Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine 861 Trust in Biobank Research Meaning and Moral Significance LINUS JOHNSSON ACTA UNIVERSITATIS UPSALIENSIS ISSN 1651-6206 ISBN 978-91-554-8585-6 UPPSALA urn:nbn:se:uu:diva-192295 2013 Dissertation presented at Uppsala University to be publicly examined in Auditorium Minus, Museum Gustavianum, Akademigatan 3, Uppsala, Saturday, March 9, 2013 at 09:15 for the degree of Doctor of Philosophy (Faculty of Medicine). The examination will be conducted in English. Abstract Johnsson, L. 2013. Trust in Biobank Research: Meaning and Moral Significance. Acta Universitatis Upsaliensis. Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine 861. 142 pp. Uppsala. ISBN 978-91-554-8585-6. What role should trust have in biobank research? Is it a scarce resource to be cultivated, or does its moral significance lie elsewhere? How does it relate to the researcher’s individual responsibility? In this thesis I draw four general conclusions. First, trust is still very much present in at least some biobanking settings, notably in Sweden, but possibly also internationally. Second, a morally relevant conception of trust entails that to be trustworthy, researchers must consider the normative expectations that people have of them, and renegotiate expectations that are mistaken. Third, this conception differs from “public trust” assessed through surveys. The main use of the latter is to legitimate policy, not to identify moral duties. Fourth, in spite of ethics review, guidelines and informed consent procedures, ethical issues will always arise during the course of a research project. Researchers can therefore never avoid their individual moral responsibility. Ensuring that one is adequately trusted is one step towards conducting morally acceptable research. Study I indicates that few Swedes refuse storage of samples in healthcare-associated biobanks and their use in research. Study II suggests that people are somewhat more willing to donate samples than surveys indicate, especially when approached face-to-face by health care personnel. Relationships of trust might thus be important in people’s decision-making. Study III investigates trust as a moral concept. The trustee is often in a unique position to determine what the other’s trust amounts to. When it is mistaken, the trustee has an obligation to counteract it, compensate for it, or renegotiate the expectations that cannot be met. In Study IV, I critique the feasibility of guaranteeing the trustworthiness of the research apparatus through formal measures such as ethics review and guidelines. Not only are there limitations of such measures to consider. They also risk blinding researchers to ethical issues that are not covered by the rules, fostering moral complacency, and alienating researchers to ethics. Keywords: Biobank, biobank research, bioethics, biobank ethics, research ethics, trust, trustworthiness, moral responsibility, informed consent, ethics review, ethics guidelines Linus Johnsson, Uppsala University, Centre for Research Ethics and Bioethics, Box 564, SE-751 22 Uppsala, Sweden. © Linus Johnsson 2013 ISSN 1651-6206 ISBN 978-91-554-8585-6 urn:nbn:se:uu:diva-192295 (http://urn.kb.se/resolve?urn=urn:nbn:se:uu:diva-192295) To Hannalina and Hilda I hope that you will find both trust and trustworthiness in your lives. List of Papers This thesis is based on the following papers, which are referred to in the text by their Roman numerals. I JOHNSSON, L., HANSSON, M. G., ERIKSSON, S. & HELGESSON, G. (2008) Patients’ refusal to consent to storage and use of samples in Swedish biobanks: cross sectional study. BMJ 337:a345. II JOHNSSON, L., HELGESSON, G., RAFNAR, T., HALLDORSDOTTIR, I., CHIA, K. S., ERIKSSON, S. & HANSSON, M. G. (2010) Hypothetical and factual willingness to participate in biobank research. Eur J Hum Genet, vol. 18, no. 11, pp. 1261-4. III JOHNSSON, L., HELGESSON, G., HANSSON, M. G. & ERIKSSON, S. (2012) Adequate Trust Avails, Mistaken Trust Matters: On the Moral Responsibility of Doctors as Proxies for Patients’ Trust in Biobank Research. Bioethics. doi: 10.1111/j.1467- 8519.2012.01977.x. IV JOHNSSON, L., ERIKSSON, S., HELGESSON, G. & HANSSON, M. G. Making researchers moral: Why institutionalised distrust might not work. Manuscript. Reprints were made with permission from the respective publishers. Contents Introduction ..................................................................................................... 9 Elephants and agendas—Personal reflections .......................................... 12 Biobanks in modern medicine ...................................................................... 14 Current challenges and promises .............................................................. 15 Ethical issues and concerns ...................................................................... 17 Clinical biobanks in Sweden .................................................................... 18 The shaping of biobank ethics ...................................................................... 22 Critical events in invasive research .......................................................... 23 Research ethics as a regulatory framework .............................................. 27 Critical events in biobank research .......................................................... 34 The role of informed consent in biobank research ................................... 40 The quest for public support ......................................................................... 47 Public trust in health care ......................................................................... 48 Public trust in biobank research ............................................................... 51 Aims .............................................................................................................. 63 Methods ........................................................................................................ 64 Empirical methods .................................................................................... 65 Philosophical methods .............................................................................. 67 Summary of Findings .................................................................................... 73 Study I: Patients’ refusal to consent to storage and use of samples in Swedish biobanks ..................................................................................... 73 Study II: Hypothetical and factual willingness to participate in biobank research .................................................................................................... 73 Study III: Adequate trust avails, mistaken trust matters: On the moral responsibility of doctors as proxies for patients’ trust in biobank research .................................................................................................................. 74 Study IV: Making researchers moral: Why institutionalised distrust might not work ................................................................................................... 75 Discussion ..................................................................................................... 77 A naïve interpretation ............................................................................... 77 The concept of trust .................................................................................. 80 Public trust revisited ............................................................................... 103 Proper use of trust .................................................................................. 108 Future Work ................................................................................................ 122 Conclusions ................................................................................................. 124 Acknowledgements ..................................................................................... 127 Summary in Swedish – Sammanfattning .................................................... 128 References ................................................................................................... 133 Introduction If this book were a novel, I would not fret about the opening line. “It all began with a newspaper scoop.” Whatever doubts one might have about the factual accuracy of the series of articles in Aftonbladet—one of Sweden’s largest tabloids—that hauled the biobank phenomenon from the relative safety of laboratory storage rooms into the public eye in 1999, at least it had an impact. Not that many people outside of the biobanking community have necessarily become much acquainted with them (with the notable exception of health care personnel, who tend to associate the term with unwelcome paperwork). But the attention that the scoop stirred up did contribute—at least if the tabloid’s own reports on the matter are to be believed—to the enactment of a new law. In its aftermath, with all the legal and ethical inconsistencies and other issues that now need to be discussed and resolved, many bioethicists have secured their employment for many years to come. The newspaper scoop in question lacked every hint of subtlety. “Scientists perform secret experiments on parts of your body,” page one proclaimed in customary huge, bold letters (Trägårdh and Ringman, 1999). Reality, as usual, did not quite live up to the drama. The “body parts” in question were leftover samples from health care,