Convergence of views

Our findings suggest that the groups of stakeholders with whom we engaged share common views regarding many of the strategies to adopt to move the PM agenda forward. As an illustration, the PIO representatives supported initiatives from policymakers to modernize drug approval mechanisms that were experienced to be too burdensome and time consuming [125]. Recently, efforts have been made to simplify bureaucratic requirements for drug approval [139], for instance through the implementation of innovative mechanisms such as adaptive pathways (mentioned in section 2.4), a progressive approach cited by some PIO representatives in paper III. The PIO representatives also called for increased efforts to contain costs of targeted treatments and genetic tests [125]; something that policymakers are trying to achieve by testing new ways to limit drug prices. One example is the use of value-based approaches for drug pricing under which drug reimbursement is tied to the actual value that the drug brings to health care systems [140]. Using such approaches, drugs that offer limited benefit (e.g. overall patient survival of less than one month) are priced lower than

those that offer long-term benefit (e.g. additional years of life). Value-based pricing, an approach also cited by the PIO representatives, is currently being explored in the United Kingdom and may contribute to contain costs of targeted treatments and genetic tests over time [141]. The PIO representatives also largely agreed with policymakers regarding the importance of developing infrastructures and tools for data sharing, and the need for conducting more genetic and epidemiological research [125]; policymakers have continuously encouraged the development of biobanks and research programs during last decades (see section 2.4).

Another area of convergence relates to the development of new and modern approaches for the collection and management of informed consent, and for communication and interface between researchers and research participants. During our consultations for the work presented in papers I, III and IV, PM researchers and PIO representatives repeatedly mentioned dynamic consent as a tool to implement in research projects [125-127]. Dynamic consent is a personalized, online consent and engagement tool which primarily aims to enable research participants to modify their consent preferences over time [115]. In addition, it can be used to establish two-way, ongoing communication between researchers and research participants [142]. For instance, researchers can provide regular updates about research discoveries, invite participants to discuss the research through online forums, and the participants can upload additional health information or fill in health questionnaires whenever needed [127]. As discussed in section 2.4, policymakers have encouraged the development of new forms of consent and communication between researchers and research participants over the last years. This is interesting knowing that, traditionally, there has been some reluctance toward fostering an ongoing researcher-research participant relationship [143]. For instance, in biobank research, the normal practice has been to collect the one-time, paper-based consent

of research participants to the broad use of their biological samples and associated health data within broadly defined research areas. No other forms of communication are usually expected to take place between researchers and research participants after the consent is collected, with the exception of some feedback of general information regarding the research, for instance through a Newsletter or a website. Increased interest in dynamic consent, also among PM researchers involved in biobanking [144], could be explained by a recognition that, in a continuously changing research environment, managing research projects is becoming increasingly difficult if no possibility exists to interact with research participants, for instance, to know whether they support data sharing, or to collect enrichment data. Increased interest in dynamic consent may also be explained by a recognition that research participants have an active role to play in research, and can contribute to improve its quality and relevance [145].

Finally, our findings suggest that there is some consensus among stakeholders regarding the need to facilitate interdisciplinary, cross-milieus collaborations, and challenging the traditional schism between research and health care. As reported in paper III, the PIO representatives explained that health care systems should do more to support and facilitate the recruitment of patients in clinical trials [125]. In paper I, the PM researchers recommended the establishment of expert networks spanning across clinical and research milieus in order to facilitate the feedback of genetic research results to research participants [126]. Policymakers increasingly encourage initiatives to create bridges between clinical and research milieus. As an illustration, in the recently launched Genomics England project [146] which aims to sequence the DNA codes of a 100,000 patients, expert networks similar to those recommended by the COST Action members have recently been implemented. These networks, called Clinical Interpretation Partnerships (GeCIP) [147], enable clinicians and researchers to work together to interpret the genetic and genomic data produced in the

research project and improve the clinical care provided to patients, but also to undertake complementary research [147]. The development of such networks is motivated by the observation that maintaining a clear separation between research and health care, as traditionally practiced, may no longer serve the interests of patients [148, 149]. As stated by Lyon and Segal, “the goal [of research] is increasingly patient-focused and intended to find information of clinical benefit to the participant, indeed blurring the lines between “patient”

and “research participant” [148]. Genomic research has the potential to contribute to yield a diagnosis [150] or identify a course of treatment within a clinically actionable time frame [148]. If such applications of genomics are to become common, increased collaboration between research and clinical milieus makes sense. However, as I will discuss later, it may also lead to a number of consequences for patients and research participants that should be considered.