Conducting public involvement in dementia research: The contribution of the European Working Group of People with Dementia to the ROADMAP project

Abstract Background Dementia outcomes include memory loss, language impairment, reduced quality of life and personality changes. Research suggests that outcomes selected for dementia clinical trials might not be the most important to people affected. Objective One of the goals of the ‘Real world Outcomes across the Alzheimer's Disease spectrum for better care: Multi‐modal data Access Platform’ (ROADMAP) project was to identify important outcomes from the perspective of people with dementia and their caregivers. We review how ROADMAP's Public Involvement shaped the programme, impacted the research process and gave voice to people affected by dementia. Design The European Working Group of People with Dementia (EWGPWD) were invited to participate. In‐person consultations were held with people with dementia and caregivers, with advance information provided on ROADMAP activities. Constructive criticism of survey content, layout and accessibility was sought, as were views and perspectives on terminology and key concepts around disease progression. Results The working group provided significant improvements to survey accessibility and acceptability. They promoted better understanding of concepts around disease progression and how researchers might approach measuring and interpreting findings. They effectively expressed difficult concepts through real‐world examples. Conclusions The role of the EWGPWD in ROADMAP was crucial, and its impact was highly influential. Involvement from the design stage helped shape the ethos of the programme and ultimately its meaningfulness. Public contribution People with dementia and their carers were involved through structured consultations and invited to provide feedback on project materials, methods and insight into terminology and relevant concepts.


| INTRODUC TI ON
Alzheimer's disease is the most common cause of dementia, accounting for 50%-70% of cases. 1,2 It is associated with a range of outcomes, including memory loss, language impairment, increased dependency, reduced quality of life and personality changes. 3,4 Research has suggested that outcomes selected for clinical trials in dementia might not be important to people with the condition and their caregivers. [3][4][5][6] Selecting outcomes of importance to key stakeholders reportedly improves the relevance and quality of research results, makes studies more acceptable to potential participants and increases participant recruitment and retention. [7][8][9] One of the goals of the 'Real world Outcomes across the Alzheimer's Disease spectrum for better care: Multi-modal data Access Platform' (ROADMAP) project 10 was to identify the outcomes of Alzheimer's disease that are most important to people with the disease and their caregivers, understand what would constitute a meaningful delay in disease progression from their perspectives and create a disease progression matrix that highlights the outcomes of greatest importance at the various stages of the disease (i.e. from pre-clinical Alzheimer's disease to Alzheimer's dementia). Findings would inform the development of a data platform that integrates multiple sources of real-world data related to Alzheimer's disease, called the Data Cube, 11 to enhance researchers' understanding of disease progression in the real world. To achieve this goal, ROADMAP involved people with dementia and caregivers in the research process, not only as participants but also as advisors. This is in keeping with the concept of 'Public Involvement' (PI) (formerly known as Public and Patient Involvement [PPI]). 12 PI is not a specific method, but an approach to involving people in research other than as research participants. It is about conducting research with and by people with a particular condition and/or members of the public, rather than research being done to, for or about them. [13][14][15][16] PI can be seen as a 'means to an end', in the sense of attempting to improve the quality, relevance and utility of research (from both research and user perspective), but also as 'an end in itself', in the sense of being linked to democracy (e.g. democratic decision making, public accountability, legitimization and transparency), people having rights (e.g. a right to voice, a right to be involved in research relevant to one's own condition) and ethical principles of justice and fairness. 12 Involvement can occur at all stages of research, including prioritization of studies, 17,18 design and management of research, 19 identification of health outcomes, 20,21 data collection and analysis 22,23 and dissemination of findings. 24 Over the last few decades, there has been an increased interest in involving people living with dementia in PI in dementia research. 25 Several national and European-funded research projects have developed different approaches and methodologies to achieve this, [25][26][27] although information regarding the impact of PI activities for researchers, the people involved and society is still lacking. From the perspective of people affected by the condition, PI in dementia research is considered as fundamental and should become the norm, but efforts still need to be made to ensure PI is inclusive of a diverse set of people. 28

| Aims
This article reviews how ROADMAP's PI consultations 1. shaped the design and content of the stakeholder surveys, 2. had an impact on other issues related to the research process in general and 3. gave voice to people with dementia and caregivers.
We believe it is a success story for meaningful and active involvement of people with dementia and their caregivers in the research process.

| People with dementia
People with dementia from the European Working Group of People with dementia (EWGPWD) took part in this consultation.
The EWGPWD is composed of people from different countries and was set up by Alzheimer Europe in 2012. 29 The members of the group had all been nominated by their national Alzheimer Associations. They work in collaboration with Alzheimer Europe, contributing towards Alzheimer Europe's work and research projects in which it is involved. The group works to ensure that the Public contribution: People with dementia and their carers were involved through structured consultations and invited to provide feedback on project materials, methods and insight into terminology and relevant concepts.

K E Y W O R D S
Alzheimer's disease, dementia, patient empowerment, patient engagement, patient involvement, public participation activities, projects and meetings of Alzheimer Europe duly reflect the priorities and views of people with dementia. All members of the EWGPWD are in the mild or moderate stages of dementia and have capacity to understand what is being asked of them.
Alzheimer Europe was a member of the ROADMAP consortium, and the EWGPWD agreed to be involved in this consultation activity as part of their PI remit. Overall, nine people with dementia (eight female and one male) from the Czech Republic, Germany, Portugal, the Republic of Ireland, Sweden and the United Kingdom were involved in the consultations.

| Informal caregivers
Members of the EWGPWD are typically accompanied to meetings by a relative, friend and occasionally by a member of staff from an Alzheimer Association. However, on this occasion, the accompanying individuals were invited to be involved in a separate consultation for caregivers. Overall, seven caregivers (five female and two male) from Portugal, the Republic of Ireland, Sweden and the United Kingdom were involved in the consultations.

| Planning and conducting the public involvement consultations
Consultations with the EWGPWD and their caregivers were held in Luxembourg on the 4th and 5th of September 2017 and were conducted in accordance with Alzheimer Europe's position on PI, 16 which was developed in collaboration with the EWGPWD and the INTERDEM research groups. 30 Alzheimer Europe has great experience in preparing and conducting PI work with people with dementia.
The schedule was developed by Alzheimer Europe in collaboration with ROADMAP members from the University of Edinburgh. In addition, the Dementia Engagement and Empowerment Project (DEEP) guidelines 31 were taken into account when developing the materials and the schedule.
Involvement consisted of two concurrent consultations (one for the members of the EWGPWD and the other for their caregivers) that ran over the course of 2 days. Consultations were conducted in English, which is standard practice for the EWGPWD.
All people involved received information about the project and the topics to be discussed 2 weeks prior to the consultation. In addition, on the first day of the consultation, a presentation about ROADMAP, relevant concepts in the project (e.g. real-word evidence and outcomes in research) and why PI was important in the project, was provided.
The consultations were organized in such a way as to ensure that the EWGPWD and their caregivers were actively involved in the design and interpretation of ROADMAP's research activities. They facilitated productive interaction between the research team and stakeholders and informed and guided the translation of the results.
The key discussion topics aimed 1. to provide constructive criticism of the stakeholder surveys, relating to its content, layout and accessibility and

| Synthesis of discussions
With the authorization of all members of the EWGPWD and their caregivers, all consultation activities were audio-recorded and notes were made. This enabled the moderators of the consultations to be fully involved in the discussions and to be attentive to possible needs of members of the EWGPWD (e.g. signs of fatigue, irritation or difficulties understanding). After the consultation, recordings were transcribed verbatim. The notes taken during the meeting and photographs of the different activities (e.g. flipchart summaries and prioritization activity) were also used for the analysis.

| Survey refinement
The survey had been drafted with the intention that it would be completed online and in paper by people with dementia, their caregivers and health-care professionals throughout Europe. It included a list of outcomes related to Alzheimer's disease, with the intention of asking participants to rate how important each outcome was with regard to assessing a meaningful delay in disease progression, in addition to questions relating to disease stage, caregiving duties and demographics. 33 The EWGPWD provided constructive criticism of the survey design and content. An important issue addressed by the members of the EWGPWD and caregivers was related to the terminology used to refer to Alzheimer's disease in the survey. Important conceptual changes were made in this regard based on the feedback provided in the consultation. The original survey used the terms Alzheimer's disease and dementia. Members of the EWGPWD felt this was confusing and that many people with dementia and caregivers would not be familiar with the difference between both terms (i.e. new conceptualization of Alzheimer disease including pre-dementia as well as dementia stages). It was therefore suggested to use the term dementia and, if necessary, to separately refer to the pre-dementia stages. It was also advocated to include people with any type of dementia in the study and not just Alzheimer's as often; in 'real life', people are not aware of their differential diagnosis, or their diagnosis is mixed (e.g. Alzheimer's and another type of dementia). Another important concern was in relation to the accessibility of the survey.

| Enhancing our understanding of key concepts
The consultations provided relevant input for the ROADMAP project regarding the topics of important outcomes, disease progression, staging of dementia and meaningful delay in dementia progression. It was recommended that the researchers should consider the following series of issues when thinking about disease progression.

Assessing and measuring progression
Overall, the groups were of the opinion that brain scans can be useful for confirming disease progression and diagnosis, but they currently hold little meaning in terms of experience of symptoms in everyday life, hence lacking tangible benefit. Overall, this lack of connection between the experience of symptoms and the results of scans was considered to limit their usefulness when identifying disease progression.
In relation to measurement scales and tests (e.g. pen and paper tests), they highlighted that completing these tests can be disheartening for some people (like '…watching yourself decline'). This, it was suggested, can lead to a loss of confidence, whilst offering little clarity with regard to disease progression. They also raised the issue that such scales and tests often focus solely on cognitive measurements, with other important outcomes being overlooked, that is assessing cognition in the absence of everyday functioning.
I have a test every six months… And I've actually just told them that I'm not doing it anymore, because every test I've done every six months it's never even, I'm not asking for miracles, but it's never even been the same.
I always lose a point or a couple of points, so I've said right, I don't want to lose any more confidence, so I'm not bothered now. I know it's progressing, yeah.

Personalization and building the individual's story
People with dementia and caregivers suggested that assessing progression should be personalized, as everyone's story is different and there is huge variability in symptom experience. Caregivers suggested that living with the person with dementia is important for assessing and understanding progression, as new and recurrent behaviours can be observed. This allows for differentiation between true progression and simply having a 'bad day'.

Taking a holistic perspective of people with dementia
It was highlighted that when looking at the progression of dementia, other comorbidities and impairments should be taken into account and, in particular, the potential impact of these on the progression or the perceived progression of the disease. This is important as they can directly influence assessment, for example poorer cognitive or functional performance due to a comorbidity, as opposed to dementia.

| Staging dementia
It was generally perceived that existing scales to stage dementia do not always capture the individual's lived experience. However, there was a general consensus that the terms 'mild -moderate -severe' adequately captured the stages of the condition in a way that could be easily understood by people with dementia, as they provide a good approximation of how far a person's dementia has progressed.
A number of issues were raised in relation to staging disease progression, which should be considered by researchers, discussed in turn.

Inconsistency and susceptibility to external influences
Some people felt that judgement of a person's stage can differ between clinicians, leading to inconsistencies. Further, assessing stage using clinical measures only provides a snapshot of the condition, which can be heavily influenced by factors like mood and the environment in which the assessment took place. It was concluded that the assessment of stages should therefore be tailored to the individual, given differences in the type of symptoms experienced and rate of progression.

Implications for daily life
The groups pointed out that staging dementia has implications for everyday life, such as the loss of a driving licence, self-determination, freedom of movement and management of finances. In some coun- couldn't function and work in his 50s. (Caregiver)

Preferences for new staging frameworks
The EWGPWD and caregivers stated that new staging frameworks should account for fluctuations or variability in a person's condition, as current frameworks can be rigid and linear. This could be achieved by providing more flexibility between the boundaries of the stages.
The EWGPWD's feedback on the conceptualization of stages was fed into the survey design 33 and ROADMAP's Data Cube 11 as it was considered useful and meaningful to people with dementia and their caregivers.

| Meaningful delay
Three relevant, interconnected issues were addressed in this context, namely delay before the onset of dementia, delaying the progression of dementia and the balance between longevity and quality of life.

Delaying the onset of dementia
It was felt that meaningful delay is a complex concept, which might not be clearly understood by some people. It would be important to clearly distinguish between cure, prevention and meaningful delay.
Meaningful delay was described in positive terms and often linked to the idea of 'enjoying or having some extra time'. Important issues when talking about a meaningful delay of the onset of dementia were related to the certainty that the person would eventually develop dementia, how long such delay would be, the balance between potential risks and benefits (e.g. side-effects, having a faster deterioration after the delay, better quality of life/independence) and the particular circumstances of each person (e.g. comorbidities, age, frailty). It was highlighted that the meaning of the delay may also depend on whether the person is or is not familiar with dementia.
Okay. It's to delay the onset. Onset to me means the

Longevity vs quality of life
Whilst some common themes were detected in relation to the concept of meaningful delay, the groups agreed that an individual's own set of values was of priority when determining choices related to their life and treatment and believed that respect for such individual differences was paramount.

| Democracy, rights and justice
The EWGPWD and caregivers were keen to have their voices heard in the ROADMAP study (in the sense of having a 'right to voice'), as well as sharing their experiences and perspectives based on living with dementia with the ROADMAP researchers. This was seen as involving mutual respect and being based on the principle of reciprocity. Table 2 provides a summary of feedback provided by members of the EWGPWD when asked to reflect on the PI process and to identify the most important messages they wished to pass on to ROADMAP researchers. 34

| Key outputs and deliverables
The input of the EWGPWD and their caregivers has been incor-  being involved in this way in the project. In addition, the discussions helped the researchers to understand better the perspectives of people affected by dementia with regard to issues that were important for the project, such as meaningful outcomes, progression and delay of the disease, and also to identify issues and concerns that should be taken into account when carrying out research. The progression and potential delay of dementia are very important and sensitive topics for people affected by dementia.
Assessing the progression of the disease is important and necessary, but the potential impact that this may have on a person's well-being and quality of life also needs to be taken into account and addressed. The potential delay of the disease can give people hope, but it is also important that people understand clearly what it entails and its potential limitations. The individual experience and considering the person as a whole (including comorbidities, social and personal factors) were key messages for ROADMAP researchers working on these topics.

Involvement of people with dementia and caregivers in research
through PI is paramount, but requires careful consideration of how to plan and conduct it. This includes, amongst other issues, ensuring that relevant expertise, time and budget are available.

ACK N OWLED G EM ENT
The authors would like to thank the current and past members of the European Working Group of People with Dementia and their carers/ supporters who contributed towards this work.

DATA AVA I L A B I L I T Y S TAT E M E N T
Data sharing is not applicable to this article as no new data were created or analysed in this study.