New avenues within community engagement: addressing the ingenuity gap in our approach to health research and future provision of health care

Abstract

The proliferation of large biorepositories and the staggering advances in our ability to analyse large numbers of human genomes relatively quickly and cost-effectively have been important drivers in the move towards personalised medicine. However, our advances in the development of these tools have outstripped our performance in addressing the issues of ethics and consent surrounding health policy and governance of such repositories, the implications of proliferation of genomic information for the individual and its potential for misuse. Public consultation is urgently needed in the development of ethical guidelines for these emergent issues; however, effective strategies for facilitating community engagement and informed debate have been lacking. Public consultation through deliberative democracy is bringing an essential new dimension to public engagement in the genomic medicine era.

Keywords:

• public engagement
• biobanking
• personalised medicine
• deliberative democracy
• community consultation

Background

Revolutionary advances in science and technology over the last 20 years, including sequencing the human genome, have enhanced our understanding of susceptibility to disease. Similarly, the ability to collect and manipulate large volumes of data has permitted us to dissect the causes of disease at a population level. Population biobanks are key drivers of these new approaches to genomic science and are considered essential tools for the translation of biomedical research into real improvements in the delivery of health care. Biobanks challenge and extend the limits of traditional health care research beyond the “one project, one centre, one point in time” paradigm and raise a range of unique ethical and legal issues. Many of these issues have been widely discussed over the last decade, yet the issues of privacy, long-term consent, data management and access, and overall governance oversight remain hotly debated. Whilst these issues largely remain unresolved, there has been recognition of the need for novel strategies to engage populations in informed debate on these contentious issues. To be useful, such strategies need to engage communities in meaningful dialogue and engender public trust. One strategy gaining acceptance globally is deliberative democracy.

The demand for informed public debate in future health research and health care

Population biobanks have been hailed as one of the flagships of the personalised medicine era. If personalised medicine is to become the cornerstone of clinical care in the twenty-first century, population biobanks will need to become embedded not only in the medical research environment, but also in the health care system (Chalmers et al. 2013). What has become clear is that establishing and retaining public trust is central to the success of population biobanks. This necessity was demonstrated in the failure of Iceland's Health Sector Database, and it has subsequently been explicitly recognised in the public consultation strategies employed by the UK Biobank and Generation Scotland (Haddow et al. 2008; Levitt 2005; Winickoff 2006).

Incorporating community input is vital for designing and operating governance frameworks for biobanking that reflect the public interest (Laurie 2011). Sustainable biobanking will necessarily require widespread public support to ensure government funding and investment. Yet mechanisms for engaging informed public debate on biobanking are limited; surveys and focus groups have well-recognised limitations, especially in dealing with complex issues, and there is a strong need to provide workable tools to facilitate this type of community engagement (Gauvin et al. 2010; Rowe and Fewer 2000). This demand has been articulated by Gaskell and Gottweis (2011), who recognised a promising community consultation model built around the concept of deliberative democracy, referring specifically to the employment of this methodology in Tasmania, Australia.

Providing a platform for informed debate in community engagement

Deliberative democracy can perhaps best be thought of as “debate and discussion aimed at producing reasonable, well-informed opinions in which participants are willing to revise preferences in light of discussion, new information, and claims made by fellow participants” (Chambers 2003). It is recognised as a useful tool engendering trust (Gaskell and Gottweis 2011; Niemeyer 2011), and as such has application in areas where citizen opinion is integral to a successful outcome, such as is the case for population biobanks. Deliberative community engagement can take many forms, including citizens' juries, consensus conferences and deliberative polls (Niemeyer 2011). One common feature is the involvement of mini-publics that reflect the diverse voices in society (Fung 2003). Selected categories of individuals reflecting the “community” are generated through recruitment from the general public.

In the context of this paper, the epithet “deliberative democracy” describes a process of community engagement in complex and emerging areas of technology. One feature of these engagements is that participants have little knowledge of the technology and the difficult policy issues that it raises prior to the deliberation. As a consequence “the design of the pubic engagement needs to include carefully constructed elements to inform deliberants about the topic under consideration” (O'Doherty 2013). Deliberative democracy events of this nature have been undertaken in relatively few centres around the world, notably in Canada, the USA, Australia and New Zealand (Burgess, O'Doherty, and Secko 2008; O'Doherty, Hawkins, and Burgess 2012; Rychetnik et al. 2013).

The aim of the recruitment process in this context is to generate a group representing diversity in social, religious and ethnic views, including those whose views are typically missing from the debate, such as people from indigenous or low socio-economic backgrounds. Participants are not chosen at random, nor screened for statistical representation but rather representativeness (Einseidel, Jelsøe, and Breck 2001; Longstaff and Burgess 2010). Willing participants are provided with information presented by relevant experts in the field on a range of important issues, and invited to deliberate on them (Einseidel, Jelsøe, and Breck 2001; Longstaff and Burgess 2010). Deliberative processes are then employed to generate informed public debate, providing normative outcomes to inform policy development and governance structures. Significantly, participants are given the opportunity to raise their own preferences, judgments, expectations, values, hopes and concerns. Through iterative dialogue, participants prioritise the issues according to their own expertise and life experiences, and they produce recommendations that collectively encompass the diversity of perspectives. Importantly, the object is not to press for a consensus, but rather to acknowledge differences of opinion. Recording these areas of persistent disagreement can also be valuable in shaping policy. Whilst the process is similar to other public consultation methods, there are important differences in terms of participant recruitment. The focus of recruitment is to reduce bias and self-selection. Self-selection is a recognised limitation skewing outcomes, for example, vested interest groups may be over-represented.

The Australian state of Tasmania is in many ways an ideal venue to conduct a deliberative democracy event. The Tasmanian population shares features in common with other isolated populations such as that of Iceland. Benefits include a significant depth of genealogical information and a relatively stable, historically isolated population with a strong history of positive engagement among researchers, health care providers and the wider community. There are also logistical benefits for research studies arising from the small geographic distances. Genetic disease research in the Tasmanian population has a long history, including one of the earliest records of Huntington's chorea (Brothers 1964), multiple endocrine neoplasia (Burgess et al. 1996), and, more recently, genetic eye disease (Mackey 2012), multiple sclerosis and cancer (FitzGerald et al. 2009; Rubio et al. 2002). Moreover, a national biobanking strategy is currently on the agenda of the Australian National Health and Medical Research Council.

The Tasmanian deliberative democracy team recruited 25 Tasmanian residents to attend the event. Sessions comprised small and large group discussions. The small group discussions were a forum for canvassing individual participant concerns. The large group discussions, in contrast, focused on debating and attempting to reach consensus on issues of concern chosen by participants. The overall design of the event was consistent with previous events (Burgess, O'Doherty, and Secko 2008; O'Doherty, Hawkins, and Burgess 2012). However, one innovative aspect of this event included our recruitment strategy: It was the first time that recruitment had been conducted in-house, through random selection of telephone numbers and screening for representative diversity. This process provided participants the opportunity to engage with members of the research team at an early stage, which proved to be important in the retention of participants for the period of the consultation. Note takers recorded the names of speakers (participant, expert or facilitator) during all deliberations as well as transcribing the whole-group resolutions. Moreover, the entire consultation was recorded for transcription and further analysis. Participants also completed satisfaction surveys at the conclusion of workshops each weekend of the event.

Advantages and limitations

Deliberative democracy brings to medical research and health care policy a method by which the community can gain standing in the development of policy on a range of issues previously dominated by researchers, lawyers and ethicists. The advantage of this method over previous ones is that it is a two-way, iterative process of information exchange. A recent event held in New Zealand on breast cancer screening policy demonstrated the power of this approach (Rychetnik et al. 2013). In particular, the authors of that study report significant shifts in participants' thinking associated with access to information and dialogue with researchers. This finding contrasts with other methods such as public opinion surveys or focus groups where concerns are frequently raised that the outcomes are influenced by ill-informed or ill-considered views and skewed by vested interests that compromise the representation of alternate points of view (Abelson et al. 2007).

The benefits of iterative dialogue were readily apparent in the Tasmanian deliberative democracy event. Significant shifts in opinions were observed in response to new information, discussion and deliberation. One example was the way participant concerns surrounding the use of their tissues in biobanking research evolved over the course of the event. Initially, participants voiced few concerns and were largely indifferent to the intended uses of their tissue post-donation. For example, one participant initially noted that she was the type of person who does not “give a bugger, take whatever you like, I don't want to know about it, you know, you just do it”. Others generally agreed that once someone makes a donation, they do not require more information. For example, two other participants separately noted:

Oh no, … we don't need to know every little thing they're doing with our samples, do we? We've donated them, ‘here – it's yours'. You know, do your best with it, so, I just don't think we need to know everything they're doing.

I spoke to friends over the week, I explained to them biobanking and they thought, um, it was just like, you know that, you donate blood with the broad consent, and they didn't want to know anything else.

Throughout the event, experts provided more information regarding potential uses of donated samples where participants felt clarification was required. The discussions among participants about the implications of this information led to a significant shift in the opinions outlined above. In particular, participants became concerned about commercialisation, patents and the misuse of samples. One participant commented on the proceeds from commercialisation

Yes, I'm a bit concerned now from the commercialisation aspect … I think that it's really important that any commercialisation process resulting from what's in the bank, that that money comes back to the bank to keep [it] going. Now is that going to happen or not?

Whilst another noted that “[She] mentioned something, that I think you missed, that our group … discussed as well. And that was monopolisation. Someone taking copyright or a patent out on genetic information.”

As the deliberation continued, more information was presented in response to participant questions and participant-led identification of issues and concerns. The discussion that ensued led to a third shift in opinion. Initial intuitive responses were reconciled with subsequent appreciation of the potential risks, as well as balancing the societal benefits of biobanking against the practical limitations, leading to a considered group consensus. Through their discussions, participants revealed that there were issues that they were concerned about, and that whilst they had idealised solutions to these (e.g. return of all individual research results relevant to biobank donors, whether clinically relevant or not), they also recognised practical limitations that might inhibit these (e.g. the cost of doing so). The participants' final position was therefore that they were prepared to place their trust in biobanks, provided their operations were transparent and accountable but, at the same time, that “red tape” should not inhibit research. As one participant commented in response to the suggestion that biobanks should be able to remove a person's sample after their death. “If they can't accommodate it, then they need to tell you.” And more generally, another said, “You need to give some information because otherwise people aren't going to feel that they can trust [the biobank] … .”

The importance of this evolution of opinion is that it demonstrates the limitations of traditional methods of evaluating community attitudes, such as surveys or focus groups, which tend to elicit extempore responses that may not accurately reflect how they would act in a real-world setting. This approach therefore adds substantially to efforts to improve policy and to effectively address public concerns. This is not to suggest that this method of deliberative democracy provides a panacea for the problems of public engagement. Careful thought must be given to the question posed, the representation of experts, the recruitment process and the intended end users of a deliberative democracy event. Employment of this methodology does, however, add a new dimension to this field, which can supplement traditional methods of public consultation.

Another advantage of this deliberative democracy methodology is that it permits effective consultation with cohorts of people who are traditionally difficult to engage, including people who live in remote locations, who are from lower socio-economic strata and who have lower literacy levels. This benefit was underscored in the Tasmanian event where it is an issue of particular relevance, as 42% of Tasmanians live outside of the two major metropolitan areas (ABS 2011) and 49% of Tasmanians are prose illiterate (ABS 2006). These factors can be barriers in the engagement and recruitment of representative populations in health-related research where typically these groups are poorly represented in such studies.

The purposive and stratified sampling techniques employed for the Tasmanian deliberative democracy event facilitated recruitment of people from a range of geographical locations, educational backgrounds and income levels. The level of engagement and retention of participants in the Tasmanian event, and, in particular, their ability to come to resolutions on the complex issues surrounding biobanking, demonstrates that this method is effective at engaging participants across a spectrum of backgrounds. Further, the event itself was conducted in a face-to-face manner, which facilitated the transfer of ideas and information between participants, presenters and the research team. Free-flowing discussions, where participants could phrase their concerns, questions and experiences, helped their own understanding and also that of other participants. In a post-event survey, participants reported feeling that they had gained a good understanding of the issues and that they had confidence in the resolutions they produced. This experience suggests that barriers to recruitment frequently accepted as a standard limitation in consultation can be mitigated within this framework.

Conclusions and future directions

While health researchers in all countries face the same kinds of legal and ethical challenges, substantial disparities have arisen in response. For example, the recently released American College of Medical Genetics and Genomics (ACMG) recommendations on incidental findings in clinical whole exome and whole genome sequencing are heavily focused on the fiduciary duty of clinicians, whilst the equivalent European Society of Human Genetics recommendations are more influenced by considerations of personal autonomy (Green et al. 2013; Van El et al. 2013). It should be noted, however, that recent revisions to the ACMG recommendations have, to some extent, addressed this disparity through the introduction of an opt-out clause.

As the recent Tasmanian event demonstrated, deliberative democracy events enable public engagement of sufficient depth to obtain useful input on complex questions, whilst simultaneously building public trust. It is the engagement of the participating community that is important. In developing biobank protocols, governance bodies will need to engage their communities to find suitable solutions to the increasing demands for resolution of the challenging questions posed in the next era of personalised medicine, such as prioritisation of health care resources, health care database linkage and population screening. The outcomes of such deliberations will have implications for health researchers, law reform and policy-makers, ethicists and social commentators.

The utility of the deliberative democracy methodology has been demonstrated in various settings outside that of biobanking, as reviewed by Secko et al. (2008). Even so, the success of this methodology is more illustrative of the role engagement with the public can have on a deeper level, forming an integral part of the consultation phase for any proposal reliant on wide public engagement, trust and ongoing support. To move genomic medicine forward in the era of spiralling health costs, successful public engagement in decisions on public purse spending is more important than ever. There is increasing evidence from around the world that deliberative democracy-style public engagement is an effective complement to existing consultation strategies, and one that is contributing significantly to what is a challenging era for health care provision.

Acknowledgements

We are greatly indebted to the participants in the Biobank Project Tasmania deliberative democracy event. We would also like to thank the expert presenters and stakeholders who contributed to the event. This work was supported by Australian Research Council (ARC) Discovery Project DP11010069 and National Health and Medical Research Council (NHMRC) Program Grant 490037. JLD was supported by ARC Future Fellowship FT120100623. All authors contributed to the design of the DD event, conception and drafting of the article, and reviewed the final manuscript.

Notes on contributors

Don Chalmers is a distinguished professor of law and director of the Centre for Law and Genetics at the University of Tasmania. Internationally, he serves as deputy chair of the Human Genome Organisation Ethics Committee and the chair of the International Cancer Genome Consortium Data Access Committee. He was chair of the National Health and Medical Research Council (NHMRC) Australian Health Ethics Committee from 1994 to 2000 and has served on a number of other national ethics committees. He currently serves as NHMRC Commissioner of Complaints. His major research interests are in health, law and genetics, research ethics and law reform, in particular biobanking, stem cells, and data sharing and transfer.

Rebekah E. McWhirter is a postdoctoral research fellow in the Cancer, Genetics and Immunology group at the Menzies Research Institute, University of Tasmania. She currently serves as a member of the Human Research Ethics Committee for Tasmania. Her nationally competitively funded research focuses on the genetics of human disease, in particular cancer, with a strong interest in the ethics of human genetic research.

Dianne Nicol is a professor in law, deputy director of the Center for Law and Genetics and current chair of the Academic Senate at the University of Tasmania. Her research focuses on intellectual property, commercialisation, data sharing, health and genetics and she has extensive publications in these fields. She currently is the lead chief investigator on two Australian Research Council-funded projects focusing on governance of personalised medicine and legal issues associated with the transfer of research materials and data.

Tess Whitton is research assistant to Professors Nicol and Chalmers at the Centre for Law and Genetics, and A/Prof. Jo Dickinson in the Cancer, Immunology and Genetics Theme, Menzies Research Institute, University of Tasmania. Her research interests include material transfer agreements, patenting of genetic information, and personalised medicine, and genetics in biobanking.

Margaret Otlowski is the dean of law and deputy director of the Centre for Law and Genetics at the University of Tasmania. She is currently member of the Tasmanian Anti-Discrimination Tribunal. She serves on various state and national boards and committees including as member of two of the National Health and Medical Research Council's principal committees: the Australian Health Ethics Committee and the Human Genetics Advisory Committee. Her research interests include privacy and discrimination particularly in the health law and genetics areas.

Michael M. Burgess is Professor and Chair in Biomedical Ethics at the W. Maurice Young Centre for Applied Ethics and the Department of Medical Genetics at the University of British Columbia. He combines ethical and social theory with social science methods related to policy in health care, health research and biotechnology.

Simon J. Foote has been appointed as Director of the John Curtin School of Medical Research at the Australian National University, Canberra, and is former Dean of the Australian School of Advanced Medicine at Macquarie University, New South Wales. An internationally renowned researcher in human genetics, he currently serves on the National Health and Medical Research Council and his research focuses on the genetics of human disease, in particular infectious disease.

Christine Critchley is associate professor in statistics and Chair, Department of Statistics, Data Science and Epidemiology, School of Health Sciences, Swinburne University and Deputy Director of the research facility housing computer-assisted telephone interviewing (CATI) technology. Her research focuses on public opinion research in the areas of public understanding of science and trust.

Joanne L. Dickinson is an Australian Research Council Future Fellow, and leader of the Cancer, Genetics and Immunology theme at the Menzies Research Institute, University of Tasmania and Deputy Chair of the Human Research Ethics Committee for Tasmania. Her nationally competitive funded research focuses on the genetics of human disease in particular, with a strong interest in the legal and ethical aspects of human genetic research.