent panel. I would definitely love to be part of any future projects like this and the whole team at UCL and NHS England treated us with respect and as a colleague with experience rather than just having to have us there as part of guidelines for the project. I found the face to face meetings beneficial as we all felt part of a wider team and network and they weren’t as daunting as online meetings. I think having face-to-face meetings before the COVID-19 pandemic resulted in better working when we had no alternatives than working online as relationships had already been formed. I am extremely proud of this project and how they altered as much as possible to meet the COVID-19 requirements. I hope that it results in helping families struggling with their children and would have benefitted from it when my children were that age.

Parent 2

I was given the opportunity to join the parent advisory group for the EPICC-ID clinical trial as I was involved with various projects run by Contact – the Charity for families with disabled children. I did not know what to expect from the trial as I have never been involved with anything like this before. I knew that the trial was studying the impact of a particular intervention for children with intellectual disabilities who exhibited challenging behaviour. As this is something that affects myself and my family personally I felt very fortunate to have been asked to take part. I can honestly say I have learnt so much from the whole experience. From finer details such as defining exactly what challenging behaviour is, to the bigger picture of how National clinical trials actually work and the complexities of randomisation and blind controls. It was a steep learning curve but always a privilege to share time with so many brilliant, respected consultants working in the field of Child and Adolescent Psychiatry. Throughout the trial I gained a lot of confidence and was encouraged by the professionals to share views and opinions about the process and the Triple P intervention itself. I always felt my contributions were valued and it was amazing to be involved in the drafting of a journal for the BMJ. This is not something many people outside of medicine can say! I have seen how clinical trials – like EPICC-ID open doors to advancing prevention and improving lives. The team always made it clear that clinical trial volunteer participants like myself play an important part in the trial process and I would not hesitate to be involved with a parent advisory group again. I am very grateful to Contact, Professor Angela Hassiotis and all the team for inviting me to take part.

Parent 3

I really enjoyed being on the SSTP trial, from sitting on an advisory parent’s group to analysing data. Progressively, I’ve seen perquisite effort to adapt contextual factors to improve recruitment numbers and resilience to complications like COVID-19 pandemic. Thank you for asking me to be part of the trial, I would love to do it again. It’s very rewarding.

Participant flow and retention

A total of 261 participants were enrolled in the trial and randomised. Of these participants, 155 were allocated to the SSTP intervention, and 106 were allocated to TAU. Figure 1 shows a CONSORT flow diagram summarising the flow of participants into the trial.

Other caregiver reported Child Behaviour Checklist caregiver−teacher report form at 4 and 12 months

A linear mixed-effects model was fitted with C-TRF total score at 4 months as the outcome variable. This model included only those participants for whom total C-TRF scores were available at baseline and 4-month follow-up. The intervention increased the C-TRF total score at 4 months by an average of 6.17 points (95% CI −7.05 to 19.38; p = 0.351). As such, there is no statistical evidence to suggest a difference in the C-TRF total score at 4 months between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with C-TRF total score at 12 months as the outcome variable. This model included only those participants for whom total C-TRF scores were available at both baseline and 12-month follow-up. The intervention increased the C-TRF score over 12 months by an average of 10.920 points (95% CI −4.07 to 25.91; p = 0.147). As such, there is no statistical evidence to suggest a difference in the C-TRF total score at month 12, between the TAU and SSTP groups. The intervention effect estimate is given in Table 10.

General Health Questionnaire-12 at 4 and 12 months

A linear mixed-effects model was fitted with GHQ-12 total score at 4 months as the outcome variable. The intervention reduced the GHQ-12 score over 4 months by an average of 0.23 points (95% CI −1.75 to 1.29; p = 0.764). As such, there is no statistical evidence to suggest a difference in the GHQ-12 total score at 4 months between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with GHQ-12 total score at month 12 as the outcome variable. The intervention reduced the GHQ-12 score over 12 months by an average of 0.42 points (95% CI −2.37 to 1.52; p = 0.666). As such, there is no statistical evidence to suggest a difference in the GHQ-12 total score at month 12, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate. The intervention effect estimate is given in Table 11.

Questionnaire on Resources and Stress-short Form at 4 and 12 months

Similarly, a linear mixed-effects model was fitted with QRS-F total score at month 4 as the outcome variable. The intervention reduced the QRS-F score over 4 months by an average of 0.38 points (95% CI −1.40 to 0.63; p = 0.456). As such, there is no statistical evidence to suggest a difference in the QRS-F total score at month 4 between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with QRS-F total score at month 12 as the outcome variable, the intervention effect estimate is given in Table 11. The intervention increased the QRS-F score over 12 months by an average of 0.09 points (95% CI −1.02 to 1.20; p = 0.874). As such, there is no statistical evidence to suggest a difference in the QRS-F total score at month 12, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate. The intervention effect estimate is given in Table 12.

Caregiving Problem Checklist at 4 and 12 months

The same linear mixed-effects model was fitted with Caregiving Problem Checklist total score at 4 months as the outcome variable. The intervention increased the Caregiving Problem Checklist total score over 4 months by an average of 0.09 points (95% CI −2.08 to 2.25; p = 0.937). As such, there is no statistical evidence to suggest a difference in the Caregiving Problem Checklist total score at month 4, between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with Caregiving Problem Checklist total score at month 12 as the outcome variable. The intervention reduced the Caregiving Problem Checklist total score over 12 months by an average of 2.05 points (95% CI −4.72 to 0.61; p = 0.129). As such, there is no statistical evidence to suggest a difference in the Caregiving Problem Checklist total score at month 12, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate. The intervention effect estimate is presented in Table 13.

Parenting Sense of Competence Scale at 4 and 12 months

The same linear mixed-effects model was fitted with PSOC total score at month 4 as the outcome variable. The intervention reduced the PSOC score over 4 months by an average of 1.66 points (95% CI −4.14 to 0.81; p = 0.186). As such, there is no statistical evidence to suggest a difference in the PSOC total score at month 4, between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with PSOC total score at month 12 as the outcome variable. The intervention increased the PSOC score over 12 months by an average of 2.20 points (95% CI −5.29 to 0.88; p = 0.160). As such, there is no statistical evidence to suggest a difference in the PSOC total score at month 12, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate. The intervention effect estimate is given in Table 14.

Internalising and externalising domains of Child Behaviour Checklist at 4 and 12 months

Table 15 provides a summary of CBCL internalising and externalising scores, stratified by allocation arm, at baseline, 4 and 12 months.

Two linear mixed-effects models were fitted with CBCL internalising score at 4 months and at 12 months as the outcome variable. The intervention reduced the CBCL internalising score over 4 months by an average of 0.32 points (95% CI −2.37 to 1.72; p = 0.756). As such, there is no statistical evidence to suggest a difference in the CBCL internalising score at 4 months, between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. The intervention increased the CBCL internalising score over 12 months by an average of 0.21 points (95% CI −2.12 to 2.54; p = 0.856). As such, there is no statistical evidence to suggest a difference in the CBCL internalising T-score at 12 months, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate. The intervention effect estimate is given in Table 16.

Child Behaviour Checklist externalising score at 4 and 12 months

The same linear mixed-effects model was fitted with CBCL externalising score at 4 months as the outcome variable. The intervention reduced the CBCL externalising score over 4 months by an average of 1.21 points (95% CI −2.97 to 0.54; p = 0.174). As such, there is no statistical evidence to suggest a difference in the CBCL externalising score at 4 months, between the TAU and SSTP groups. The model assumptions were checked and found to be appropriate. Similarly, a linear mixed-effects model was fitted with CBCL externalising score at 12 months as the outcome variable. The intervention effect estimates of both models at 4 and 12 months are given in Table 17. The intervention reduced the CBCL externalising score over 12 months by an average of 1.59 points (95% CI −3.58 to 0.39; p = 0.114). As such, there is no statistical evidence to suggest a difference in the CBCL externalising score at 12 months, between the TAU and SSTP groups. Again, the model assumptions were checked and found to be appropriate.

Parent–child interaction measured by the Revised Family Observation Schedule

Table 18 provides a summary of child per cent negative behaviour, indicated by the percentage of 30-second observation intervals where at least one (often more than one) of the negative behaviours had occurred (Non-compliance, Complaint, Physical Negative, Oppositional, Interrupt). Table 19 provides descriptive statistics for the total number of intervals that each child and parent behaviour was observed, at baseline, 4 and 12 months, respectively.

Mullen Scales of Early Learning

The summary statistics of child level of disability of cognitive functioning measured using the Mullen Scales of Early Learning is provided in Table 20.

Client Satisfaction Questionnaire

Forty-nine participants from the SSTP group provided feedback at 4 months of their experience of the intervention, including ease of use, format and helpfulness. The total score of the questionnaire is 32. The average score from 49 participants is 27.6 points [SD = 3.0; median = 29 (18, 30)].

Baseline characteristics by primary outcome completion

Whether the participants in the primary analysis were representative of those randomised was initially investigated by comparing the baseline characteristic of the trial participants who had complete primary outcome data to those who did not. Summaries of demographic and outcome variables at baseline, stratified by if the primary outcome is missing, are provided in Tables 21 and 22 for children and parents, respectively. p-values indicate that there is no evidence to suggest a difference between the two groups, except for increased rates of mental health problems in parents who dropped out of the study (40% vs. 25%).

Baseline demographic characteristics by adherence

Participants’ adherence is defined as attendance at the majority of the planned group and individual sessions, that is participation in at least 4 (out of 6) group sessions and 2 (out of 3) individual sessions. Of the 155 participants who were randomised to the SSTP arm, 50 participants were adhering to the SSTP intervention. Participants who were adhering to SSTP were compared descriptively with participants who did not adhere to the intervention in terms of their baseline characteristics and results are provided in Tables 23 and 24 for children and parents, respectively. p-values indicate there is a difference between the groups based on adherence in children’s mobility problems, parents’ living situation, relationship status and the receiving of state benefits.

Per-protocol and CACE analyses were performed as the participant adherence to intervention was relatively low.

Per-protocol analysis

To account for non-adherence in the SSTP arm, a per-protocol analysis was calculated to measure the effect of the intervention on CBCL total score at 12 months, using a linear mixed regression. This analysis excludes non-adhering participants from SSTP and compares the adherence with the TAU group. The effect of the SSTP on CBCL total score at 12 months was estimated as −10.77 (95% CI −19.12 to −2.42). This implies there is a large difference to the previous conclusions concerning the effect of the SSTP intervention on CBCL total score at 12 months. The per-protocol analysis results are presented in Table 25.

Complier-average causal effect analysis

A CACE estimate was also calculated to measure the effect of the intervention on CBCL total score at 12 months, using a two-stage least squares approach. This analysis adjusts for non-adherence and maintains randomisation balance by comparing SSTP compliers with TAU participants who would have complied if randomised to SSTP. The effect of the SSTP intervention on CBCL total score at 12 months was estimated as −11.53 (95% CI −26.97 to 3.91). The treatment effect is similar to that from the per-protocol analysis, and the p-value tends to be similar to that from the primary (ITT) analysis. CACE analysis results are presented in Table 26.

Imputing Child Behaviour Checklist total score at baseline, 4 and 12 months

Fifty out of 261 participants were missing in the ITT analysis. Therefore, we imputed the primary outcome data using multiple imputation techniques based on chained equations. Specifically, we imputed the total score of the CBCL by two arms using information from the baseline (child age, child sex and baseline CBCL), 4- and 12-month CBCL total scores and other variables (site and level-LD). A total of 19 imputed data sets were created, as there are 19% missing values in the CBCL total at 12 months. Estimates from imputed data sets were combined using Rubin’s rule. The effect of the SSTP intervention on CBCL total score at 12 months was estimated as −4.85 (95% CI −10.24 to 0.54). The results of this analysis are presented in Table 27. The treatment effect is slightly larger than that of the primary analysis, and the p-value indicates that there is weak statistical evidence to suggest a difference in the CBCL total score at 12 months, between the TAU and SSTP groups.

Subgroup analysis based on group size

We also considered whether the intervention effect depends on the size of the session groups. Separate analyses were conducted for: (1) the overall group size; (2) the average group size in sessions attended. We note that these analyses feature post-randomisation variables (session size) and that groups are not balanced by randomisation. Hence these analyses should be regarded as exploratory.

The overall group sizes

The participants were assigned into the following four groups:

• 0 = TAU

• 1 = SSTP (not assigned to SSTP group)

• 2 = SSTP (assigned to SSTP group with at most five people)

• 3 = SSTP (assigned to SSTP group with more than five people).

The same linear mixed-effects model was fitted with CBCL total score at 12 months as the outcome variable. The estimates of the effect of the three intervention groups on CBCL total score at 12 months are given in Table 28.

This exploratory analysis suggests that children allocated to smaller groups saw bigger reductions in CBCL scores (compared to TAU).

The average group size in sessions attended

The participants were again assigned into the following four groups:

• 0 = TAU

• 1 = SSTP (no group sessions)

• 2 = SSTP (average size 1–3)

• 3 = SSTP (average size > 3).

A linear mixed-effects model was fitted with CBCL total score at 12 months as the outcome variable. The estimates of the effect of the three intervention groups on CBCL total score at 12 months are given in Table 29.

This exploratory analysis again suggests that parents of children allocated to smaller session groups saw bigger reductions in CBCL scores (compared to TAU).

Baseline demographic characteristics before and after 16 March 2020

To study the effect of the COVID-19 pandemic on this trial, the baseline characteristics of children and parents were summarised in Tables 30 and 31 by allocation arm, before and after 16 March 2020.

Adherence to the programme and attrition

The adherence to the programme and attrition are also summarised before and after 16 March 2020 in Table 32.

Subgroup analysis based on the coronavirus disease 2019 impact

We performed a subgroup analysis to investigate whether the intervention effect differed depending on whether recruitment was before or after 16 March 2020, using the primary mixed model with an interaction term. In this model, the mean difference of the effect of the SSTP intervention on CBCL total score at 12 months between those participants who were recruited before 16 March 2020 and those after 16 March 2020 were estimated as −7.12 and 7.61, respectively. The interaction p-value of 0.046 suggests the effect of SSTP was significantly different pre-pandemic compared to during the pandemic. In addition, the point estimates suggest the direction of effect may have reversed after the start of the pandemic, though the confidence interval for the ‘during’ estimate is wide due to small patient numbers. See Table 33 for further details.

Cost of training and delivery of Stepping Stones Triple P

In total, 11 therapists across the four services received training in delivering SSTP. The cost of the training was £26 per participant (Table 34). Details of the activities carried out by therapists were estimated at an average cost of £270 (see Report Supplementary Material 4). Training and delivery of the SSTP were estimated at a total average cost of £296 per study participant.

Delivery challenges

To understand the challenges of implementation of the SSTP intervention in healthcare settings, we interviewed three service managers. They were interviewed remotely during the first 9 months of the study as part of the internal pilot. The interviews discussed the recruitment and training of the therapists, any issues with delivery, and how they felt about facilitating the adoption of the intervention into their services. All interviews were audio-recorded, transcribed verbatim and entered into the NVivo^® (2020) (QSR International, Warrington, UK) software for further analysis. The transcripts were read and coded by the research assistant who identified all important information that came up during the interviews.

The service managers discussed the perceived interest in a group parenting programme like SSTP and expressed concerns about whether parents would be interested in SSTP, specifically since their services already offered a variety of therapies, including Early Bird or the My Child Programme.

I was slightly concerned that we already offer quite a lot of interventions for families that meet the criteria anyway. So, I wasn’t sure about the difference between the intervention group and the control group. So that’s basically what my feeling about it was first, but I thought that if we can enhance anything then we would want to.

Service Manager 1

Despite this, service managers said they felt SSTP would be a valuable addition to their service, especially as it addresses behaviours that challenge displayed by young children. However, they also expressed concerns about costs and funding.

I thought this was going to be very helpful for families particularly coming through our child development team, which is often children aged nought to five with very complex needs. Behaviour is a very big issue and one that comes up from parents quite a lot. So, the idea of doing this enhanced Triple P seems like a very good thing to be able to offer them.

Service Manager 3

I would like to mainstream it and when that funding is coming to an end, so we have been thinking about we might mainstream the skills, but as with everything I really would struggle to fund it.

Service Manager 1

Running the sessions smoothly required careful preparation, including finding the right venue with appropriate equipment and setting up the room before sessions. Service managers described difficulties with finding a venue with a good location, size, equipment and parking facilities.

And that’s been tight in terms of getting rooms that are large enough to accommodate all of this that also provide the audio-visual facilities to play the videos, you know, which is what the therapy requires. So that’s caused a lot of challenges. We’ve eventually found the space, but parking isn’t great, and travelling is not great from here, you know, which is probably why we found the space. But no, eventually it worked out. But it has caused some challenges.

Service Manager 2

When providing SSTP in their service, managers emphasised the need to consider the timing of the sessions to ensure regular attendance by parents, who are often busy and cannot afford to spend long hours in group sessions.

My only concern would be that our experience of running groups and I think in particular with this research project is that it does take quite a lot of man hours to set up and run the groups. Also, some families that we work with really struggle to participate in the group setting so they would be my two caveats really …

Service Manager 1

Service managers also described difficulties with finding therapists to be trained in delivering SSTP. For example, service providers had to externally recruit a therapist to deliver three groups due to the limited availability of therapists in London and due to another therapist taking unexpected sick leave.

We had quite a lot of difficulty recruiting because we did have some people in the borough who are already trained, which we hoped we would be able to link with, but we actually weren’t able to do that because the service they were local authority employed and that service were not keen to give up their skills.

Service Manager 2

However, once recruited, service managers were happy with the skills and provision of the therapy.

The people that we did recruit, we were particular pleased that they both were embedded in our local specialist schools, so we thought would be a good link and also once they were trained that they maybe would be able to continue using those skills, so it’s all skill development.

Service Manager 1

Fidelity

The fidelity of the intervention was reviewed by an independent reviewer with experience delivering SSTP. A fidelity form was adapted from the i-Basis Intervention Fidelity rating scale (first developed for use in the Preschool Autism Communication Trial study). 76 This form balances the contribution between session content and delivery style. The therapists completed checklists from SSTP providers and attendance worksheets for every session.

The fidelity form includes:

• general group procedures;

• interpersonal effectiveness;

• specific criteria for each session;

• deviations from the manual;

• video and sound.

Each section included items scored as ‘0’ for No, ‘1’ for Yes or N/A. These scores are summed to give a total fidelity score (max score = 10). A quality score is also given on a rating of 1–5 (poor–excellent). Sessions were video recorded and stored in the online platform, DSH.

Ten per cent of sessions were randomly selected from the total number of sessions, ensuring an even spread of site, group and session number. The selection was generated by the study statistician. This resulted in a total of 13 sessions being reviewed by an external reviewer (Maeve Darroux – SSTP deliverer at Brilliant Parents). Three out of the 10 sessions reviewed were delivered remotely. Fidelity scores ranged from 7 to 10 (M = 9.38, SD = 0.96). Eight sessions (62%) were scored as having the maximum score for fidelity. In terms of quality, two sessions were rated as 3 – adequate, with the remainder of the sessions being rated at 4 – good (N = 11).

Across the 13 sessions, therapists reported some deviations from the manual:

1. A practitioner referred to ‘quiet time’ being discussed before ‘logical consequences’ rather than after.

2. A practitioner demonstrated role play which the parent was not required to copy, but this was still described to be beneficial by the external reviewer.

3. Practitioner did not use a whiteboard or flip chart to support the strategies as recommended by Triple P. This occurred in three sessions, all at different sites. On one of the occasions, the whiteboard was not used because only one parent was present in the session.

4. Due to a small number of participants in one of the group sessions (N = 1), the therapist accommodated the attending parent’s preference to have an individual phone call session instead of a face-to-face session.

Therapist training and Stepping Stones Triple P delivery

Therapists were health professionals involved with assessing or treating children with behaviours that challenge. The trained therapists had the following professions: community social worker, specialist health visitor, assistant psychologist, specialist nurse, family and inclusion mentor and occupational therapist. The therapists were not involved in the routine care of study participants and were based at separate facilities from the researchers to avoid contamination and unblinding. We recruited 11 therapists to deliver SSTP: 2 therapists from Sites 1, 2 and 4, and four therapists from Site 3.

All therapists were trained in the Group Stepping Stones Training and Accreditation programme. This included three training days and a further half-day accreditation workshop after 6 weeks. SSTP requires a therapist to spend an average of 37.5 hours on each family receiving the intervention, including time spent on pre-session preparation, post-session reporting, fidelity checklist completion and supervision. The training was conducted by Dominic Weston, Deputy Head of Training (UK) and Implementation Consultant of Triple P UK and Ireland. Dominic Weston provided 28 hours of supervision to therapists. Triple P trainers observed therapist sessions to help build therapist competence to run the groups. Monthly supervision was provided to maintain and monitor therapist skills over time by a co-applicant trained in SSTP (seven times during the study duration). Each therapist took part in the supervision and had access to the wider Triple P network of practice, which provides ongoing advice about the programme delivery and a range of clinical resources. The learning objectives focus on maintaining behavioural change, using skills within a group learning environment, learning from peers in the group and sharing difficulties or achievements, providing support, considering if more intensive work is required, referring further if needed, and talking about risk and protective factors operating within families.

All groups were delivered by at least one therapist and six groups were delivered by two therapists.

Adaptations

The therapists discussed any adaptations they made to sessions during supervision meetings. These included:

• Therapists agreed for parents to bring very young children if they struggled to find child care during the session time.

• Parents were allowed to bring partners to the sessions, as this was the most common request at all sites.

• To boost the number of people attending group sessions, we obtained ethical approval to include parents of children who had been screened for the study but were ineligible to take part due to not fulfilling the inclusion criteria.

• As per protocol, therapists contacted parents who missed a session to remind them of the next scheduled meeting and asked parents to read the relevant chapter in the workbook. Due to the concerns about the impact of the missed session, therapists offered a catch-up session to explain the missed content. The catch-up sessions were helpful, but therapists had concerns about how many catch-up sessions could be provided as it interfered with their work schedules, was time-consuming, and was difficult to provide sufficient information about the session on the phone call.

Dose and reach

Participants were recruited through participating NHS healthcare services (including primary care), parent support groups and online multimedia channels through the charity Contact. This charity reaches 20% of families with disabled children each year. The majority of participants were recruited through Child Development Centres which provide the main point of access for assessment and diagnosis of children with developmental delay, and outpatient clinics provided by the CAMHS. It was not possible to keep a structured record of all patients approached due to the number of PIC and the number of clinicians supporting the project. However, expressions of interest were obtained from 583 patients. Data on the number of participants approached were available from a Site 3 PIC, and this provides an example of the proportions of people being approached that were then randomised into the intervention (Figure 10). A total of 79 participants were approached at Site 3 PIC and 54% of these expressed an interest in taking part in the study, with 17 people (22%) being randomised into the study. Based on these figures, we can approximate that the reach of the trial may have been around 1100–1300 people.

FIGURE 10.

An example of parents approached and reasons for exclusions from the Site 3 PIC.

A total of 155 parents were invited to attend the SSTP group and 91 attended at least one session (59%). Sixty-four people did not attend any of the intervention sessions. The minimum acceptable dose of the intervention was stipulated to be four group sessions and two individual sessions. A total of 50 participants received this level of dosage (see Table 38 for the attendance breakdown by type of session).

Therapists were asked to record reasons for non-attendance at sessions. Some reasons included the parent being in education, the child being unwell, lack of support with child care and clashes with other obligations or medical appointments (see Figure 11 for further details).

FIGURE 11.

Reasons for non-attendance reported by parents.

Therapist views

Therapists noticed that the impact of the group depended highly on the parents’ abilities to comprehend and apply the skills presented:

I found the way the therapy is set up, to go through one session each week, is a lot of material and so the demand on the parent is quite large in terms of their ability to follow along, to understand and to be coached. It requires that both the person administering it has confidence in their coaching skills and that the parent is coachable. I think that some considerations didn’t go all the way to ensure participants are coachable.

Therapist 1, Interview

One therapist expressed the need for flexibility in offering more sessions depending on how quickly parents grasp the information being covered:

I would like to have more flexibility to gear the program to each particular group, i.e., to be able to add in an extra week if we weren’t getting through the sessions as quickly as we had hoped or to spend a bit more time on something or go back to something.

Therapist 1, Interview

One therapist expressed their thoughts on the length of sessions and potential need for splitting the sessions into shorter or more frequent sessions to be able to cover all material:

I could have easily used an extra day and could have broken down the 5 sessions into 6. The sessions are really long for everyone and that is definitely something to consider. By the end of running one, you are exhausted. You need to go through the material and watch the room to ensure everyone is engaged and be the cohesive glue for the group. Whoever is running it really needs experience in coaching. It is not an entry level experience programme and people really need coaching experience.

Therapist 1

Besides the struggles to find the best time for each parent in the group, the timing highly depended on the flexibility of the therapist, which was restricted by working hours:

One significant reason could be related to the timing of the groups, Site 1 has tried an evening group. One parent requested this, and it had to be early evening which didn’t suit everyone else. In most sites the therapists are only employed for 1 day and have other commitments so they cannot be flexible in offering differing days of the week.

Therapist supervision meeting

There were also concerns about parents with English as a second language and their understanding of the content.

Some of my personal experiences were that there were parents who had significant learning needs that meant they were not coachable and there were participants who struggled because English was not their first language. A lot of the material is explained clearly but I felt that some of the participants struggled due to their understanding of English.

Therapist 1, Interview

Several aspects of the SSTP therapy made it easier for parents to follow, even if their comprehension of the session was lower. For example, providing workbook exercises and a handbook that can be taken home were seen as helpful to keep parents on track. Moreover, the SSTP handouts are translated into five languages, which is useful for parents whose first language is not English.

When delivering the remote sessions, some parents found it difficult to log into MS Teams, and therapists found it challenging to not be able to see all participants when presenting slides. Zoom seemed to work better for therapists and allowed them to see more people on the screen. Session videos were shown by sharing the screen and the therapists acted out role plays with a second therapist remotely.

Therapists discussed the benefits and challenges of remote delivery of SSTP therapy. Some of the benefits included increased group sizes, the opportunity to be more flexible with timing and session length, and a greater possibility of additional caregivers joining sessions. Many parents preferred the convenience and flexibility of online delivery, which likely boosted attendance.

More parents have been able to attend, however some have been more distracted. Some of them have their child at home so there were situations where parents had to step away to manage something. I experienced better attendance and great opportunities by delivering remotely but this was balanced by distraction.

Therapist 1, Interview

Some challenges also included a limited opportunity for informal conversation between parents and therapists, which is important for building rapport and making parents more comfortable sharing with others. Therapists noticed fewer natural discussions, which meant they had to be more directive in their approach and invite people to comment. One way of tackling this was by using breakout rooms to encourage conversations between the parents without the therapist present.

Another challenge was internet and connectivity issues. Many people did not have unlimited data, and sometimes their broadband width was insufficient, making it hard to see their faces or resulting in crashes. This interrupted the flow of the session and wasted valuable time. Some parents commented that 2.5 hours spent online is a long time for many parents with young children at home.

1. Treatment as usual Group Results: ‘Need for intervention’

Parents in the TAU group described their experiences of receiving usual care that was locally offered to their children and described what they felt worked well and what did not work well.

2. Stepping Stones Triple P Group Results: ‘Does the SSTP meet parents’ needs?’

Parents who received SSTP provided their reflections on which aspects of the intervention they found helpful.

3. Results from all parents (Stepping Stones Triple P and treatment as usual arms): ‘What are families looking for in a group intervention?’

The parents from both trial arms talked about their experience with group interventions, and their concerns and expectations if they were to be offered one.