The results are presented in three sections. Section one provides results from the stages outlined in Figure 1 (Fig 1), section two shares the perspectives of ASPREE-XT participants and other stakeholders, and in section three we describe how participant involvement impacted study design in seven ways. We quote participants directly, assigning each a unique number. Figure 2 summarises the entire process, results and impacts (Fig 2). For quantitative data from the telephone interviews, see Additional File 3.
Section 1: Results from stages 1-4
Results from Stage 1: Planning
Data from the study team suggests that the input of the participant advisor during the early co-design stage had a number of benefits, in particular in identifying the best approaches for engaging the broader ASPREE population; improving the wording of participant information resources and improving question design for the interviews.
Results from Stage 2: Recruitment and telephone interviews
After reading the advert in the newsletter, 76 ASPREE-XT participants agreed to participate in the process. We interviewed 59 people by telephone, with the remaining 17 not responding to a number of follow-up calls. Participants were a mixture of people living in metropolitan and rural areas.
Interview participants expressed a strong interest in receiving results from genetic research. The strongest preference was for genetic results of medical significance (rather than ancestry or drug-response).
All telephone interview participants expressed willingness to provide feedback throughout the process of planning the proposed MGRS. While most participants understood the concept of involvement, 9 of the 20 analysed expressed self-doubt about how they could be involved. Of 20 analysed telephone interviews, five participants seemed unclear about what tasks they could be involved in, suggesting the concept of participant involvement was new to them. Six participants sought clarity about what was expected of them when being involved, or discussed the skills and knowledge required. Further, participants stated that the goals and processes of involvement needed to be clear in order to avoid “spending too much time” and were willing to be involved as long as the task had purpose and was not “just for the sake of chatting” (P1).
During the telephone interview stage, the interviewer initially “struggled to explain” the concept of involvement (as distinct from participation in an active study) (MS). Co-designing and making changes to the language used in the telephone interviews (version 1.5 onwards) appeared to improve participant understanding of the concept of involvement. Changes included providing a clearer definition and pre-question script and including actual examples of involvement (such as overseeing ethical decisions about data access), thereby avoiding jargonistic abstract concepts such as ‘involvement’.
Table 3: Summary of data from telephone interviews
Questions (closed)
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Results
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% (number)
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1. Do you think participants like yourself should be involved in helping design research projects, or just researchers?
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Just researchers
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39 (11/28)
|
Participants involved
|
46 (13/28)
|
Unsure
|
14 (4/28)
|
2. Would you be willing to provide feedback and share your views and perspectives by commenting throughout the research process?
|
Yes
|
100 (32/32)
|
No
|
0 (0/32)
|
3. Would you like to be more involved in study design rather than just participating?
|
Yes
|
65 (11/17)
|
No
|
12 (2/17)
|
Unsure
|
23 (4/17)
|
4. Would you be more or less likely to participate in research if participants were involved in design
|
More likely
|
25 (2/8)
|
Less likely
|
0 (0/8)
|
Unsure
|
13 (1/8)
|
Wouldn’t influence participation
|
13 (1/8)
|
5. What is your preferred mode of communication for being involved
|
Face to face event
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25 (15/59)
|
Post
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31 (18/59)
|
Online discussion
|
8 (5/59)
|
Online questionnaire
|
17 (10/59)
|
6. If genetic research results were made available, which types of genetic testing would you be interested in? *
|
Medical
|
97 (32/33)
|
Ancestry
|
97 (32/33)
|
Drug response
|
94 (31/33)
|
7. If only one option for genetic testing was available, which one would you prefer?
|
Medical
|
65 (11/17)
|
Ancestry
|
12 (2/17)
|
Drug response
|
6 (1/17)
|
No preference
|
18 (3/17)
|
*Note that percentages indicate the percentage of participants who responded to that particular question with that response. On question six, participants could select multiple answers, so totals do not add up to 100%. The number in brackets represents (number of people with that response/ number of people asked that question).
Results from Stage 3: Event
Eighteen of the invited 76 participants attended the face-to-face event. Participants were a mixture of people living in metropolitan and rural areas, with some travelling for a number of hours in order to attend. When specific tasks were discussed at the event, 10 participants expressed interest in being involved in recruitment and communication tasks, 7 in data access decisions, and 11 in ethical decisions. When surveyed after the event, the study team reported that involving participants positively impacted the proposed study design by improving the research objectives, developing protocols, influencing funding decisions and improving ethics applications. Videos of the event discussions and interviews with participants will be shared in the public domain.
Results from Stage 4: Evaluation and reflection
Participant feedback from the workshop event was analysed, with participants reporting it as a positive experience, one describing it as “brilliant” (P7). When surveyed, all members of the study team reported that the event achieved its intended aims and was a positive experience. No negative impacts were reported from any participants or study team members at any stage of the process.
One study team member described that through involving people, their perceptions “significantly changed” as they learned how participants could have “valid, interesting, and often novel ideas that researchers may not have considered”. The lead investigator (PL) noted:
“What I learnt is that sometimes the researchers’ assumptions about certain things can be off or even incorrect, and that researchers can miss critical points that are important to participants and the study”.
During the process, both participants and study team members reported changed views about the value of involvement in research, demonstrating ‘transformative learning’ and co-construction of knowledge39,50,51. One member of the study team (MS) stated in the survey at the end of the process:
“I came into this project wondering how participants could be involved…I was unsure how people without a science or health background could contribute. I have learned that participants have valid, interesting, and often novel ideas that researchers may not have considered. My perceptions of involving participants in planning medical research has significantly changed”
A number of significant learning points were identified by the study team when responding to the question “do you have any advice to other researchers planning involvement for their research”. Significant learning points are summarised in Table 4.
Table 4: Summary of learning points from study team data
Summary of learning points from study team data
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1. Fund and prioritise involvement, make it a requirement
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2. Ethics processes take time, but can improve plans
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3. Know your audience – don’t make assumptions
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4. Value diversity in experience and knowledge
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5. A supportive team improves the experience for all
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Cost and value
The entire process of involving people was estimated to cost $10,000 AUD, including staff time, catering and event venue hire (see Additional File 1 for more detailed information). The value of the process was summarised by the lead investigator who stated “I learnt a lot from the process and am very glad we made the effort”.
Section 2: Perspectives of ASPREE participants and other stakeholders
Recurring themes were identified by the study team from the data sources analysed, and seven specific impacts were reported as a result of involving participants (summarised below). The analysis is divided into two sections: (1) participant views about the proposed study, (2) stakeholder views about involvement in the proposed study.
1: Participant views about the proposed study
Participants were very positive about the proposed MGRS, with one stating ‘I think it sounds very good’[P20]. Altruism was a primary motivator for participation, with participants suggesting that outcomes of a MGRS could include benefits to themselves (personally and for their families); improving healthcare more generally; and the potential for saving lives, preventing diseases, improving quality of life, and improving future research.
Participant views on types of information they would like returned from genomic research and how the study should be funded (commercial funding versus public funding) were diverse and did not always align with the study team’s prior expectations. For example, two thirds of event participants wanted access to their own genomic data, which was described by the lead investigator as ‘very at odds with the current system’ (PL).
2: Stakeholder views about participant involvement in the proposed study
Participants were supportive about being involved, with all participants supportive of being involved by providing feedback throughout the research process (100%, 32/32), with a typical participant response being ‘I’d be happy to be involved’ [P3]. Views about enablers were shared in three of the 20 interviews coded, by all 18 of the event participants and all study team members surveyed. Views about barriers were shared in eight of the interviews with participants that were coded, and by half of the study team surveys. Further data is categorised in Table 5. Additional mapping of preferences for involvement using the STARDIT-PM tool can be found in Additional File 1 25.
Table 5: Enablers and barriers for involving participants
Enablers
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Quotations
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Barriers
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Quotations
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Financial remuneration for people’s time; financial support for travel and accommodation
|
Running a business ‘limits me and my time’ [P15]
|
Living in rural areas and travel logistics a barrier to participation in face-to-face events
|
‘I’d like to be involved online rather than face to face because of travel difficulties’ [P6]
|
Learning and development opportunities for participants and researchers
|
‘if you tell me what would be useful, I could do it’ [P13]
|
Poor explanation of abstract concepts such as ‘involvement’, which can be jargonistic
|
‘there seemed to be confusion, or a lack of understanding of what this 'involvement' would actually look like’ (MS)
|
Small groups at events gave more people a chance to share perspectives
|
‘Small group’ discussions at the event ‘worked well’ (Study team member)
|
Self-doubt about their skills or knowledge mean they don’t think they could be involved
|
‘I’d probably ask a stupid question’ [P2]
|
Early notice of events
|
‘give me enough notice’ [P10]
|
Some people not comfortable being part of a face-to-face group
|
‘I get very uncomfortable in a group of people’ [P12]
|
Clear information about timings and time commitments, frequency of involvement and available support
|
Ensure ‘people are advised what’s going to happen at the workshop’ [P8]
|
Lack of clarity about expected time commitments
|
Being involved ‘depends on what’s involved and time’ [P1]
|
Clear information about purpose and expectations of involvement, feeling their involvement has consequences
|
‘What’s the endpoint – what’s the goal?’ [P11]
|
Unclear about what tasks they could be involved in
|
‘I don’t know how I could but willing to help’ [P13]
|
Independent facilitator when working in groups (either face-to-face or online)
|
I’d feel more comfortable if I had someone who was facilitating [P14]
|
Face to face discussions ‘dominated’ by more confident or knowledgeable people (Study team member)
|
‘Participants from professional backgrounds to some extent dominated some of the discussions’ (Study team member)
|
Short events ensure people do not get fatigued
|
‘any longer and I think fatigue would have dampened the enthusiasm’ (Study team member)
|
People feel they have limited time, are busy with work or social commitments
|
‘I don’t have a lot of time left in life’ [P1]
|
Having access and literacy in using computers and online tools
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‘if I could negotiate [online discussions] I’d be happy to do that’ [P15]
|
Lacking access, literacy or trust in using computers, smartphones or online tools (including social media)
|
‘I’m hopeless with computers’ [P16], ‘I don’t have internet’ [P12]
|
A selection of flexible communication modes (such as face to face and facilitated online discussion forums)
|
‘I’d be happy to be involved – more online but if there was an occasional need to come into the city I’d be happy to do that’ [P3]
|
Only one mode of communication, such as expecting people to travel to events
|
‘online is often easier’, face to face only ‘as long as it’s not too far’ [P22], ‘travel distance is an issue’ [P20]
|
Involving people in research ethics and governance
|
‘ethics is the difference between right and wrong – you know what’s right and you don’t do what’s wrong’ [P18]
|
A ‘researchers know best’ attitude that doesn’t value the process of involving people (Study team member)
|
Researchers ‘don’t see the forest for the trees’ [P19]
|
Table 5: Enablers and barriers for involving participants
The data from Table 5 is organised into themes identified during analysis, with the quotations provided as illustrative of those themes.
The lead investigator stated “the feedback has been so valuable” and that it will be “built into the design” of future research. However, the study team also identified barriers to involving people which exist for researchers. One study team member reported that at the start of the project they were “unsure how people without a science or health background” could be involved (MS). They reported a personal shift during the involvement process from not understanding how participants could be involved and being concerned about “asking too much” to believing that, “with adequate resources (financial, training, time...), people can be involved in all aspects of genomics research”.
Other barriers identified by the study team included delays in obtaining ethical approvals for involving people and the cost of involvement in both time and money. One study team member reflected in the follow-up survey that they had “worried too much” about the time-burden of involving participants (MS). The concern of not putting further pressure on participants was a theme in email communication between the study team when making decisions to limit contact with participants throughout the process of involving them.
Survey data from the study team also suggests that adequate funding, a supportive team, involving participants in the very earliest stage of research planning and co-designing inclusive methods of involvement all contributed to the impacts reported.
Section 3: Impacts on study design from stakeholder involvement
Involving stakeholders in the co-design process impacted the study in seven specific ways. By asking for participants’ views on aspects of the proposed study design, the study team gained insight into participant preferences and opinions. While there was diversity in views, the process allowed the study team to improve aspects of the study design. As a result of the process described, the ASPREE-XT investigator agreed a number of changes to the proposed study design. These proposed changes as summarised as impacts in Table 6 ‘Summary of impacts on study design’
Table 6 ‘Summary of impacts on study design’
Impact on planned research
|
Summary of impact
|
1: Recruitment and sample collection
|
Recruitment and consent for the MGRS will occur online wherever possible, and saliva samples (rather than blood) will be sent by post to be used as biospecimens for DNA analysis.
|
2: Participant communication
|
A short video and ‘information pack’, which will explain the MGRS study, will be created to assist with recruiting family members.
|
3: Participant involvement in governance
|
Participants will be invited to be involved in overseeing governance, including funding decisions.
|
4: Data access
|
Study participants should be involved in controlling data access decisions and policies.
|
5: Communication and ways of involving participants
|
Participants will be included on study advisory groups, including for study recruitment and communication, data access and ethical oversight using multiple communication modes.
|
6: Provide feedback to participants about the research
|
Participants will be informed about the impact of the research, and how their involvement has affected the design and management of the study.
|
7: Create learning and development opportunities
|
Learning and development opportunities will be created for potential participants (developing knowledge of research), researchers (developing skills in involving people) and other stakeholders.
|
Impact 1: Recruitment and sample collection approach
A discussion about participants’ adult children being ‘time-poor’ highlighted the importance of a study design which minimised the time burden on younger generations. Only a third of participants thought their children would be willing to do a blood test. This allowed the research team to make a more informed decision about ‘trade-offs’ between the data that can be collected via blood or saliva, versus the potential effect on recruitment.
Impact 2: Participant communication
Most participants reported willingness to be involved in recruiting family members to a new study, if given appropriate information and supporting documents. Relatively inexpensive information resources, such explanatory videos, could have a significant impact on the success of the recruitment and the study as a whole. One participant also advised that information produced for participants by researchers can be confusing, and that laypeople can help simplifying it.
Impact 3: Participant involvement in governance
Event participants unanimously agreed that they should be involved in all aspects of the research, whereas 11 of participants from the 20 analysed interviews thought that they should be involved in study design. One interview participant felt only researchers should be involved as they are “the qualified people” (P8). Five of the interview participants expressed the view that non-researchers are required in research as they provide an important alternative perspective.
Participants had mixed views about commercial organisations funding research. Four event participants were against it, some were ambivalent, and the majority indicated no objections. One participant was concerned about the risks posed by involvement of commercial organisations with opaque “vested interests”, asking “what is in it for them?” (P2), referring to individuals and organisations with real or perceived conflicting, competing or commercial interests. Participants suggested that involving people in governance (including funding and ethical oversight) was a way of mitigating this risk.
Study team members reflected that public funding would be preferable to commercial funding, as the responses at the event suggested that a commercially-funded study might negatively impact recruitment. As a result, the study team altered the proposed design to involve participants in governance, oversight and funding decisions.
Impact 4: Data access
Participants shared the view that they would like different kinds of genetic information returned from the research (see Table 3), including personal medical, ancestry and pharmacogenomic results. Two-thirds of event participants wanted access to their own genomic data, and had mixed views about who else should have access. All event participants stated they were comfortable with their data being held by academics, and none were comfortable with data being held by a commercial company. However, one participant suggested not “ruling private companies out completely” from research (P9).
General practitioners (GPs) were generally trusted to access and interpret genomic data, but participants felt GPs should not have access to data that they did not. All but two event participants agreed they should exclusively control access to their own data, with those disagreeing mentioning cognitive decline as a reason for a co-managed access model.
Some participants had concerns about themselves or their biological relatives (especially offspring) finding out information they “might not want to know” (P6), or having it imposed on them.
Impact 5: Communication and ways of involving participants
Preferences for communication mode differed between interview participants and event participants. Interview participants preferred questionnaires via post (30%, 18/59), and face-to-face events (25%, 15/59), over online questionnaires (17%, 10/59) and online discussions (8%, 5/59). Event participants suggested face-to-face meetings were helpful but only when there was an “occasional need” (P3). Participant responses also suggested that limiting face-to-face events where possible (in favour of telecommunication) may mean involvement is more inclusive.
Event participants felt certain tasks (such as reviewing information) could be done “more online” (P3). Participants spontaneously suggested using online, moderated forums and suggested that these should be hosted by trusted institutions (such as universities) rather than commercial organisations, as some ‘don’t trust’ social media companies [P17].
Impact 6: Provide feedback to participants about the research
Event participants stated that keeping people informed about what has been learned from the study is a good way of keeping people engaged in the study and improving retention. For example, ASPREE-XT sends a regular newsletter to participants. Participants also stated that they would like to be informed about when their involvement has made a difference.
Impact 7: Create learning and development opportunities
Participants often stated their willingness but also their uncertainty about how they could be involved. Learning and development opportunities were identified as an area of involvement by both a participant advisor and participants. Learning and development opportunities will be need to be created for different stakeholder groups’ learning needs, including developing a knowledge of the research process for research participants and developing skills in involving people and community engagement for researchers.