In order to co-design the study from an early stage, a representative from AusEE was contacted, inviting the organisation to partner with the research team, with a representative from AusEE invited to be part of the study team and another invited to give feedback on the proposed study design. The representatives were involved in a number of tasks including reviewing and improving the written information, online survey questions, and the facilitation plan for the online discussion.
In order to facilitate comparison with other studies, we used the novel tool ‘Standardised Data on Initiatives - Alpha Version 0.1’ (STARDIT) to map preferences, plan involvement and report any outcomes from the process16. This included reporting co-design positively impacted the study. STARDIT is an open access data-sharing platform being developed to standardise the way that information about initiatives is reported across diverse fields and disciplines, including information about which tasks were done by who. Quantitative data was analysed descriptively and STARDIT was also used in parallel with a thematic analysis, which organised data into pre-defined ‘super-categories’ which allow consistent comparison with other data using STARDIT.
Learning resources were both co-created and selected by the investigator team, working in partnership with the Australian Genomics Health Alliance and co-refining the selection with potential participants, including working in partnership with potential participants and the Australian Genomics Health Alliance, using the outcomes of a landscape analysis of educational materials as a starting point for selecting resources17. Final resources were checked by a medical professional specialising in EoE.
Study Design
A participatory action research (PAR) paradigm was chosen to guide the process with co-design and reporting informed by guidance from a number of sources18–20. PAR is an umbrella term which describes a number of related approaches, including forms of action research which embrace a participatory philosophy and include ‘co-design’ and ‘co-production’ of research 21. It is a process whereby researchers, the public and other relevant stakeholders “work together, sharing power and responsibility from the start to the end of the project”,22 including knowledge generation and translation22. Elements of this study were co-designed in parallel with another similar study, therefore some aspects were inflexible and thus ‘co-refined’ rather than ‘co-designed’.
We used case study methodology to record and describe our experience involving participants in an online discussion about genomics research. The selection of the population for this case study was informed by a number of factors which were appraised by the study team, including ethical, pragmatic and population considerations23,24. One of the investigators (PL) had a professional relationship with the charity AusEE, which was used as a starting point by the study team to explore the appropriateness of the case study.
The case study is presented as an instrumental case study, where the purpose is to understand the particular case and can attempt to provide data that could produce useful generalisations by using inferences from the data 25(p109). We collected and analysed both qualitative and quantitative data during the involvement activities, informed by a number of frameworks and standards26,27.
In addition, we analysed other data from participant survey responses, online discussions, meeting notes, emails, surveys of the study team and reflexive diary entries of one member of study team (JN). Coding and thematic analysis of qualitative data was carried out by two authors independently (JN, KG) and checked by another author (PL), following best practices for enhancing validity in qualitative methods28. Two authors of this paper also shared personal comments in the online discussion (KG, SG), which have been anonymised using participant numbers. In order to aid analysis and comparison with other case studies, we used a novel way of sharing data in a standardised way (Standardised Data on Initiatives - Alpha Version 0.1) to map preferences for involvement, plan involvement, report and evaluate how people were involved in the PAR process, and how this positively impacted the study design16.
STARDIT is an open access data-sharing platform being developed to standardise the way that information about initiatives is reported across diverse fields and disciplines, including information about which tasks were done by who. It also offers a way to add updates throughout the lifetime of an initiative, from planning to evaluation and reporting any impacts. Authors from this paper were involved in co-creating STARDIT, and learning from this process informed the development of the reporting tool29.
Participants and recruitment
This study recruited participants from an existing online community managed by the Australian-based charity AusEE. A link to the informed consent form and learning resources was shared by AusEE on a closed Facebook group. We recruited people if they were either a parent, a carer, a partner, family member or loved one of someone with EGID who is under 18; or someone who was over 18 with EGID. If people gave consent, they were invited to complete an online pre-discussion survey and sent instructions for joining the online discussion.
Stages of research
There were four stages of the research process: 1. Co-design; 2. Recruitment and surveys; 3. Online discussions and 4. Evaluation and reflection. The multiple stages of the co-designed research are summarised in ‘Figure 1: Stages of research’.
Initial contact was made with AusEE in July 2018 and two investigators from AusEE were invited to join the study team (SG, KG). After ethics approval was obtained, an invitation to participate in the study was shared in the AusEE Facebook group and member newsletter in September 2018. This group has members who live in both Australia and New Zealand. Before being asked to give consent, the study was summarised in plain English and two learning resources were shared with participants in order to provide context to the study (see Additional File 1 ‘Data and analysis’).
The invitation to participate contained a link to the participant information document and the informed consent form. If people gave consent to participate, they were invited to complete an online pre-discussion survey, which had demographic data categories informed by other similar studies to allow comparison30.
Participants who gave consent were then contacted by email, with information about joining the discussion shared alongside relevant learning resources. Participants were also sent a follow-up survey after participating in the online discussion. Questions relating to ‘Who should be involved in research’ were the same as in the pre-discussion survey to allow comparison. Participants could choose from the categories outlined in Figure 2, with a change in direction towards more people being involved labelled as ‘widening’, the inverse as ‘narrowing’.
Online discussion methodology
We conducted an online discussion to maximise flexibility about when participants could contribute (for example, people in different time-zones or those with different caring responsibilities). A significant advantage of online discussion platforms is that they are accessible to a greater number of people. This creates a more equitable platform for people to participate, compared to a synchronous (simultaneous ‘real-time’) discussion.
We offered anonymity in online discussions to avoid people inadvertently disclosing sensitive or personal information, which may have serious implications, for example, impacting on eligibility for future health insurance. Participants could choose to use their real name and email address, or remain anonymous by using pseudonyms or temporary email accounts.
Before participants joined the online discussion, seven learning resources were shared with them. This included a short 60 second online video about the study context and purpose31, a one-page infographic summary of a scoping review about genomics research32, and a short two-page summary of genomics and contemporary research relating to EoE was co-created with AusEE, the study team and experts in genomics (see Additional File 1 ‘Data and analysis’)33.
An open-source software web application ‘Loomio’34 was installed on virtual machines hosted by an Australian Government initiative called ‘Nectar Cloud’35. Participants could securely log-in from anywhere in world and participants’ data was stored securely on servers physically located inside Australia.
Two people facilitated and moderated the discussion for 14 days (JN, KG), one of whom was a parent of a person affected by EoE (KG). Participants were invited to co-create their own boundaries for the group discussion at the start by reviewing provided statements and suggesting amendments. The facilitators judged when to introduce new topics (depending on the engagement with each topic) with the recommended schedule below in Table 1 used as a template.
Table 1: Online discussion overview
Question
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Day
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What do you understand by the word ‘research’?
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Day 1
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What do you understand by the phrase ‘genomic research’?
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Day 1
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Why do we do research?
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Day 2
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Which aspects of any future genomic research should be influenced by the following groups of people?
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Day 4
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What methods do you think could be used to involve those people in future genomics research?
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Day 6
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Do you have any ideas, thoughts or reflections that have not been shared yet?
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Day 7
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Discussion closed
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Day 14
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