The COVID-19 pandemic has been an unprecedented event within this century and has had a significant effect on every clinical trial running during this time. Many trials halted recruitment, stopped delivering interventions or closed down. This was due to the impact of the pandemic and concomitant restrictions and redeployment of clinical staff leading to loss of capacity and necessary resource. In addition, staff sickness and participants unwilling to take part due to the risk of catching COVID-19 created additional challenges.
On 23 March 2020, the UK entered a strict lockdown, which included the enforcement of rules relating to leaving your property, which was only permitted for specific reasons and for restricted time periods. At this point, URB was still in the recruitment phase. As well as recruiting to the study, the URB and LAA intervention groups were running in person, mainly on clinical premises, facilitated by clinical personnel. Several decisions had to be made as to whether and how the trial could continue. These decisions included how to undertake informed consent with participants, how to deliver the interventions and how to manage follow-up assessments. NCTU worked closely with the TMG and sponsor to discuss each element of the trial and how to develop methods of ensuring its continuation. The development of methods to enable continuation was further complicated by local variations in how different sponsors at the three trial sites interpreted and enforced government guidelines.
With agreement from sponsor (CNTW), REC and the TSC, including patient and public involvement (PPI) members, the following activities were delivered remotely or via online secure platforms to ensure that the trial could continue:
Consent
Verbal/e-mail consent became the primary method for participants to provide consent. The PIS was read out to the participants in full over the telephone and e-mailed where possible. A consent proforma was then completed, ensuring that each point matched the approved consent form, to document that the participant agreed to each point.
This consent process was found to be both acceptable and feasible to potential participants – a total of 62 participants were consented using this method. Consent was briefly paused while these measures were developed, approved and put in place. Unfortunately, permission was not gained to reopen recruitment in Scotland, leading to the Lothian recruitment site closing permanently in March 2020. The closure of the Lothian recruitment site necessitated an increase in target recruitment at the remaining two research sites in order to achieve the required sample size. It also led to a requirement that additional groups were then run at the two remaining sites to accommodate the additional participants recruited at each site.
The delivery of the Understanding Repetitive Behaviours and Learning About Autism parent groups
A significant challenge following the onset of the COVID-19 restrictions was to determine how the interventions could be delivered via remote methods. Many factors had to be taken into consideration such as data protection for the participants and therapists, the use or potential limited use of technology and any associated costs and whether delivering groups online would be feasible for both staff to deliver and parents/carer to attend and could be delivered with fidelity. As both interventions had been designed to be delivered in a face-to-face format, the programmes had to be reviewed to ensure that the activities could be delivered in an online format and some adjustments needed to be made to the materials. At the time of lockdown, one group in CNTW was midway through delivery; the delivery of the group was paused while appropriate adjustments were made to the materials and the final four sessions of the group were then delivered to participants using an online format.
After March 2020 all LAA and URB groups were conducted entirely online using a range of free platforms; Microsoft Teams and Attend Anywhere. The platform adopted differed across NHS trusts based on local policies/guidance. The use of online platforms to deliver the LAA and URB groups presented some challenges for group leaders. The majority of group leaders had not delivered therapeutic groups or teaching/training online previously and were therefore unfamiliar with the online platforms. Additional time was required at the start of each session to support parent/carers to access the online platform and some parents/carers required individual support with technology between sessions. Earlier versions of the online platforms had limited features, for example only four people were visible at once on screen and parent/carers were not visible at all while the group leader was displaying session slides. This meant that group leaders may have missed nonverbal cues that a parent/carer wished to contribute to a discussion or was finding the content of a session difficult. Similarly, managing group dynamics (e.g. dominating personalities) was potentially more difficult online. Due to COVID-19 restrictions many children did not attend school during lockdowns, this meant parents/carers’ attention was often divided during the group sessions.
The move to online delivery of groups also brought some positives for parents/carers. Once initial technological challenges were overcome, some parents/carers reported that it was easier for them to attend the groups online as this greatly reduced the time commitment required as they did not need to travel to/from the group or organise child care. Parents/carers were also able to share visually with the group any environmental factors and strategies utilised within the home (e.g. social stories and visual timetables).
Baseline and follow-up assessments
After March 2020 the majority of baseline and follow-up assessments at weeks 10, 24 and 52 were conducted by telephone by the RAs. It was agreed that these assessments could take place over multiple sessions within a given time period to allow for more complete data collection and to reduce pressure on the participants to be available for a lengthy phone call, particularly as in many instances their children were not attending school due to the lockdown and therefore there was an increase in their parental duties at this time. In exceptional cases, and during periods of lockdown easing, as a reasonable adjustment some home visits were made to participants to undertake assessments if the participant was not able to participate comfortably by telephone. These visits were undertaken with the relevant social distancing requirements and following approval of the necessary risk assessments by senior research staff.
Fidelity assessments were also completed remotely due to COVID-19 restrictions. This involved the fidelity raters, who were located in Tyne & Wear (CNTW), reviewing a sample of LAA and URB session recordings via a virtual private network at Edinburgh University (Lothian NHS) or via Egress NHS encryption service (TEWV). While it was possible to complete the fidelity ratings remotely this method proved to be costly timewise due to ongoing server issues and difficulties streaming the videos.
Impact on the primary outcome
After reviewing all the baseline and outcomes measures, it was confirmed that all measures could be conducted over the telephone, with the exception of the ADOS-2 assessment. This measure, which is an observational play-based assessment with the child, is designed to be conducted face to face. Data from this measure were originally planned to be gathered at baseline and contribute to the CGI-I to provide raters with some detailed clinical insight into the child’s level of ability and autistic traits. It was not planned that the measure would be repeated at follow-ups. From March 2020 onwards, ADOS-2 assessments were not undertaken with children due to the implementation of national governments’ and NHS COVID-19 restrictions. Consequently, ADOS-2 scores were only obtained for two-thirds of the participants (155/227) and may therefore not be clinically representative of the full sample. Given this, the ADOS-2 scores were not included in the CGI-I analysis.
Impact on secondary outcomes
While it was determined that, with the exception of the ADOS-2, all other assessments could be collected remotely, the pandemic may have had an additional impact on responses to some or all of these other measures. During the pandemic, schools were closed for considerable amounts of time and when they were in session they were subject to changes to procedures. These school-based changes may have impacted on teaching staff responses on the RBQ Teacher version; most particularly, they may have spent far less time with the children they were reporting upon than would have previously been the case. In addition, the changes to everyday life that were a consequence of restrictions may have impacted on children’s RRB directly, for example the setting for the RRB may have no longer been somewhere that was accessed during lockdown (e.g. school, shopping centres or playgroups); indeed, a small number of scenarios identified during the TBVS were no longer relevant.
Restricted and repetitive behaviours were also assessed more generally using the RBQ-2, which requires parents/carers to rate their child’s engagement in a range of RRB. It is difficult to determine the full impact of COVID-19 on the families who were part of the study. Many parents/carers reported changes in their child’s well-being as a consequence of the pandemic; for some these changes may have been negative with their autistic child feeling anxious about how the pandemic would unfold and upset about disruptions to their routine, for others the changes may have been more positive with the child benefiting from a less demanding lifestyle with the focus on staying at home with family. For many families it may have been a combination of the two at different times. These circumstances therefore may have impacted in unanticipated ways on the behaviours captured by the RBQ-2.
In addition, for parents/carers the closure of schools and services meant that they often lost their expected everyday support networks and often became full-time caregivers of their autistic child. Recent research indicates that some autistic children experienced worsening in behavioural, social and developmental domains during the pandemic, while others thrived in a more restricted environment.
It is difficult to pinpoint what impact COVID-19 may have had on the sensitivity of our outcomes measures and the study is not powered to provide a definitive answer to these questions.
Impact on economic outcomes
In addition to the clinical effectiveness measures, there is a possibility that COVID-19 had an impact on the economic conclusions. With regard to the effectiveness measures, the impacts that have been noted for the primary and secondary outcomes will have also affected the economic evaluation when comparing the costs and the benefits. In addition, with regard to the measurement of resources used, the impacts of COVID-19 meant that there may have been less availability or willingness to access services as a result of lockdown measures. As such there may have been less services utilised by participants. However, given the unprecedented and stressful nature of the restrictions, the opposite may also be true if children needed greater support during this time. What should be noted is that whatever the effect may have been on resources, it would have likely had the same impact on both arms of the evaluation, in an equal manner, and therefore we would not expect to see significant differences between the arms.
Statistical considerations
Future practice
One unanticipated benefit of the COVID-19 pandemic was the opportunity to deliver the assessments and interventions via remote means. Our fidelity analysis, which included ratings of sessions delivered in the original face-to-face and remote formats, indicates that both methods of delivery are feasible, can be done with fidelity to the manual and are acceptable to parents/carers. Indeed, informal feedback from parents/carers during COVID-19 suggests that they were appreciative of the efforts of the research and clinical teams to continue with the study during a time when many clinical, educational and support services were closed. As such, we do now have some evidence that both URB and LAA are feasible and acceptable to delivery via remote methods. This has significant implications for the implementation of the programmes via clinical services to families who may struggle to attend the groups in a face-to-face format. A separately funded qualitative study is currently evaluating the findings of a series of focus groups undertaken following the completion of the main trial to investigate the impact of COVID-19 on delivery of the interventions.