“More powerful than the march of mighty armies is an idea whose time has come”
– Victor Hugo
The intellectual legacy of Rosamund Snow
Rosamund Snow was a social scientist with type 1 diabetes; she was an academic at the University of Oxford and a Patient Editor for The BMJ. She led the field in questioning conventional approaches to patient and public involvement in research and in mainstreaming a patient-led way of doing research.
Rosamund’s PhD was entitled “The role of patient expertise inside and outside the health system.” She addressed, from the patient’s perspective and using critical social science methodology, what it was like to attend a diabetes clinic.  This work led to a widely-cited academic paper “What happens when patients know more than their doctor?” which addressed knowledge imbalances between educated patients and non-specialist GPs.  She was critical of “partnership” approaches to research priority-setting, which she viewed as both tokenistic and paternalistic (she claimed that patients had been accused, for example, of proposing the “wrong kind of research questions”).  At The BMJ, she led a radical re-engineering of processes for including the patient voice in the journal. She co-authored (with me) another highly-cited paper addressing the question of whether evidence-based medicine is “biased” against the patient. 
Tragically, Rosamund died on 2nd February 2017. Her parents made a significant financial donation to help create an institute in her memory; this funding is being used to pump-prime the initiative with some doctoral fellowships. The applications are still open until January 2020. 
Patient involvement in research—a brief history
There is a well-described mismatch between the research that is done on a particular condition and the research that patients themselves would like to see done. [4,6] Formal research priority-setting partnerships aim to reduce this mismatch by involving patients in the selection of topics for research. [7, 8] The National Institute for Health Research (NIHR) has strongly supported patient and public involvement (PPI) in research, produced national benchmarks for PPI, funded INVOLVE (www.invo.org.uk) which promotes patient involvement in all aspects of biomedical research, and written up some exemplar case studies.[9, 10, 11] Co-design and co-delivery of research with patients and communities using “partnership” models is increasingly popular. [12,13]
While these and similar approaches have merits, all are designed and run by researchers (with greater or lesser efforts to achieve democratic governance); they are not led by patients.  Sarah White distinguishes nominal involvement of patients and the public (undertaken to confer legitimacy on a project), instrumental involvement (to improve its delivery and/or efficiency), representative involvement (to avoid creating dependency) and transformative involvement (to enable citizens to influence their own destiny).  Arguably, most of what is called “patient involvement” in medical research is nominal or instrumental in nature.
Transformative involvement of patients: a question of power
Social scientists have highlighted the persistence of power imbalances when well-meaning clinicians and scientists seek to “involve” patients in research.  In a paper called “Beware Zombies and Unicorns,” Mary Madden and Ewen Speed cautioned against aligning with an uncritical (instrumental) agenda for PPI and called for models of patient-led research that address fundamental questions about who holds the power and sets the agenda in research.  Helga Nowotny, past President of the European Research Council, has highlighted the need to “democratise expertise” when undertaking science with citizens (everyone is an expert—in different aspects of the problem).  Such partnerships, run democratically and with careful attention to the processes of governance and power-sharing, would align well with Simon Lock’s call for a People’s Research Council. 
A different kind of knowledge
Researchers in all fields become patients (and vice versa). Patient-led research may therefore include conventional forms of objective knowledge such as randomised controlled trials or bench science in the researcher’s own illness. More uniquely, patients bring experiential knowledge—the subjective, lived-body knowledge of what it is like to live with a particular illness or condition.
Experiential knowledge can be systematically explored through phenomenology (the study of what we can discern through our consciousness and senses ) and auto-ethnography (the study of one’s own experience in an unfamiliar world ). Experiential knowledge is complementary to knowledge generated in the laboratory or the clinical trial, potentially producing “multiple realities” as accounts of the patient experience clash with textbook descriptions of disease or the unsurfaced assumptions of clinicians and researchers. 
Another kind of knowledge that is unique to the patient experience (and ripe for research) is the collective knowledge generated by online communities. The growth of the “social web” has enabled the emergence of large (and increasingly research-aware) communities of individuals with a particular disease. Some of these communities share ideas for research and self-organise to undertake self-experimentation, self-surveillance and even analysis of their own genomic data. [23, 24]
Governing patient-led research: scientific rigour and ethics
If patient groups are to undertake and/or commission research, academic input (to match patients’ priorities and questions with appropriate theories and methodologies, and to support analysis and writing up) and capacity-building (training patients in research methods and techniques) are surely essential to ensure that patient-led research is scientifically defensible (and hence has credibility with clinicians and policymakers). [25,26]
The question of what counts as scientific rigour may itself be contested if the term is defined narrowly using the traditional scientific criteria of objectivity and distance. Martha Nussbaum, for example, has vigorously challenged the (arguably, flawed and gendered) view of science as necessarily dispassionate, uninvolved and emotionless; she considers emotion to be a dimension of scholarship without which science is impoverished and uncreative. 
Patient-led research raises both similar and different ethical challenges to conventional research. A comparison of patient-led research with standard research, for example, revealed six areas that are of potential relevance to ethical oversight: institutionalization, state recognition and support, incentive structures, openness, bottom-up approach, and self-experimentation. 
The productive role of conflict
In the best research programmes, the (productive) conflicts generated when patients’ experiential knowledge meets conventional research paradigms not only informs the wider research agenda, but transforms conventional researchers into more creative scientists who prioritise different questions and study them in imaginative and flexible ways. Vololona Rabeharisoa distinguishes between conventional “researchers in the lab” and patient groups, which she calls “researchers in the wild”.  Citing her own empirical work on the research interactions in rare diseases, she comments:
“We witnessed the trajectory of scientists who had started on the bench as biologists, then, as they exchanged with patient organizations, oriented themselves towards the clinic, and then returned to the bench with new research questions stemming from their observations, enriched by patients’ observations.”
Notwithstanding the potential for such creative conflict, a significant challenge for patient-led research is that it is often (understandably) underpinned by “cognitive passions”—that is, deeply-held, emotionally-charged perspectives on a condition. While such passions give energy and focus to a patient-led research agenda, they may mean that patients find it difficult to approach research into their own condition with the equipoise expected in science. However, while one high-profile patient-scientist conflict seemed to generate negative tension (chronic fatigue syndrome ); there are many counter-examples of conflicts that were highly productive, including in rare diseases, HIV/AIDS, mental health, and breast cancer. [22,29,30,31]
In short, there are many questions—both scientific (in the broad sense) and philosophical—that could be taken forward by an institute for patient-led research.
Whilst substantial additional funds will need to be raised to create a full-blown institute, a preliminary vision for such an institute is set out in Box 1.
|A vision for an institute for patient-led research
Such an institute would:
Trish Greenhalgh is professor of primary care health sciences at the University of Oxford.
- Snow R, Fulop N. Understanding issues associated with attending a young adult diabetes clinic: a case study. Diabetic Medicine 2012;29(2):257-59.
- Snow R, Humphrey C, Sandall J. What happens when patients know more than their doctors? Experiences of health interactions after diabetes patient education: a qualitative patient-led study. BMJ open 2013;3(11):e003583.
- Snow R, Crocker J, Crowe S. Missed opportunities for impact in patient and carer involvement: a mixed methods case study of research priority setting. Research Involvement and Engagement 2015;1(1):1-13.
- Greenhalgh T, Snow R, Ryan S, et al. Six ‘biases’ against patients and carers in evidence-based medicine. BMC medicine 2015;13(1):200. doi: 10.1186/s12916-015-0437-x [published Online First: 2015/09/02]
- Anonymous. The Rosamund Snow scholarship for patient-led research. Oxford: Green Templeton College. Accessed 1.1.19 on https://www.gtc.ox.ac.uk/students/how-to-apply/scholarships-bursaries/the-rosamund-snow-scholarship-for-patient-led-research/ 2019.
- Chalmers I, Bracken MB, Djulbegovic B, et al. How to increase value and reduce waste when research priorities are set. The Lancet 2014;383(9912):156-65.
- Petit-Zeman S, Firkins L, Scadding JW. The James Lind Alliance: tackling research mismatches. The Lancet 2010;376(9742):667-69.
- Deane KH, Flaherty H, Daley DJ, et al. Priority setting partnership to identify the top 10 research priorities for the management of Parkinson’s disease. BMJ open 2014;4(12):e006434.
- Denegri S. Going the extra mile: Improving the nation’s health and wellbeing through public involvement in research. Available on https://www.rds-yh.nihr.ac.uk/wp-content/uploads/2015/06/Going-the-Extra-Mile-Final.pdf. London: National Institute for Health Research 2015.
- National Institute for Health Research. UK Standards for Public Involvement in Research. . Southampton: NIHR. Accessed at https://sites.google.com/nihr.ac.uk/pi-standards/home on 1.11.19. 2019.
- National Institute for Health Research. The Public as our Partners. NIHR CCF Highlights from 2015/16. Available on https://www.nihr.ac.uk/about-us/how-we-are-managed/managing-centres/nihr-central-commissioning-facility/Documents/CCF_PPIEreport15_16SHORT.pdf. London: NIHR 2016.
- Greenhalgh T, Jackson C, Shaw S, et al. Achieving research impact through co-creation in community-based health services: literature review and case study. The Milbank quarterly 2016;in press
- Jagosh J, Macaulay AC, Pluye P, et al. Uncovering the benefits of participatory research: implications of a realist review for health research and practice. Milbank Quarterly 2012;90(2):311-46.
- Greenhalgh T, Hinton L, Finlay T, et al. Frameworks for supporting patient and public involvement in research: Systematic review and co‐design pilot. Health Expectations 2019
- White SC. Depoliticising development: the uses and abuses of participation. Development in practice 1996;6(1):6-15.
- Edelman N, Barron D. Evaluation of public involvement in research: time for a major re-think? Journal of health services research & policy 2015:1355819615612510.
- Madden M, Speed ES. Beware Zombies and Unicorns: Towards critical patient and public involvement in health research in a neoliberal context. Frontiers in Sociology 2017;2:7.
- Nowotny H. Democratising expertise and socially robust knowledge. Science & Public Policy (SPP) 2003;30(3)
- Lock SJ. People’s Research Council : A report on proposals for its implementation. Accessed 7.6.17 on https://jeremytill.s3.amazonaws.com/uploads/post/attachment/51/PRC_report.pdf. London: Wellcome Trust 2010.
- Pols J. Knowing patients: turning patient knowledge into science. Science, Technology, & Human Values 2014;39(1):73-97.
- Greenhalgh T. Adjuvant chemotherapy: an autoethnography. Subjectivity 2017;10(4):340-57.
- Rabeharisoa V. The multiplicity of knowledge and the trembling of institutions. Revue d’anthropologie des connaissances 2017;11(2):ao-au.
- Vayena E, Tasioulas J. Adapting standards: ethical oversight of participant-led health research. PLoS medicine 2013;10(3):e1001402.
- Swan M. Health 2050: The realization of personalized medicine through crowdsourcing, the quantified self, and the participatory biocitizen. Journal of personalized medicine 2012;2(3):93-118.
- Wenner DM, Kimmelman J, London AJ. Patient-Funded Trials: Opportunity or Liability? Cell stem cell 2015;17(2):135-37.
- Steuli J, Vayena E. The promising revolution of participant-led research in rare neurological diseases; Potential benefits and pitfalls. Accessed 7.6.17 on https://www.researchgate.net/profile/Juerg_Streuli/publication/293769190_The_Promising_Revolution_of_Participant-Led_Research_in_Rare_Neurological_Diseases/links/56bb11cb08ae0a6bc9560d68.pdf: Epileptologie, 2016.
- Nussbaum MC. Upheavals of thought: The intelligence of emotions: Cambridge University Press 2003.
- McKie R. Chronic fatigue syndrome researchers face death threats from militants. Accessed 9.7.17 on https://www.theguardian.com/society/2011/aug/21/chronic-fatigue-syndrome-myalgic-encephalomyelitis. The Guardian 2011;21st August
- Epstein S. The construction of lay expertise: AIDS activism and the forging of credibility in the reform of clinical trials. Science, Technology, & Human Values 1995;20(4):408-37.
- Tomes N. The patient as a policy factor: a historical case study of the consumer/survivor movement in mental health. Health Affairs 2006;25(3):720-29.
- Osuch JR, Silk K, Price C, et al. A historical perspective on breast cancer activism in the United States: from education and support to partnership in scientific research. Journal of Women’s Health 2012;21(3):355-62.