Aim
The overarching aim of the PPI work in the ORBIT trial was to collaboratively involve CYP with tic disorders and their parent/carers at all stages of the ORBIT trial. Specific objectives included:
- to inform the refinement of the intervention from Swedish to English
- to incorporate the experiences of young people and their parent/carers to inform patient-facing documents
- to advise on how to maintain patient and public interest in the trial throughout its lifespan
- to assist in interpretation and presentation of the data to the public.
Method
We partnered with Tourettes Action [Tourettes Action (tourettes-action.org.uk)], a national charity, in order to facilitate PPI in this study.
As part of this, we developed a core PPI panel, consisting of four young people with TS and their parent (in all instances, the parent was the mother). The panel was recruited via Tourettes Action and known by contacts through clinical co-applicant.
As the initial step, the method in which the families wanted to be involved was established. All families wanted to remote contact, which was partially driven by concerns of ‘ticcing’ in front of other families/new people, and partially driven by the fact the PPI group were geographically spread over England. Although a budget was provided for PPI travel, the families stated they would find it difficult to find the time.
There were no specified meetings with the PPI group, although they received a monthly update from the trial manager to ensure they were kept informed of progress.
The PPI group were invited by the trial manager to contribute to all relevant tasks. The tasks were outlined via emails, with suggested deadlines for response to agree/decline the request. If they agreed to provide feedback/support to the task, the trial manager either setup a phone/videoconference or emailed further details (mode of contact was chosen by the families). Most contact was arranged for the evenings and/or weekends to avoid conflicting with school/work time. The task was then discussed in more detail, with opportunities for questions. Feedback on the task was either provided to the trial manager in real-time (i.e. a discussion over phone/videoconference) or in written form at a later agreed date. In all instances, the trial manager provided summary feedback explaining how their support had informed the development or design of the trial documents/materials/procedures.
The families were informed they could decline any request and they would still be part of the PPI group, unless they decided they did not want to have continued involvement. No families withdrew from PPI support.
PPI input was conducted throughout the trial, from start to finish. The key areas of input were:
- refining the therapy from Swedish to English
- advising on how to deal with Christmas and other holiday breaks in providing continuing access to the ORBIT intervention
- developing patient-facing documents and reviewing outcome measures
- advising on promoting continued engagement/recruitment to the trial (with specific input from Tourettes Action)
- inputting into interview schedules as part of the process evaluation
- supporting analysis of interview data
- reviewing dissemination plan and contributing to lay dissemination materials.
We also had a lay member on our TSC, Neelam Gurvav, a parent of a young person with tics, attended all of our TSC meetings, and she provided excellent input throughout.
Results
The resulting impacts of PPI will be discussed in relation to each objective.
Refining the therapy
Once the initial content of the interventions had been finalised, both interventions were sent out to review with two parent–child dyads from the PPI panel and two members of the MindTech Involvement team.
The two parent–child dyads were given a login respectively to the child and parent/supporter interventions. Each PPI member was provided with a feedback form (Microsoft Word format) to complete alongside going through the intervention. They were provided with 4 weeks to review the intervention.
Several changes were made to the two interventions (child and parent/supporter) based on feedback:
- inclusion of a ‘warning’ at the beginning of both interventions that the intervention would be about tics, therefore potentially making the child tic more when engaging with it (suggestibility of tics)
- reducing length of some videos and/or cutting one video into two videos
- ensuring all audio on videos was the same volume
- reducing text on lengthy webpages
- correcting spelling errors/typos and clarifying sentences and text lost in translation
- both interventions having three ‘characters’ with tics – feedback suggested that these were better as cartoons and not as photos.
These changes resulted in the final versions of the ERP and psychoeducation interventions described in Chapter 2.
Therapy breaks
As a result of consultation with our PPI panel, there was unanimous agreement that continuing therapy over festive periods such as Christmas would be too difficult for families. Issues around changes in routine, family visits and festive activities would make it difficult for families to log in. As a result of this, we placed a substantial amendment for REC/HRA approval in April 2018, which allowed the 10-week intervention to be delivered over a 12-week period, with a maximum of 10 weeks of therapist support. This allowed families to take a holiday or deal with unexpected life events.
Developing patient-facing documents and outcome measures
Our PPI panel reviewed all of our patient-facing documents, such as participant information sheets, therapy manuals and supporting documents. The main feedback was to make these as brief as possible; as such, where possible, information was removed or shortened and subheadings were put in to break up the information.
Our PPI panel also reviewed our chosen outcome measures. The group was informed as to which documents were standardised and so changes to wordings or removing specific items were not possible and which measures had been created by the trial team and were able to be changed. The group was supportive of all of the chosen outcome measures, indicating that they felt they were all important. The group particularly commented on the need to measure other outcomes (such as anxiety and low mood). As such, no changes to outcome measures were made based on the PPI review.
Encouraging engagement and study uptake
As noted previously, potential participants could register their interest in the trial through the Tourettes Action webpage as well as through our own study webpage. Thus, our PPI partners were a key part of trial recruitment. Indeed, 251/445 (56%) people who registered interest in the trial were self-referred.
Alongside our charity partners, Tourettes Action, we developed a social media strategy to ensure that the ORBIT trial was maintained in the public eye. Consultation with our PPI panel indicated that families would rather view/listen to a video blog than read a written blog.
Our research team created monthly video blogs and ‘spotlight on researcher’ profiles that were hosted on the Tourettes Action webpage. Each member of the research team completed their blog, and in their blog they informed the public what it is like being a researcher/therapist/trial manager.
This approach kept the public informed about ORBIT and served to educate on what research entails. Anecdotally, the team noticed an increase in self-referrals to the trial after a blog was posted, suggesting that the approach may be successful in promoting recruitment to trials.
When we reached key milestones, such as finishing recruitment, we also created video blogs to inform the public and wider audience of this. These were hosted on the Tourettes Action webpage and on our study webpage.
Interview schedules
As part of the process evaluation, we developed interview schedules in order to carry out interviews with a subset of children and parents involved in the trial. The interview schedules were drafted and underwent initial revision from the process evaluation research team. The team then sent the revised drafts to parent–child dyads of the PPI group – including two young people with tics – for feedback. Most of the comments from the young people related to making sure the questions asked were simple enough to be understood by the younger participants. After their feedback, the schedules were revised accordingly. The PPI input at this stage was invaluable, as it ensured that the questions were understood by children as young as 9 years of age.
Analysing data
We also approached two members of our PPI group to help analyse a proportion of the interview data emanating from the process evaluation; however, due to time constraints and the COVID-19 pandemic, they were not able to do this task. Moreover, the young people from our PPI group felt that the analysis would be too complex to carry out with no formal training.
Dissemination
Members from our PPI group – including two young people with tics – have provided extensive feedback on aspects of our dissemination strategy.
This PPI panel supported our researcher in co-creating a lay summary of the main trial findings. This summary document has been emailed to all participating ORBIT families and includes a link to an online video blog. The video blog provides a brief, spoken summary of the results aimed at a lay audience.
The PPI panel also advised that video blogs were again preferable over written summaries for wider dissemination. The PPI panel reviewed draft video scripts, provided feedback and approved the final product. To achieve this wider dissemination, the video blogs were hosted on the Institute of Mental Health social media accounts (Twitter, webpage) and on those of Tourettes Action.
We had planned to host a free, online event to be held on an evening and aimed at families to discuss the findings of ORBIT. However, on advice of our PPI panel, this has been placed on hold. The PPI group felt that until the intervention was closer to being offered outside the trial, families may be disappointed or frustrated to learn that this clinically effective intervention is not available in routine care. We have thus postponed this event until we are closer to achieving this aim. The agenda is drafted and includes one of our PPI parent–child dyads giving a short talk where they will discuss their experiences of being involved in the ORBIT trial.
We also invited ORBIT PPI members to the MindTech National Symposium in December 2021 as part of their continued development and training on digital technologies in mental health. At this event, there was a talk provided by Seonaid Anderson (formally of Tourettes Action) that was dedicated to the PPI activities. The talk detailed the methods of involvement and outcomes/impacts on the trial and was well received.
Equality, diversity and inclusion
Participant representation
Our participants were predominately white, British; however, this is reflective of the clinical population presenting to clinical services with tics in England. We adopted a wide approach to recruitment, including self-referral via adverts from national charities. By allowing referrals from people not being seen by specialist services we hoped to broaden the range and characteristics of people who could participate. We also noted that although our participants did encompass a range of socioeconomic groups, overall our group was of slightly higher socioeconomic status than the general population. We anticipate that this may be due to having one of our research sites in London, where people are likely to have higher earning status than those in rural/northern areas. We did, however, have patient identification sites in the north to try to ensure wider representation. Our process evaluation revealed that treatment outcome was not significantly affected by the sociodemographic or clinical characteristics of participants. Thus, participant characteristics did not mediate/moderate the study outcome.100
Research team
Our research team included patients, public members and research members of different ethnicities and socioeconomic backgrounds and with representation from all areas of England. Our PPI activities have been described. Our team also included a mixture of male and female clinicians and academics, including senior and junior members. Junior members were provided with training and experience in clinical trial design, management and PPI.
Discussion
Patient and public involvement was an integral part of the ORBIT trial, integrated from start to finish and involved in all key steps.
Our two core channels of PPI involvement were through the national charity Tourettes Action and through our panel of PPI parents and young people. We believe combining both was a particularly effective approach in PPI. Tourettes Action allowed us to reach a larger audience and to develop a national profile with regards to the trial. This platform was pivotal in not only facilitating recruitment, but also educating the public on the aims and outcomes of the trial. In contrast, our PPI panel allowed us to get in-depth critical opinions and advice on key study documents and dissemination materials, as well as informing our dissemination strategy. Both approaches bought a different but complimentary angle to our PPI.
The key areas where PPI was particularly important were recruitment, retention and dissemination. Facilitating self-referrals into the study via Tourettes Action contributed over half of the total number of initial referrals into the trial and was a key part in achieving the recruitment target within time. Furthermore, not only did our PPI provide direct input into the patient-facing documents, they also co-developed the strategies for engagement/retention and dissemination. The success of this is likely to be reflected in our excellent recruitment rate and retention to the primary outcome, continuing until the 18-month follow-up.
We would have liked to have had face-to-face communication with our families as well as attendance at our team meetings. Although we did offer this, it was not taken up by any of our PPI members. Reasons for this included issues with time to travel, taking time away from school/work and not feeling comfortable with ‘ticcing’ during meetings or around strangers. However, by being flexible in our communication style with families we were able to keep our original PPI panel throughout the trial. We believe that offering the mode of communication that suited them at a time that suited them (usually outside traditional work hours) and providing sufficient time and feedback on tasks were key to facilitating this. It is also important to note that the members of our PPI group were reimbursed for all of their activities in line with INVOLVE guidelines on PPI payments.
There were some limitations in what we achieved with regards to PPI involvement in terms of analysis. As mentioned, we approached members of our PPI team to analyse a subset of interview transcripts for the process evaluation; however, this was not feasible as a result of the COVID-19 pandemic, lack of training for the young people and time constraints. There have been studies in which PPI members have been formally trained in qualitative analysis and in which the analysis is conducted collaboratively with researchers in face-to-face workshops. Due to the pandemic and time constraints, such an approach was not feasible. In hindsight, we could have conducted workshops via videoconferencing; however, this would have been difficult to achieve due to the complexity of the qualitative analyses.
Despite this, the PPI involvement in ORBIT was largely successful. Dr Seonaid Anderson, who supported the trial as part of her previous role in Tourettes Action as well as her position as a freelance neurodiversity consultant (Neuro-Diverse | Dr Seonaid Anderson, neurodiversity consultant), summarised the PPI on ORBIT:
- HUGE collective research AND clinical expertise in the ORBIT team
- genuinely include people in the different stages of their research, not just to have them as participants
- actively listened to my expertise in terms of how to reach potential participants
- were creative and adaptable in their style of reaching an audience
- innovative in creating videos to explain what the study was about and videos about results.
Dr Seonaid Anderson, December 2021
Reflections
Overall, PPI has been a crucial element of the ORBIT trial, which has been integral to the study’s design, delivery and dissemination. The impact of this involvement was overwhelmingly positive, and there were no negative outcomes associated with PPI; however, we note the limitation regarding the involvement of the PPI in the analysis of the intervention. Having an adequately costed PPI budget was crucial to support the delivery of this important aspect of the work.
Our research team consisted of a combination of researchers with previous experience of PPI (such as the trial manager), alongside more junior members with limited or no experience. Utilising the trial manager as the main point of contact enabled junior members of staff to learn about PPI involvement and thus supported their development as researchers. In the initial application, we had suggested that co-applicant S. Brown would facilitate most of the PPI activities. Brown provided support and guidance on PPI where needed; however, given the level of involvement that our PPI group had, it was more efficient and effective for this to be led by the trial manager, who had a more comprehensive understanding of the trial progress and need for PPI.
Although we did not have any set meeting dates, we still were able to facilitate and benefit from regular PPI input, and the use of monthly newsletters ensured that the PPI families were kept abreast of the trial progress. This approach of more flexibility with meetings/task involvements was novel to our research team, with previous research following more specified meetings and areas for input. However, we feel that ORBIT was a good example of true PPI collaboration that facilitated meaningful involvement. The fluid approach was preferred by our PPI families, which may be a result of the characteristics of our group (i.e. young children with tics and potential anxiety regarding participating in group meetings).
Our PPI partner, Tourettes Action, particularly our key contact, Dr Seonaid Anderson, provided the expertise in the topic area and lived experience of the population to facilitate PPI involvement. The charity played a key role in PPI and in offering a route to identify families to form the PPI panels. This meant that PPI was straightforward, present and meaningful.