Guthrie Cards in Scotland: Ethical, Legal and Social Issues

CHAPTER 8: PUBLIC ATTITUDES & PUBLIC ENGAGEMENT

8.1 This chapter discusses some of the previous work that has been conducted on public attitudes to the storage and use of newborn bloodspots obtained for screening purposes. Prior to 2009, there had been very little empirical work to examine the views of parents and the wider public on the possible research uses of these samples and associated information. However, as the scientific case for the research potential became more compelling, the associated social, ethical and legal issues remained to be characterised and solutions sought. Empirical research exploring public attitudes is central to that endeavour as this will indicate whether or not there is broad public support, what public concerns are articulated and what conditions need to be met to ensure such support and alleviate concerns. Several studies, using both quantitative and qualitative research techniques, conducted in different countries, have helped answer key questions about what the public think about research uses of stored blood spots from newborn screening. The findings from studies in different jurisdictions, utilising different methods and with different participating population groups, while identifying a range of views, demonstrate overall support for the storage and use of these samples for research purposes, with appropriate safeguards and permissions. The results of several of these studies are distilled below and the key themes identified. The chapter then suggests what research, engagement and consultation should be conducted in Scotland to inform policy in this area.

Key questions addressed by this chapter are:

• What research has been conducted on public attitudes relating to the storage and use of newborn bloodspots?
• What are the key findings from such research?
• What needs to be done in Scotland to assess views and support public engagement?

Key messages from this chapter are:

• There seems to be majority support for the storage and future use of newborn bloodspots for health related research.
• However, a minority do not support such use, with some evidence that this may be related to concerns about privacy, discrimination or lack of trust.
• Overall, permission/consent was identified as essential for such support, although there are different views on when, how and what type of consent should be sought.
• There is a lack of knowledge about retention and future use. Increased understanding was related to increased support in some studies.
• Parents and the wider public wish to have information and choice.
• Public engagement is likely to enhance trust and support for such biorepositories.
• In the Scottish context, there should be a programme of public engagement to explore the public's views and concerns and to identify ways to promote on-going public involvement and the appropriate mechanisms for information sharing.

Existing research on public attitudes

Australia

8.2 Davey et al (2005)¹⁴⁵ conducted a survey of 600 new mothers in Perth, Western Australia, using a self-administered questionnaire. This represented a response rate of 33%. The majority of women had heard of newborn genetic blood screening and over half were satisfied with the information provided. However, some commented on the need for better information given at a more appropriate time.

8.3 Twenty-nine per cent supported the current 2 year retention period and the same proportion a 3-10 year period. However, the authors note that some women said that they did not know enough to make an informed answer; others thought that two years was enough to meet the primary purpose of the test. Those supported a retention period of longer than 2 years seemed to be more keen on promoting research.

8.4 Most (85%) believed that anonymised samples should be made available for research, with 4% disagreeing and 11% being unsure. More specific questions reaffirmed this commitment and also the strong concern about the need for samples to be de-identified. Additional qualitative comments also identified the need for consent. There was also support for different types of health related research.

8.5 Survey research is of course limited by the questions asked and the scope provided for respondents' answers. Some of the question wording was rather positively framed (for example, respondents were asked the extent to which they agreed or disagreed with the statement: 'I would like to have the opportunity to contribute positively to research through newborn screening cards').

8.6 Muchamore et al (2006)¹⁴⁶ conducted qualitative research using moderated small group discussions in New South Wales. Nine discussions were conducted with a total of 40 participants (24 women and 16 men) including young adults, parents with young children and parents with older children. Each group met twice to allow a deliberative component.

8.7 There was overall support for newborn screening although little specific knowledge. Discussion of the retention and use of samples revealed little understanding that this occurred nor of why it might be useful. The vast majority felt that parents should know that the dried blood spots were stored and could be used for a range of purposes.

8.8 Use for medical research was seen positively, but in the context of parents being informed and consulted if identifying information was provided. Parental consent was only seen as important by a few in the case of anonymised samples. There seemed to be a misconception that stored specimens might have direct future benefit to families.

8.9 Views were expressed about unacceptable uses (e.g. research into human cloning; research that might lead to discrimination by insurers or employers) and about inappropriate access - some felt concerned about access by pharmaceutical and biotechnology companies because of the profit principle whilst others reflected on the need for such research (such findings resonate with Scottish research on public attitudes to Generation Scotland, Haddow et al 2007; 2008¹⁴⁷). Overall, the authors note a high level of trust in those representing the public good and therefore a willingness for them to make decisions about appropriate research.

8.10 Most saw value in stored samples being used to identify missing children and most were supportive of police access for solving crime. However, amongst the groups of men, concern was expressed as there seemed to be general distrust of the police. Concerns were also raised about security and privacy.

New Zealand

8.11 A comprehensive consultation has been undertaken in New Zealand, combining a public consultation with focus group research (Research New Zealand 2007 a and b)¹⁴⁸. The consultation included closed and open ended answers to questions on information; consent; refusals; repeat sampling/testing; and storage and use. 182 responses were received with 80% of these being from private individuals (68% of whom were women).

8.12 Focussing on the issue of storage, access and use, the consultation found that 55% agreed with current New Zealand Police access to blood spot cards (to help identify a missing or deceased person or for other coronial inquiries within a Memorandum of Understanding between the Newborn Metabolic Screening Programme and the New Zealand Police). A further 21% agreed but with some suggested changes and 19% disagreed with 5% either did not know or had no comment on this issue. Although this suggests majority support, nearly a fifth of respondents did not agree with the current policy.

8.13 Forty-six per cent of respondents said that the blood spot cards should be stored indefinitely, 16% agreed with 21 years and 15% with 10 years. Nineteen per cent suggested other time periods, ranging from three months to the average lifespan.

8.14 Respondents were asked if they agreed or disagreed with the proposed process of approving research requests (formal approval by Ethics Committee and National Screening Unit). Sixty-six percent agreed with the proposed approval process; 22% disagreed and 12% either did not know or had no comment. Areas of concern amongst those disagreeing who provided comments were that the blood spots had not been originally taken for the purposes of research or that they (respondents) did not have enough knowledge of the organisations concerned.

8.15 The focus group research involved seven groups of 37 participants of different ethnicities, including new parents and adults without children. The research aimed to understand people's perceptions and expectations about future storage and use of blood spot cards. The research found support for screening but that attitudes and beliefs varied on some of the other issues. None could recall, where relevant, receiving information about storage and future use. There were a range of views about consent, although consensus about the need for consent. Some felt that there should be consent each time their child's blood spot was being used, others felt that final consent at the time of screening was sufficient, so long as this included specific consent for storage and each possible future use. The need for more information, given at an appropriate time and in an appropriate way, was also noted.

8.16 Participants did not know that the blood spots were stored; this was an emotional issue for some due to the specific cultural meanings of blood amongst the Maori and Pacific peoples. The authors note that only some were comfortable with the idea of indefinite storage and the New Zealand Europeans expressed an attachment to their blood spots.

8.17 Responses were also varied in relation to use of blood spots for research, with some happy about this and the proposed approval process and others concerned about lack of explicit consent or about final consent. Other types of use, for example by police or for audit seemed to generate less concern.

Canada

8.18 Avard et al (2006)¹⁴⁹ report on a consultative workshop held as part of a review of the social, ethical and legal issues regarding the storage, access and uses of newborn bloodspots. The workshop included a range of participants: policy makers, consumer groups, representatives from provincial NBS laboratories, health professional associations and the research community.

8.19 In relation to uses of dried bloodspots, respondents were supportive of research but less supportive for other purposes, such as special family studies or forensic purposes. It was noted:

'It was agreed that there may be appropriate and inappropriate secondary uses of newborn dried bloodspots although there was diversity on where to draw the line and how to proceed' (p88)

8.20 Important factors raised were that secondary use should not interfere with the primary purposes of NBS; that identifiers must be removed and of course ethical approval gained. However, concern was noted about whether research ethics boards were able to determine appropriate uses. Most felt that future use for forensic purposes was a violation of individual liberties.

8.21 The workshop found no consensus over whether informed consent should be obtained before secondary use for research purposes took place. However, many seemed to think that if samples were anonymised and the research subject to ethical review, then explicit consent should not be required. Further research might help illuminate whether explicit consent should be required or assumed, but that the issue would remain complex. For example, would the primary purpose be negatively affected if consent for future use was required but then would individual autonomy and privacy rights be compromised if explicit consent was not obtained? Participants also noted that parents need to be informed about storage and future use as well as about the screening programme more generally.

8.22 Bombard et al (2012)¹⁵⁰ explored citizens' values about research with stored samples from newborn screening. Focus groups with an educational component, deliberative discussion and a pre and post interview questionnaire were employed. The study focussed on anonymous medical research and linkage to other information, as this reflects the approach in Canada as well as elsewhere.

8.23 The study found overall support for future anonymous research, with 90% (36 participants) agreeing to storing samples for such use and further support identified in the focus groups. Most suggested the need for choice in relation to whether their child's sample should be stored, although some thought that parents' should be required or at least strongly encouraged to allow such storage and use.

8.24 The researchers identified three key themes that underlay these differences: level of trust, concern about harms and where the balance between individual and population interests lay.

8.25 There was strong agreement that parents need to be informed about the secondary use of newborn bloodspots.

8.26 Given the differences between participants in relation to parental choice, the authors conclude that:

Ultimately, although public engagement exercises may elucidate values, enhance transparency, and inform policy, they may not necessarily provide explicit policy direction (p245)

USA

8.27 There have been several studies in the USA over the past few years exploring public attitudes. These have included different States and diverse methods and populations. The first are a group of publications by a research team including Botkin and Rothwell from the University of Utah.

8.28 Botkin et al (2012)¹⁵¹ conducted a multi-method study utilising a survey (paper/telephone), focus groups and an internet survey through Knowledge Network panels. A total of 3855 participants were involved in these studies. An educational movie or written information was provided.

8.29 Although the study found strong support for NBS, 30% of survey respondents indicated that they were very concerned about retention. Those with lower concern were more likely to have seen the movie rather than the written information, to be white, a mother of young children, with higher educational attainment and to have been surveyed by telephone.

8.30 However, there was majority support (80%) for potential uses of residual bloodspots whether this was for quality control, research on diseases affecting mothers and children or research on diseases of the general population.

8.31 The survey asked whether an opt-in or opt-out permission model was preferred. 62% supported an opt-in model. The factors that were associated with selecting an opt out model were movie education, older age and higher level of education.

8.32 This study reinforced the importance of providing information; this led to increased support as well as improving knowledge. The authors note that their focus group discussions demonstrated that participants were supportive of retention but wanted more information and that choice was important.

8.33 Rothwell et al (2012)¹⁵² report on the focus group element of the above study. Fourteen groups were conducted across six states, with a total of 128 participants. The groups included ones with the general population, African-American groups, Hispanic groups and mothers with young children.

8.34 The results suggest support for the retention and use of residual blood spots for research. There was a preference for an opt-in approach although some recognition of the logistical issues with this.

8.35 There was general support for research to improve health or provide medical benefit if conducted by university or medical departments rather than pharmaceutical companies.

8.36 Personal control was considered important with participants expressing their right to choose, their need to be informed and to be able to change their minds.

8.37 Rothwell's earlier study (2010)¹⁵³ also employed focus group methods, with three groups, an African American group, a Paediatrician group and a Mothers of young children group. They noted that that guidance had tended not to be informed by public input and suggest this is a failure than could become problematic.

8.38 This research identified the need for informed consent on both anonymous and identifiable residual samples, appropriate ethical review, consent for storage, fears of discrimination arising from research and lack of awareness of the programme.

8.39 Duquette and colleagues at Michigan Department of Community Health and University of Michigan report on two studies. The first comprised a set of four questions on the 2008 Behavioural Risk Factor Surveillance System (telephone survey)¹⁵⁴. These asked about support for the use of dried blood spots for research in general, for specific diseases (general health research, childhood, adult and environmental exposure). The sample was 3018 adults.

8.40 The majority (72.3%) supported the use of dried blood spots for research; those who were female, younger, white, healthy and with at least a high school degree were more likely to say that they were in favour. Only three per cent strongly opposed.

8.39 Support was high for each type of use with little variation across use; indeed it was higher than for the overall question about use. This support was associated with age, education and household income.

8.41 The second study reported by Duquette et al (2010) involved ten focus groups from diverse communities. The discussions explored public views about the Michigan BioTrust for Health, a program for storage and use of residual newborn screening dried blood spots:

'Community engagement was considered critical for assuring that samples are used in a manner acceptable to the public and to the ultimate success of the MDCH BioTrust initiative' (p147)

8.42 Four key questions were used focussing on initial thoughts about the BioTrust, types of research that would be acceptable, how consent should be obtained and how you would feel about your own or your child's de-identified blood spots being used for research. Each participant was provided with written information, there was a presentation and a pre and post survey.

8.43 There was very strong support for the idea of a BioTrust, with none saying they were opposed and a consensus being reached. The survey produced consistent results, with 86.4% strongly or somewhat agreeing. There was also broad support for any type of health research, although participants identifying cloning or cosmetic research as exceptions. The post discussion survey identified increased support. In the survey, support for use by academic institutions was higher than for private sectors, but in the focus groups there was a recognition that distinguishing these two was sometimes difficult and the type of research seemed to be more important than who was conducting it.

8.44 There was a consensus that information was required for informed decision making but different views as to how this might be provided. There was also lack of consensus in the focus groups about whether consent should be opt-in or opt-out. This was reflected in the survey responses also.

8.45 Lastly, there was general agreement about using one's own or one's child's bloodspots in de-identified research. In the questionnaires, willingness increased post discussion. Privacy concerns were expressed by a minority and opinion was divided about the benefit of receiving research results.

8.46 Tarini et al's research¹⁵⁵ comprised an internet survey of a nationally representative sample of parents, with a sample size of 1508. This examined willingness to permit use of their children's newborn samples and to allow storage.

8.47 Overall, 76.2% responded that they were willing or somewhat willing to permit use, with permissions. This dropped to 28.2% if permission was not obtained Seventy-eight per cent would permit storage; those less willing to permit use were also less willing to permit storage.

8.48 They conclude that it is important to:

'engage the public in a transparent and informative discussion that also provides parents a forum in which to voice their concerns.' (p129)

8.49 Neidich et al (2010)¹⁵⁶ conducted a survey of women in University of Chicago hospitals who had live infants. 239 women participated, with 82% self-classifying as black. The researchers wanted to explore attitudes to a hypothetical paediatric biobank as a response to ethical concerns about a proposed programme, particularly what additional safeguards might be required and what information beyond the usual consent requirements.

8.50 The results suggested some support for such a biobank, with 48% of women saying they'd be willing to enrol their child and 28% being not sure. Caucasians were the most willing.

8.51 The research also explored why people responded as they did and 58% expressed altruism for societal benefit. Exploring what that benefit might be, the survey contained questions about trust and justice. Understanding of research, trust in researchers and a belief that findings would be used fairly all correlated with hypothetical enrolment. The authors conclude that:

'It is imperative that researchers help ensure that the women's trust and justice beliefs are fulfilled' (p303)

China

8.52 Gong and colleagues conducted the first survey of Chinese parents regarding the use of newborn screening samples. This was a hospital-based survey to explore attitudes to newborn screening sample storage and permitting use for research (with or without consent). With a 52% response rate, the sample size was 378. The research was conducted in Beijing.

8.53 Sixty-eight per cent of parents would permit infants' samples to be stored but this dropped to 14% if no permission was obtained. This did not seem to be explained by a range of socio-demographic variables, such as gender, education or household income. Those who would not approve of use in those circumstances also reported that they should be given more information:

'They strongly believed that they - and not merely the researchers - should play a more important role in such research' (p191)

8.54 Although these authors note similarities between their findings and other research such as that summarised above, they also suggest that there is a basic lack of trust between patients and doctors in China and therefore concern about rights and privacy.

Japan

8.55 Fujii et al (2010)¹⁵⁷ conducted a questionnaire study to examine the attitudes of the public, parents, patient families and health professionals to extended uses of newborn screening and storage of blood spots. They found that awareness of newborn screening was low amongst the general public and they were less likely to be positive about the extended use for the study of health problems. Patient families and health professionals were more positive.

8.56 The open ended comments were analysed through text mining and three concepts were identified: personal data (for example, privacy); consent availability; and progress in medical science. As with other studies, they noted the importance of information.

England

8.57 Research by Hargreaves et al (2005)¹⁵⁸ from the Parent Support Research Team of the UK Newborn Screening Programme Centre touched on this issue. Hargreaves et al conducted a qualitative study involving telephone interviews and focus groups with 47 parents and 35 health professionals. They found that amongst some parents the need for written consent was heightened when the storage of bloodspots was considered.

Conclusions on public engagement

8.60 Research across different jurisdictions, using different methods identify overall support for the storage and research use of newborn blood spots. However, some are less supportive and support is contingent on a number of factors. Consent, control, trust and information are all identified as key to maintaining and promoting public support.

8.60 In policy terms, the research is clear about the need for public engagement, not as a one-off consultation to ascertain views but as an integrated component of the on-doing relationship between citizens and those who procure, curate and use newborn blood spot collections. This suggests an approach to engagement that embraces awareness raising, consultation and empowerment.

8.61 Research and public engagement should be conducted in Scotland to explore views and attitudes as well as to shape best practice in public involvement in governance.

POINTS TO CONSIDER AND RECOMMENDATIONS FROM CHAPTER 8

• Although previous research on public and professional attitudes does not suggest widespread distrust or concern about the storage and use of newborn bloodspots, the issues of consent, access, appropriate use and regulation are pertinent and there is a degree of ambivalence about appropriate policies. This needs to be resolved transparently and with public input.
• We recommend primary research on public and professional attitudes and concerns about the storage and further use of newborn bloodspots and a programme of public and stakeholder engagement for effective governance.