Health Care Analysis

, 19:220

Relating to Participants: How Close Do Biobanks and Donors Really Want to Be?

Authors

    • Department of Politics Philosophy and Religion, County SouthLancaster University
Original Article

DOI: 10.1007/s10728-011-0193-9

Cite this article as:
Levitt, M. Health Care Anal (2011) 19: 220. doi:10.1007/s10728-011-0193-9

Abstract

Modern biobanks typically rely on the public to freely donate genetic data, undergo physical measurements and tests, allow access to medical records and give other personal information by questionnaire or interview. Given the demands on participants it is not surprising that there has been extensive public consultation even before biobanks in the UK and elsewhere began to recruit. This paper considers the different ways in which biobanks have attempted to engage and appeal to their publics and the reaction of potential and actual donors. Whilst those organising biobanks presumably want to be as close to their publics as they need to be in order to successfully recruit and sustain participation in sufficient numbers, the closer the relationship the more obligations and expectations there are on both sides.

Keywords

Public engagementUK BiobankALSPACGeneration Scotland

Introduction

Modern biobanks typically rely on the public to freely donate genetic data, undergo physical measurements and tests, allow access to their medical records and provide other personal information.1 For this reason biobanks consisting of voluntary donors have to engage with their publics and the media before recruitment begins. This paper considers different types of relationships that a biobank might have with its publics. Those organising biobanks presumably want to be as close to donors as they need to be in order to recruit them in sufficient numbers and sustain their participation. However, donors’ concerns may not be those anticipated by research ethics committees or other experts. Can there be an optimal model for public engagement and an ideal relationship between donors and those responsible for the operation of large scale biobanks?

There are large genetic databases that do not necessarily have to think about their relationship with participants at all. Where donation is compulsory for some categories of people and the database has public support, then a large database with millions of samples may be built up without public consultation and with favourable media coverage. In England and Wales the National DNA database built up over three million samples before commissioning any public consultation and only established an advisory ethics committee in 2007 [20]. Public and media support was assumed and periodically mobilised by news stories of serious criminals apprehended due to DNA and murderers and rapists who would have ‘got away’ if the DNA from those not found guilty of an offence could not be retained.2

However, it became apparent that the National DNA database was not simply a criminal database but stored profiles for life from around one million people who had never been convicted, and, retained children’s samples on the same basis as adults. The one million innocent people included those arrested but never charged, those later acquitted and victims of crime whose profiles were loaded on the database for the process of elimination with consent. A Member of Parliament, Grant Shapps, began a campaign to remove innocent children from the database and the case of two people whose DNA had been retained despite the lack of a criminal conviction eventually went to the European Court of Human Rights which ruled that the retention of their DNA was disproportionate [8, 24]. If the National DNA database had only retained the DNA from those convicted of serious offences it is doubtful whether there would have been any adverse publicity, nor the enquiry by the Human Genetics Commission entitled ‘Nothing to hide, nothing to fear’ in 2009 [16].

A disease specific database building up samples from patients and their families may not need to consider the relationship with participants either. All those participating will have personal experience of the disease to be researched or awareness of it in their family and therefore be in a position to give informed consent. Furthermore, through their genetic connection participants have a stake in the success of the genetic research.3 However, population genetic databases do need to think about their relationship with donors because they recruit healthy volunteers whose tissue and data will be used for as yet unknown research into as yet unknown conditions. Consent is necessarily broad consent, to any research approved by the research ethics process, rather than informed. Those responsible for establishing population genetic databases have recognised the need to work at getting the relationship with their public right from the planning stage through to post-recruitment.

The relationship before and during recruitment will be considered briefly, before moving on to the problem of successfully maintaining a long-term relationship over decades which is a requirement for the successful operation of a large-scale population database like UK Biobank.

Stage 1: Pre-Recruitment

At the first stage the task is to design and construct a biobank in ways acceptable to all the stakeholders who will not have exactly the same interests. Stakeholders include the prospective funding bodies, the scientific research community and pharmaceutical companies who will be using the data, the medical profession and patient groups who would hope to benefit from the research and the relevant ethical review procedures. Media coverage has to be managed. In the case of UK Biobank the key points aimed at the media and public were that the purpose of the biobank was to advance research into common diseases that everyone had in some way been affected by, for example cancer and heart disease, and press releases stressed that treatments would be available to everyone in the UK through the National Health Service [30].

Stage 2: Recruitment

At the second stage the task is to refine recruitment methods in order to encourage participation and increase the validity of the resulting data. The Ethics and Governance Council of UK Biobank held open public meetings in each location where recruitment was taking place with members of the Ethics and Governance Council and of the senior management [28]. There were also post-visit surveys at each centre and research into the reasons for non-participation.

Stage 3: Post Recruitment

The focus of this article is on the third stage of consultation when the success of the database will be judged. To be successful it is necessary to retain those who have been recruited so that the data can be continually updated and to maximise the use of data in ways that will be acceptable to all stakeholders. Any bad publicity in the media or scandals over alleged misuse of data might lead to withdrawals. Again the needs and wants of participants and researchers may differ.

In 2010 UK Biobank reached its target of 500,000 participants in the age group 40–65 years. Table 1 lists the main official UK Biobank consultations since 2000. In addition there have been many projects on ethical and social aspects of population databases involving public consultations [1114, 17, 26, 32]. Publics have expressed the concerns that are central to traditional medical ethics and to ethical review processes, particularly the need for individuals to be able to give their informed consent, the confidentiality of data, the freedom for individuals to decline to take part, or to withdraw from the research without it affecting medical treatment. However, publics have also been interested in the motivations of, and obligations imposed on, users of the freely donated data. The potential for harm is not just to the donors and families but to society at large, if undesirable or risky research is carried out, or if research is motivated by profit rather than by health benefits to the population [17, 22, 33].
Table 1

UK Biobank: official consultations

Attitudes to the collection and use of biological samples [6]

Primary health care professionals [25]

Trust [23]

Ethics & Governance framework [22]

Industry [27]

Third party access and benefit sharing [33]

Reasons for non-participation (on-going)

Post-visit surveys at each centre on understanding of consent & areas for improvement (on-going)

Both quantitative and qualitative methods have been used in the research including focus groups, surveys, deliberative mapping exercises and a People’s Panel consulted at different stages. Efforts were made to include diverse publics, in terms of ethnicity, socio-economic status and region. Groups that were specifically targeted for individual in-depth interviews included GPs and practice nurses, people with diseases and disabilities, relatives of people with diseases and disabilities, religious and community leaders, spokespeople for organizations with special interest in the issues surrounding human biological samples and genetic research. Scientists and industry were also engaged with as stakeholders. The care taken to consult illustrates the importance of the public to the whole enterprise.

Maintaining the Relationship

The task for biobanks, once established, is to maintain the relationship with participants and regulate access to the resource by researchers in accordance with the purposes for which the biobank was established. There are cost-free approaches to participants that are used by current genetic databases and there are costly approaches. The participation and continuing loyalty of donors can be encouraged by appeals to national pride, to their part in advancing medical research, to their altruism in freely donating to help research and to aiding scientific progress in general. Generation Scotland had a national and family based appeal with phrases such as “…addressing the health and wealth of Scotland”, “the health of the nation”, “our genes, our health, our future” [10]. Referring both to Generation Scotland and Scotland’s part in UK Biobank a press release stated that “Scotland has a long and distinguished history in medical innovation and research. These two studies continue that pioneering spirit and will be seen as landmarks in the health landscape in the future” [9].

In this way there was an appeal to national pride and benefits specific to Scotland. Scotland is said to be “better placed than perhaps any other country” for this type of biobank because Scottish families to tend to stay in the country for several generations, the NHS ensures records are kept throughout life and, especially, because Scotland has a strong tradition of supporting medical research. Given that 800,000 Scottish born people live in England, Wales or Northern Ireland out of a population of around 5 million the stability of the population may be more rhetorical than reflecting reality [4].

The UK Biobank stressed the altruistic nature of donation with attractive images of older people, in the donor age range, with young children and the line “improving the health of future generations” [31]. Since the UK is made up of countries with strong national identities the appeal to the NHS, as common to the whole of the UK, was used rather than any specific reference to benefit to the UK people. The results of the health and medical research would be available to all through the NHS and the biobank would be “unique” and “the world’s largest”.

More costly approaches to maintaining the relationship include personalised on-going feedback of results, genetic counselling, dialogue with participants involving more than the supply of information and the opportunity to ask questions. A third UK project, the Avon Longitudinal Study of Parents and Children (ALSPAC), recruited 14,000 pregnant mothers in the Bristol area in South West England during 1991 and 1992 and developed cell lines from the blood samples in 2003 [1]. ALSPAC has adopted more costly approaches to maintaining the relationship and has been able to develop high levels of loyalty among participants as will be discussed later It has even been credited with a positive effect on recruitment to UK Biobank in the Bristol area. While the percentage response rate has been 10% overall it was highest in Bristol at 19%, double that of Glasgow.

Relationships

It might be thought that the closer a relationship between a biobank and its donors the better it would be for all concerned. However, the closer the relationship the more obligations and expectations exist on both sides, the more emotional investment is made and the more consequences there are if the relationship breaks down. Thinking about the different types of relationships that individuals have, other than the closest one of spouses/ partners or parents and children, people have acquaintances, colleagues, neighbours, perhaps business partners and friends. All these relationships depend on some degree of contact, not necessarily face-to-face, on communication and on some degree of mutual interests, respect, trust and means of conflict resolution. These relationships, from the casual to the more intimate, will be considered in turn as possible models for the relationship between a biobank and its donors.

Acquaintances

The relationship with acquaintances carries minimal obligations or commitments. The relationship might be casual or instrumental. An acquaintance may be the friend of a friend or someone who does work on your house or repairs your car. Such relationships may become closer over time or cease altogether without causing any great difficulties to either side. This sort of loose relationship has advantages for a biobank. If someone donates and consents to on-going use of medical records and samples they may then make no particular demands and the biobank organisers need make no particular effort to maintain the relationship. As long as the person does not leave the country the link with health records can be maintained through the NHS. However, if there is a need to make further demands on donors, say for more tests or questionnaires, then it is easy for the individual to refuse. He or she might have forgotten about the donation altogether and in any case will not necessarily feel any obligations towards the biobank.

Colleagues and Neighbours

Colleagues at work or neighbours tend to meet more frequently due to where we live or work. When changing jobs or moving house a decision has to be made as to whether to keep in touch, assuming these people are not also friends. Colleagues and neighbours may make demands on you and you might feel some obligations towards them, but the relationship will often be of mutual benefit. You water your neighbour’s garden when they are away and can expect some sort of reciprocation. You help colleagues at work to maintain a pleasant environment to work in and perhaps mutual support against ‘the boss’. When these relationships break down there is more cost to the individuals concerned than with mere acquaintances. Disputes between neighbours may turn bitter and involve court cases about boundary walls or excessive noise. Disputes between colleagues may result in grievance procedures or one party moving to another job. Thinking of participants as neighbours or colleagues, based on mutual interests, could work well for biobanks which recruit a homogeneous group of donors. Those who share a common interest in a specific disease, because of their family history, are at least hoping for research that will help their family if only in future generations. Parents from one area, recruited when pregnant, share interests by virtue of having children the same age and a link to the area even if they have relocated. The relationship would be in danger of breaking down if the biobank failed to deliver research results that the participants see as valuable to them and their families or if, in some other way, it is seen as operating against their interests. One example might be a research project that led to the development of treatments not available to all patients because of cost. It would be more difficult to establish a neighbourly or collegial relationship where the biobank is large scale, heterogeneous and recruited nationwide, like UK Biobank.

Partners

Another possibility would be to have a more formal relationship on the business model of a partnership. Again the participants have mutual interests but also some form of written and binding contract, explaining the rights and obligations on both sides and arrangements for dissolving the relationship. It might be said that biobanks already have such arrangements. There will normally be statements about the procedures for allowing access to the biobank for research including ethical approval, privacy and confidentiality clauses and information on how participants can withdraw from the biobank. However, a partnership should allow for negotiation between the parties, with all parties understanding what is involved when they enter into the contract and having some degree of control over the form it takes. The fact that parties choose to be involved and to enter an formal agreement might mean that there is more of a willingness to stick with the partnership even if there are mistakes made on one side. In the case of a biobank this might be adverse publicity over a research project or one minor example of personal information being passed on to a third party. If the mistake is recognised and attempts are made to rectify the situation the partnership may not be damaged. In a focus group study carried out before the arrangements for UK Biobank were in place, participants referred to the need for on-going obligations on both sides rather than a one off consent procedure as illustrated in this quotation from one participant:

…to a certain extent … there should be a liability from the other side … there should be … certain points say that are bulleted, what it’s going to be used for, why, and you sign it. And with that then you can’t sue or anything but if they breach that then you can … you should be kept informed what it’s used for [17, p. 317].

In this model participants would have legal rights if the agreement was broken. Currently the Ethics and Governance Council of UK Biobank is an advisory body that does not have any formal veto over UK Biobank [31]. It does of course have the power to resign en masse, just as participants have the right to withdraw.

A practical suggestion for a shareholder model was made by David Winickoff at an Ethics and Governance Council Workshop [29]. Participants would be represented in the governance structure through a Board of Representatives elected by all participants. Winickoff proposed an advisory role for this Board but also that the Chair would be a voting member on the UK Biobank Board and the Vice-Chair would join the Ethics and Governance Council (ibid, p. 5).

Friends

Finally it might seem attractive to be friends with your participants! The word ‘friend’ is used in many different ways but generally friends try to be in regular contact, whether face to face or otherwise, and have obligations to each other without clear boundaries. It is more difficult and more costly to end a friendship than a partnership because no written contract exists and the relationship may involve strong emotions. Friends will tend to have shared values and interests. In modern society it has been argued that friends are increasingly important, replacing some of the ties to relatives, and that people heap more expectations on to them [21]. At best participants and biobanks could have a relationship of friendship as utility, providing mutual benefit and dissolving once that utility has ended. The aspects of friendship that would be particularly valuable to the success of a biobank would be trust and mutual respect. As Hallowell found in her study of women at risk of ovarian cancer, the way information is given and how it was perceived was important to trust. It has been argued that where there is a dependent relationship, as between patients and doctors, or between donors and the professionals organising a biobank, those with less power are less inclined to view the powerful as trustworthy [15, p. 427].

Using this model of relationships most members of the Bristol based ALSPAC study might be characterised as colleagues. UK Biobank is still developing plans for maintaining its relationship with donors but currently it could be characterised as one of acquaintances.

ALSPAC Members

ALSPAC had advantages when it came to developing a close relationship with participants, who are addressed as members [1]. It was based on a single geographical location although as members have moved they now take part from many different countries . Participating expectant mothers could be assumed to share an interest in child development and the research carried out with their data was relevant to them. For example, research into the effect of smoking on babies and research on the benefits of pregnant women eating oily fish weekly for their baby’s eyesight could be attractively presented. Practical advice has been given to mothers as a result of the database, including advice to put babies to sleep on their backs and the link between washing too often and childhood asthma. Currently the research highlighted on the website is relevant to the now teenage members, ‘Size zero is bad news for bones’ [2]. Research results are presented in separate ALSPAC newsletters for parents and young people [3]. The children were all born within a year and have grown up as ALSPAC members sharing at least their age and a link to the Bristol/Bath area. There is regular contact through clinic visits and questionnaires, with children having their own questionnaires from the age of five. The relationship has developed as the children have grown-up and so are able to take a more active role. In 2006, when the children were around the age of 15 years, a Teenage Advisory Panel was set up. The Executive Director, Ms Molloy, was quoted as saying:

Our plan is that the Panel assists us in making decisions about ALSPAC. For example, we have asked them to give us their ideas on what the clinic environment should look like, to comment on the questionnaires, and to help us develop the new website (McCall [18]).

She hoped that participation would develop further with young people involved in the research process. The Teenage Advisory Panel has resulted in a small group becoming closely involved with ALSPAC. At a workshop organised by UK Biobank Ethics and Governance one participant reported enthusiastically on her role which included being involved in the interviewing process for new staff. Generally there are high rates of participation and a loyalty to ALSPAC, as shown by the willingness of members to tolerate intrusive tests. Williams discusses one test that induced the symptoms of asthma;

This involved testing children for asthmatic and proto-asthmatic symptoms by use of a ‘chemical challenge’ – that is, squirting doses of a chemical, methacholine, into the children’s throats. The extent of bronchial restriction was then measured, and further doses given until the child’s breath function was lowered by 20%. (About 20% of the children were then given another drug, salbutamol, to reverse the effects of the first one) (Williams [34] quoting Henderson p. XX).

To parents this test might appear to have risks yet over 95% of the children who attended agreed to take part, of whom around a third were excluded due to recent respiratory infection or asthma. Williams considers this high participation rate to illustrate the trust felt by parents and children, their feeling of commitment to the project and of an obligation to take part (Williams [34]).

Large scale national databases do not have the same basis on which to build a relationship. They lack a central location and have participants of different ages without common interests, particularly the unique interest of parents in their own child. Given the greater demands ALSPAC makes on parents and children, compared with biobanks that simply ask for a one off donation and the maintenance of a link with health records, the levels of trust and commitment have to be higher. Therefore ALSPAC members who regularly participate might be characterised as colleagues in a mutual enterprise, with a small core of close colleagues or even ‘friends’ who are more involved in activities, including social events, and have greater responsibilities as panel members.

In the case of UK Biobank it is inevitable that there will be issues that cause controversy among some of the 500,000 participants, despite the ethical review processes and safeguards over access and use of data. There are the dangers that research projects passed by the research ethics procedures will receive media attention as unethical or controversial; treatments might be developed that are not approved for use in the NHS because of high costs and limited benefits, access to data might be sought by third parties or data might be mislaid. In the future questions may arise over whether the whole UK Biobank project has provided value for money in terms of its promise to improve ‘the prevention, diagnosis and treatment of a wide range of serious and life-threatening illnesses’.

Maintaining a Relationship in the Future

The preceding discussion has demonstrated that there are costs and benefits to getting closer to participants, donors or members. ALSPAC requires more commitment from its ‘members’ than that envisaged by UK Biobank for its ‘people’.4 ALSPAC has had a close relationship to a small group of members, including those on the Teenage Advisory Panel, and inspired loyalty from a much larger group as demonstrated by the high participation rates. For the larger and more heterogeneous population databases the financial costs of a close relationship might be too high. Of course participants themselves will not necessarily all want the same kind of relationship, but perhaps different degrees of involvement could be offered on recruitment and at intervals thereafter. The costs and benefits of closer contact have to be weighed up carefully. Participants who want to be closely involved will probably not be representative of the group as a whole and, if they are already interested, putting more effort into the relationship may not have any positive effect on retention rates. On the other hand, if various opportunities for a closer involvement exist this may deflect criticisms of a biobank’s operations, whether or not a significant number of participants take them up.

For a large heterogeneous population biobank there is no tried and trusted model of engagement to adopt. One aim of the Public Population Study in Genomics (P3G) is the harmonisation of biobanks across the world. The draft framework sets minimum standards for different aspects of governance intended as a guide for population databases devising their own model [32]. For communication these are that ‘the population will be kept generally informed of the research conducted using their data and samples’ and that ‘participants will be able to register their comments, queries and complaints to the resource, with the assurance that any complaints will be addressed’ (ibid, p. 10). Such a relationship might be characterised as that of an acquaintance, with minimal obligations that can be fulfilled without major expenditure through media publicity for the population and the provision of a means for participants to make contact if they wish to do so. Closer relationships would incur more costs that have to be balanced against the benefits where the aim is to maximise the use of the resource by researchers and ultimately to improve health care for the whole population.

Footnotes
1

While it tends to be assumed that participants will be most concerned about donating genetic material, in an account of one UK Biobank participants’ experiences, it was completing the questionnaire and physical measures that ‘impinge[d] more directly upon her sense of self’ [19].

 
2

The figures given by the then Prime Minister, Gordon Brown, are critically analysed by Dr Helen Wallace in a letter to the House of Lords Constitution Committee—Surveillance: Citizens and the State 28th June 2008. http://www.publications.parliament.uk/pa/ld200809/ldselect/ldconst/18/8013007.htm.

 
3

There are of course conditions for which genetic research is not supported by all those affected. Among the deaf community there has been some opposition to genetic registers and research [5, 7].

 
4

UK Biobank participants are referred to as ‘people’ but attend a ‘participant’ research centre. http://www.ukbiobank.ac.uk/.

 

Copyright information

© Springer Science+Business Media, LLC 2011