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Abstract

‘Digital Mega-Studies’ are entirely or extensively digitised, longitudinal, population-scale initiatives, collecting, storing, and making available individual-level research data of different types and from multiple sources, shaped by technological developments and unforeseeable risks over time. The Australian ‘Gen V’ project exemplifies this new research paradigm. In 2019, we undertook a multidisciplinary, multi-stakeholder process to map Digital Mega-Studies’ key characteristics, legal and governance challenges and likely solutions. We conducted large and small group processes within a one-day symposium and directed online synthesis and group prioritisation over subsequent weeks. We present our methods (including elicitation, affinity mapping and prioritisation processes) and findings, proposing six priority governance principles across three areas—data, participation, trust—to support future high-quality, large-scale digital research in health.

Keywords

Digital Mega-Studies health research data governance law trust participation

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References

Abraham

Schneider

vom Brocke

(2019). Data governance: A conceptual framework, structured review, and research agenda. International Journal of Information Management, 49, 424-438. https://doi.org/10.1016/j.ijinfomgt.2019.07.008

American Society for Quality (ASQ) (2020). What is an Affinity Diagram? K-J Method . https://asq.org/quality-resources/affinity

Bell

J. L.

(2020). Governing commercial access to health data for public benefit: Charity law solutions. Medical Law Review, 28(2), 247-269. https://doi.org/10.1093/medlaw/fwz022

Bentley

A. R.

Callier

Rotimi

C. N.

(2017). Diversity and inclusion in genomic research: Why the uneven progress? Journal of Community Genetics, 8(4), 255-266. https://doi.org/10.1007/s12687-017-0316-6

Building digital capability (2018, October 31). Jisc. https://www.jisc.ac.uk/rd/projects/building-digital-capability

Bunnik

E. M.

Janssens

A. C. J. W.

Schermer

M. H. N.

(2013). A tiered-layered-staged model for informed consent in personal genome testing. European Journal of Human Genetics, 21(6), 596-601. https://doi.org/10.1038/ejhg.2012.237

Central Digital and Data Office, UK Government (2020). Data ethics framework . https://www.gov.uk/government/publications/data-ethics-framework

Danish National Birth Cohort (2019, July 31). About the DNBC . https://www.dnbc.dk/about-the-dnbc

Dezateux

Colson

Brocklehurst

Elias

(2016). ‘Life After Life Study’: Report of a scientific meeting held at The Royal College of Physicians. University College London. https://discovery.ucl.ac.uk/id/eprint/1485681/7/Life%20Study%20Report%20of%20Life%20After%20Life%20Study%20meeting%20January%202016%201485681.pdf

10.

Edwards

(2012). Growing up in Australia: The longitudinal study of Australian children. Family Matters, 91, 7-17.

11.

Erikainen

Friesen

Rand

Jongsma

Dunn

Sorbie

McCoy

Bell

Burgess

Chen

Chico

Cunningham-Burley

Darbyshire

Dawson

Evans

Fahy

Finlay

Frith

Goldenberg

, … (2021). Public involvement in the governance of population-level biomedical research: Unresolved questions and future directions. Journal of Medical Ethics, 47(7), 522-525. https://doi.org/10.1136/medethics-2020-106530

12.

Freeman

R. E.

(2010). Strategic management: A stakeholder approach. Cambridge University Press.

13.

Gajanayake

Lane

Iannella

Sahama

(2012). Legal issues related to accountable-Ehealth systems in Australia. In 1st Australian eHealth Informatics and Security Conference, held on the 3rd-5th December, 2012 at Novotel Langley Hotel, Perth, Western Australia, Perth, Western Australia. https://doi.org/10.4225/75/5796f5b740a96

14.

Generation Victoria (GenV) | Murdoch Children's Research Institute (n.d.). Retrieved 5 June 2020 from https://genv.org.au/

15.

Gold

Davies

Davenport

Perera

Wake

Goldfeld

(2019). GenV rapid evidence assessment report: Large research-led partnerships. Murdoch Childrens Research Institute. https://mcri.figshare.com/articles/GenV_Rapid_Evidence_Assessment_Report_Large_research-led_partnerships/10452185

16.

Haas

M. A.

Teare

Prictor

Ceregra

Vidgen

M. E.

Bunker

Kaye

Boughtwood

(2021). ‘CTRL’: An online, dynamic consent and participant engagement platform working towards solving the complexities of consent in genomic research. European Journal of Human Genetics: EJHG. 29, 687-698. https://doi.org/10.1038/s41431-020-00782-w

17.

Hagemann

Colvin

Gibson

L. Y.

Miller

S. J.

Palmer

D. J.

Srinivas Jois

Silva

D. T.

Prescott

S. L.

(2019). The ORIGINS project. In Sata

Fukuoka

Hanson

(Eds.), Pre-emptive medicine: Public health aspects of developmental origins of health and disease (pp. 99-116). Springer. https://doi.org/10.1007/978-981-13-2194-8_6

18.

Hein

I. M.

De Vries

M. C.

Troost

P. W.

Meynen

Van Goudoever

J. B.

Lindauer

R. J. L.

(2015). Informed consent instead of assent is appropriate in children from the age of twelve: Policy implications of new findings on children's Competence to consent to clinical research. BMC Medical Ethics, 16(1), 76. https://doi.org/10.1186/s12910-015-0067-z

19.

Hunter

K. G.

Laurie

G. T.

(2009). Involving publics in biobank governance: Moving beyond existing approaches. In C. Mullen & H. Widdows (Eds.), The governance of genetic information: Who decides? (pp. 151-177). Cambridge University Press. https://doi.org/10.1017/CBO9780511635274.009

20.

Kaiser

(2014, December 12). NIH cancels massive U.S. children's study. Science. https://www.sciencemag.org/news/2014/12/nih-cancels-massive-us-children-s-study

21.

Kaye

Briceño Moraia

Mitchell

Bell

Bovenberg

J. A.

Tassé

A.-M.

Knoppers

B. M.

(2016). Access governance for biobanks: The case of the BioSHaRE-EU cohorts. Biopreservation and Biobanking, 14(3), 201-206. https://doi.org/10.1089/bio.2015.0124

22.

Kaye

Prictor

(2021). Consent. In Ganguli-Mitra

Sorbie

McMillan

Dove

Postan

Laurie

Sethi

(Eds.), The Cambridge handbook of health research regulation (pp. 103-111). Cambridge University Press. https://doi.org/10.1017/9781108620024.014

23.

Kirkham

J. J.

Davis

Altman

D. G.

Blazeby

J. M.

Clarke

Tunis

Williamson

P. R.

(2017). Core outcome set-standards for development: The COS-STAD recommendations. PLoS Medicine, 14(11), e1002447. https://doi.org/10.1371/journal.pmed.1002447

24.

Koenig

B. A.

(2014). Have we asked too much of consent? The Hastings Center Report, 44(4), 33-34. https://doi.org/10.1002/hast.329

25.

Kooijman

M. N.

Kruithof

C. J.

van Duijn

C. M.

Duijts

Franco

O. H.

van IJzendoorn

M. H.

de Jongste

J. C.

Klaver

C. C. W.

van der Lugt

Mackenbach

J. P.

Moll

H. A.

Peeters

R. P.

Raat

Rings

E. H. H. M.

Rivadeneira

van der Schroeff

M. P.

Steegers

E. A. P.

Tiemeier

Uitterlinden

A. G.

, … (2016). The generation R study: Design and cohort update 2017. European Journal of Epidemiology, 31(12), 1243-1264. https://doi.org/10.1007/s10654-016-0224-9

26.

Landry

L. G.

Ali

Williams

D. R.

Rehm

H. L.

Bonham

V. L.

(2018). Lack of diversity in genomic databases is a barrier to translating precision medicine research into practice. Health Affairs, 37(5), 780-785. https://doi.org/10.1377/hlthaff.2017.1595

27.

Lawlor

D. A.

Andersen

A.-M. N.

Batty

G. D.

(2009). Birth cohort studies: Past, present and future. International Journal of Epidemiology, 38(4), 897-902. https://doi.org/10.1093/ije/dyp240

28.

Lepley

C. J.

(1998). Problem-solving tools for analyzing system problems: The affinity map and the relationship diagram. JONA: The Journal of Nursing Administration, 28(12), 44-50. https://doi.org/10.1097/00005110-199812000-00014

29.

Magnus

Haug

Nystad

Skjaerven

(2006). The mother and child cohort study will give new answers. Tidsskrift for Den Norske Laegeforening: Tidsskrift for Praktisk Medicin, Ny Raekke, 126(13), 1747-1749.

30.

Milne

Morley

K. I.

Almarri

M. A.

Anwer

Atutornu

Baranova

E. E.

Bevan

Cerezo

Cong

Costa

Critchley

Fernow

Goodhand

Hasan

Hibino

Houeland

Howard

H. C.

Hussain

S. Z.

Malmgren

C. I.

, … (2021). Demonstrating trustworthiness when collecting and sharing genomic data: Public views across 22 countries. Genome Medicine, 13(1), 92. https://doi.org/10.1186/s13073-021-00903-0

31.

Murtagh

M. J.

Turner

Minion

J. T.

Fay

Burton

P. R.

(2016). International data sharing in practice: New technologies meet old governance. Biopreservation and Biobanking, 14(3), 231-240. https://doi.org/10.1089/bio.2016.0002

32.

Narayanan

Huey

Felten

E. W.

(2016). A precautionary approach to big data privacy. In Gutwirth

Leenes

De Hert

(Eds.), Data protection on the move (Vol. 24, pp. 357-385). Springer. https://doi.org/10.1007/978-94-017-7376-8_13

33.

National Data Guardian, & Caldicott

(2020, May 27). No surprises. GOV.UK. https://www.gov.uk/government/speeches/no-surprises

34.

Nilsen

E. S.

Myrhaug

H. T.

Johansen

Oliver

Oxman

A. D.

(2006). Methods of consumer involvement in developing healthcare policy and research, clinical practice guidelines and patient information material. Cochrane Database of Systematic Reviews, 3, https://doi.org/10.1002/14651858.CD004563.pub2

35.

Norwegian Mother, Father and Child Cohort Study (MoBa) (n.d.). Norwegian Institute of Public Health. Retrieved 5 June 2020 from https://www.fhi.no/en/studies/moba/

36.

O’Doherty

K. C.

Burgess

M. M.

(2013). Public deliberation to develop ethical norms and inform policy for biobanks: Lessons learnt and challenges remaining. Research Ethics, 9(2), 55-77. https://doi.org/10.1177/1747016113488858

37.

Ocloo

Matthews

(2016). From tokenism to empowerment: Progressing patient and public involvement in healthcare improvement. BMJ Quality & Safety, 25(8), 626-632. https://doi.org/10.1136/bmjqs-2015-004839

38.

OECD (2019). Enhancing access to and sharing of data: Reconciling risks and benefits for data re-use across societies. OECD Publishing. https://www-oecd–ilibrary-org.eu1.proxy.openathens.net/sites/276aaca8-en/../sti-2019-1215-en/index.html

39.

S. S.

Galanter

Thakur

Pino-Yanes

Barcelo

N. E.

White

M. J.

de Bruin

D. M.

Greenblatt

R. M.

Bibbins-Domingo

A. H. B.

Borrell

L. N.

Gunter

Powe

N. R.

Burchard

E. G.

(2015). Diversity in clinical and biomedical research: A promise yet to be fulfilled. PLoS Medicine, 12(12), e1001918. https://doi.org/10.1371/journal.pmed.1001918

40.

Olsen

Melbye

Olsen

S. F.

Sorensen

T. I.

Aaby

Andersen

A.-M. N.

Taxbøl

Hansen

K. D.

Juhl

Schow

T. B.

Sørensen

H. T.

Andresen

Mortensen

E. L.

Olesen

A. W.

Søndergaard

(2001). The Danish national birth cohort – its background, structure and aim. Scandinavian Journal of Public Health, 29(4), 300-307. https://doi.org/10.1177/14034948010290040201

41.

Pearson

(2015). Massive UK baby study cancelled. Nature, 526(7575), 620-621. https://doi.org/10.1038/526620a

42.

Phillips

Knoppers

B. M.

(2016). The discombobulation of de-identification. Nature Biotechnology, 34, 1102-1103. https://doi.org/10.1038/nbt.3696

43.

Prictor

Hemsley

Taylor

McCarthy

(2018, September 21). If privacy is increasing for My Health Record data, it should apply to all medical records. The Conversation. http://theconversation.com/if-privacy-is-increasing-for-my-health-record-data-it-should-apply-to-all-medical-records-102654

44.

Prictor

Huebner

Teare

H. J. A.

Burchill

Kaye

(2020). Australian Aboriginal and Torres Strait Islander collections of genetic heritage: The legal, ethical and practical considerations of a dynamic consent approach to decision making. The Journal of Law, Medicine & Ethics, 48(1), 205-217. https://doi.org/10.1177/1073110520917012

45.

Prictor

Teare

H. J. A.

Kaye

(2018). Equitable participation in biobanks: The risks and benefits of a ‘dynamic consent’ approach. Frontiers in Public Health, 6, 253. https://doi.org/10.3389/fpubh.2018.00253

46.

Richards

N. M.

King

(2014). Big data ethics. Wake Forest Law Review, 49, 393-432.

47.

Richter

Norris

Pettifor

Yach

Cameron

(2007). Cohort profile: Mandela's Children: The 1990 birth to twenty study in South Africa. International Journal of Epidemiology, 36(3), 504-511. https://doi.org/10.1093/ije/dym016

48.

Sanson

Johnstone

(2004). Growing up in Australia takes its first steps. Family Matters, 67, 46-53.

49.

Sumathipala

Fernando

D. J. S.

Siribaddana

S. H.

Abeysingha

M. R. N.

Jayasekare

R. W.

Dissanayake

V. H. W.

Silva

N. D.

(2000). Establishing a twin register in Sri Lanka. Twin Research and Human Genetics, 3(4), 202-204. https://doi.org/10.1375/twin.3.4.202

50.

Taylor

M. J.

Dove

E. S.

Laurie

Townend

(2018). When can the child speak for herself? The limits of parental consent in data protection law for health research. Medical Law Review, 26(3), 369-391. https://doi.org/10.1093/medlaw/fwx052

51.

Taylor

M. J.

Wilson

(2019). Reasonable expectations of privacy and disclosure of health data. Medical Law Review, 27(3), 432-460. https://doi.org/10.1093/medlaw/fwz009

52.

Teare

H. J. A.

de Masi

Banasik

Barnett

Herrgard

Jablonka

Postma

J. W. M.

McDonald

T. J.

Forgie

Chmura

P. J.

Rydzka

E. K.

Gupta

Brunak

Pearson

Kaye

(2018). The governance structure for data access in the DIRECT consortium: An innovative medicines initiative (IMI) project. Life Sciences, Society and Policy, 14(1), 20. https://doi.org/10.1186/s40504-018-0083-0

53.

Teare

H. J. A.

Prictor

Kaye

(2021). Reflections on dynamic consent in biomedical research: The story so far. European Journal of Human Genetics, 29(4), 649-656. https://doi.org/10.1038/s41431-020-00771-z

54.

The British Academy and the Royal Society (2017). Data management and use: Governance in the 21st century . https://royalsociety.org/∼/media/policy/projects/data-governance/data-management-governance.pdf

55.

UK Biobank (2007). UK biobank ethics and governance framework (Version 3.0) . https://www.ukbiobank.ac.uk/wp-content/uploads/2011/05/EGF20082.pdf

56.

Understanding Patient Data (n.d.). Retrieved 4 June 2020 from http://understandingpatientdata.org.uk/

57.

University College London (2015). Why did life study close? Life Study. http://www.lifestudy.ac.uk/about/faqs.html

58.

Vayena

Blasimme

(2017). Biomedical big data: New models of control over access, use and governance. Journal of Bioethical Inquiry, 14(4), 501-513. https://doi.org/10.1007/s11673-017-9809-6

59.

Wilkins

C. H.

(2018). Effective engagement requires trust and being trustworthy. Medical Care, 56(10 Suppl 1), S6-S8. https://doi.org/10.1097/MLR.0000000000000953

60.

Winickoff

D. E.

Winickoff

R. N.

(2003). The charitable trust as a model for genomic biobanks. New England Journal of Medicine, 12, 1180-1184. https://doi.org/10.1056/NEJMsb030036

61.

Zwitter

(2014). Big data ethics. Big Data & Society, 1(2), 1-6. https://doi.org/10.1177/2053951714559253

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Digital Mega-Studies as a New Research Paradigm: Governing the Health Research of the Future

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