Citizens' conceptions of the genome: Related values and practical implications in a citizen forum on the use of genomic information

Abstract Background The development of large data sets, including genomic data, coupled with rapid advances in personalized medicine where citizens increasingly face complex choices about the use of their genomic information implies that citizens are essential stakeholders in genomics. They should be engaged in the ethical, legal and societal issues to produce a framework that fosters trust and allows them to guide the technology based on their values. Objective This article highlights that citizens' conceptions of the human genome inform about and make sense of their main values regarding the use of genomic information, which is critical for policymakers, experts and stakeholders to understand to maintain the public support in genomics. Method Through an inductive thematic approach, we reanalysed data collected for the Belgian citizen forum, which aimed to produce recommendations for the Ministry of Public Health and other stakeholders. Results Citizens expressed four conceptions of the genome that determined which uses of genomic information they supported: the most intimate part of individuals; ‘I am more than my genome’; the individual's property vs the common good; and uncertainty and fear. Conclusion Diversity in their conceptions reveals remaining conflicts of values among citizens, mainly regarding a conception of the genome as an individual property or a common good. However, despite differing conceptions, shared values emerged such as solidarity, privacy, no genetic discrimination and the right to an open future, where individual and common interests coexist. Patient or public contribution The panel of the citizen forum consisted of 32 citizens.


| INTRODUC TI ON
Rapid advances in personalized medicine and genomic technologies raise new questions and issues from conception (eg prenatal and neonatal screening) to adulthood (eg genealogy, carrier screening, precision medicine). 1 Consequently, citizens increasingly have to make complex and personal choices about the use of their genomic information. To support the implementation of these technologies, governments are spending many public funds worldwide as well. 2 Additionally, the successful deployment of genomic data for preventive and medical purposes requires the development of a large representative data set, including long-term health and environmental data of patients and healthy individuals. Hence, the use of genomic information and related technologies calls for the support of the public. Public engagement on the ethical, legal and societal issues (ELSI) in genomics allows citizens to guide the technology based on their values and principles, resulting in a framework that fosters trust in the use of genomic information in society.
Public engagement on ELSI in genomics can take many forms.
It often involves questioning patients, professionals and research participants through surveys and interviews (eg Dheensa et al 3 ; Middleton et al 4 ). Some studies have also described attitudes towards genomics in the general public. 5,6 These studies are very informative, but most of them remain descriptive and do not explicitly translate the results into policy and regulatory recommendations. Recently, several countries have decided to tackle the issue of developing a policy framework for genomics by using deliberative processes with citizens. In France, the public, experts and other stakeholders were consulted as part of the review of the French law on bioethics with a specific interest in genomics. 7 Genomics England reported on their public engagement efforts regarding genomics that citizens call for a new social contract in health care. 8 Within the European Horizon 2020 programme, a stakeholder involved ethics project (SIENNA) was launched with genomics as one of the core subjects. 9 In Belgium, Sciensano and the King Baudouin Foundation orga-

| The citizen forum as a method
A citizen forum is an internationally approved method where a small group of selected citizens, generally 16-32 individuals, debate and reflect in-depth on complex and controversial societal issues. 10,11 It is a way to create practical policy input with involvement from citizens, experts, stakeholders and policymakers. 13

| Participant selection and recruitment
With the help of 10 non-profit civil organizations, targeted media calls and the Bpact online recruitment database, 492 citizens responded to an initial open call to participate in the citizen forum.
Candidates received an online questionnaire with socio-demographic questions, and questions about their motivation to participate and about their knowledge of the topic. The goal was to form the most diverse citizen panel possible to generate high-quality discussions. With this in mind, the King Baudouin Foundation and the independent research agency Indiville selected 160 citizens who answered the online questionnaire, according to criteria such as age, gender, language, level of education, work and family situation (Table 1). People with severe genetic disease and professionals in the field of genomics-doctors, researchers or experts-were excluded.
From these 160 citizens, 32 were selected to form the most diverse panel possible. Participation was voluntary-three participants did not come to the last weekend-and they were paid a small fee to reimburse travel costs. informing the family; carrier screening; prenatal and neonatal genetic testing; secondary findings; managing databanks; directto-consumer testing; and non-medical screening-eg behavioural traits and talents). 14 A team of facilitators, translators and 14 resource persons with different expertise related to the topic (law, genetics, biobank, ethics, patient associations, et cetera) helped the citizens formulate balanced and well-informed opinions by informing them, answering their questions and challenging them through various deliberation methods. Among others, we used the journalism method (in groups of four, each citizen had to interview the rest of the group like a journalist about one case of their choosing in the booklet) and role-plays (citizens simulated a discussion between a patient, a doctor-researcher and a patient organization about data sharing; and an employer asking his employee to access his genomic data, with the intervention of the trade union).

| The Belgian citizen forum
Those methods foster critical attitudes, individual and collective reflection, facilitate a better mutual understanding, pinpoint remaining areas of tension and highlight opportunities for improvements on current practices. Those exercises led to the creation of a mind map, which summarized the main themes, their related issues and values identified by participants ( Figure 1). The second weekend aimed at deepening all themes; by interacting with experts on issues, citizens indicated they needed more information to formulate their opinion. This allowed them to voice their values correctly and to write the final recommendations during the last weekend. Citizens criticized and improved their recommendations before indicating which ones they prioritized and the ones that still included areas of tension ( Figure 2).
An advisory committee supervised the quality of the informative background material and supported the selection of resource persons. Two independent researchers assessed the quality of the process. They published their assessment in a separate report. 15 Throughout the citizen forum, particular attention was paid to transparency and to give participants an active role both on content and methodology. For instance, participants chose the areas of expertise of the resource persons they needed, they reviewed and criticized the content of the deliberations thanks to detailed summary reports we sent them after each weekend, and we took their remarks into account on the final report, which they approved. 16 This article is a qualitative analysis of a public engagement initiative, not a qualitative study per se. Nevertheless, the citizen forum was conducted in a way that followed the Declaration of Helsinki: participation was voluntary, participants could refuse to engage in discussions at any point and consented to the recording of all discussions.

| Stakeholder involvement
Stakeholders were involved in all stages of the process. The citizen forum started with an issue framing workshop, where more than 50 Belgian and international experts and policymakers framed and identified the ELSI that citizens should discuss. 17 An advisory committee composed of experts from various fields continually monitored and mentored the project. During the 3 weekends, several experts were invited to engage in the discussions. After the citizen forum, we organized a stakeholder workshop where experts and stakeholders analysed the implementation of the citizens' recommendations into the Belgian health-care system. 13

| Data analysis
All discussions, both between citizens and experts and among citizens, were audio-recorded and transcribed. As participants were Dutch-and French-speaking, simultaneous translation was provided in all plenary and some subgroup sessions. We ensured that all researchers performing the data analysis were fluent in both languages and that at least one researcher was a native speaker.

Results of the general inductive thematic analysis of citizens'
recommendations have been written up in a report that answers to the research question 'How should we deal with genomic information in society?'. 18 We started with an open-coding process. 19 One researcher coded the transcriptions manually, another checked the codes and entered them in Nvivo, while a third researcher resolved disparities between interpretations. The codes were kept close to the original data, which allowed themes to emerge naturally. These themes were grouped in bigger themes with careful consideration for interconnectedness by two researchers who were constantly debating alternative interpretations. We stayed Work situation

Student 4
Level of education Primary education 2 Technical education 1 Vocational education 1

Secondary education 8
Artistic education 1

Higher education 19
Family situation Alone 7 Cohabitation with spouse/children/parents/ parents-in-law

23
Community housing (eg student housing, nursing home)

2
With children 20

Without children 12
Connection to the health sector Is or was professionally active

| RE SULTS
The inductive thematic analysis of the discussions among participants showed four conceptions of the human genome: 1. The genome is the most intimate part of individuals that warrants protection to avoid a breach of privacy.
2. The genome does not tell the whole story about individuals, or 'I am more than my genome'.
3. The individual's property vs the common good.
4. The genome implies uncertainty and fear. [DNA] says something fundamental about who I am.

| The most intimate part of individuals
It contains more information about me than the result of a blood test, which only reveals specific diseases.
DNA defines to a great extend who we are Citizens felt like they were laying themselves bare when sharing their genomic data and feared that it could turn against them or their relatives, which would make them vulnerable. They referred in particular to weaknesses in health (eg diseases) or personality traits that are negatively considered by society (eg low tolerance to stress).
Citizen 1: There is a high level of distrust in this group.
We distrust the secondary use of data after a person agreed to share their genome or their genomic data. […] Citizen 2: We have the feeling that it will escape us and that there is some kind of danger Therefore, citizens concluded that privacy should be one of the fundamental principles to guide the use of genomic information in society.
However, citizens pointed out that the legal protection of confidential data is worth nothing if individuals are not aware of their right to privacy, or act in a way that undermines it. Since they     Whether an individual should be free to decide to share their genomic data remained a point of discussion. Some citizens suggested to leave the decision up to the individual, but to offer incentives for data sharing, to make it the default option or to raise awareness about the importance of data sharing to nudge citizens into acting in support of the common good. Still, all citizens except one argued that individuals should never be legally obliged to share their genomic data.

| The genome implies uncertainty and fear
Citizens identified different levels of uncertainty in genomics, un- The right to be yourself must be guaranteed; the right not to correspond to the cult of performance and perfection. This is related to our wish for a world with diversity, and to our fear of a world where everyone is conformed due to genomic manipulation and the like.
We want to avoid that kind of world Nevertheless, the majority of citizens supported the individual's right to use genomic technologies (eg prenatal screening, embryo selection, carrier screening) to avoid children with genetic diseases or disabilities, which may lead to the reduction of human diversity in the long run. A minority disapproved of this individual right because it discriminates and categorizes disabled and sick people, and may blame parents who choose to keep an at-risk or a sick child. These citizens warned against the human tendency to 'play God'.
It is the respect for human beings as they are, no matter if they are disabled or not […]. If we begin to select to that point, we take a role that is not ours, because nature already makes the selection. At a given moment, we may need people who carry this or that feature we considered as a disability, or people who don't have the perfect genome, but who resist one specific disease others don't resist. It is how nature has always worked While the present results focus on the negative aspects of genomics according to the citizen panel, they recognized the tremendous potential for good inherent in genomics too. They supported genomic testing to help prevent diseases by detecting predispositions and inherited conditions; they encouraged genomic data sharing to develop better treatments and improve medical knowledge, which contributes to a fair society where everyone has an equal opportunity to a healthy life. In short, genomics brings many hopes and fears simultaneously.
To preserve the support of the public and avoid their fear becoming a reality, the citizen panel demanded a clear legal and ethical framework that inspires trust by using a soft precautionary principle to guide the governance of genomic data.

| D ISCUSS I ON
The four conceptions of the genome from citizens illustrate the ambiguity and complexity of the ways citizens approach genomics. These conceptions lead to various fundamental norms and values concerning the use of genomic information in society. We argue that it is important to understand this framework of different conceptions to be able to interact with citizens about their values and opinions.
Firstly, citizens considered that their genome is part of their intimacy and that the use of their genomic information entails vulnerability for themselves and their relatives. Hence, citizens expected the government to protect their privacy on the one hand by   25 German participants holding an exceptionalist position argued that the genome is unique, is the most personal information of individuals and belongs to them. 29 Furthermore, a recent qualitative study indicated that participants consider genomic data as exceptional not because of an inherent difference, but merely because of its uses outside of health care and its familial implications. 8

| CON CLUS ION
Without public trust and support, genomic medicine will not be able to fulfil its commitment to personalize care and improve pub-

ACK N OWLED G EM ENTS
The authors would like to thank all participants of the citizen forumincluding the citizen panel, coordinators, facilitators, translators and resource persons-the advisory committee who supervised the process, the two independent experts who assessed the quality of the methodology and the King Baudouin Foundation for their financial, technical and communication support.

CO N FLI C T O F I NTE R E S T
All authors have no conflict of interest to declare.

DATA AVA I L A B I L I T Y S TAT E M E N T
The data that support the findings of this study are available from the corresponding author upon reasonable request.