4.1.1. Paper I - Email survey and Coimbra workshop
Paper I aimed to investigate the views of PM researchers involved in the COST Action regarding potential challenges to the feedback of genetic research results to research participants in Europe, and solutions to address these challenges. The method selected to investigate this issue was guided by a requirement of the Action in its Memorandum of Understanding [19] to hold an annual workshop among its members on this topic, and produce a working paper (paper I). The workshop was planned to take place in October 2014.
To lay the ground for discussions among workshop participants, we decided to collect data prior to the workshop (in July 2014) by sending an email survey to the Action members in the 25 participating countries. The survey consisted of an open-ended questionnaire inquiring about challenges and practical issues that the Action members could encounter related to providing feedback of genetic research results to research participants (Appendix A). The questionnaire also asked for suggestions to address these challenges. The survey respondents were asked, whenever possible, to coordinate responses within their country. In total, 19 questionnaires were collected through the email survey, and they provided information from 14 of the 25 COST Action countries.
In October 2014, the Research Ethics working group (WG1) of the Action organized a one-day workshop at the University of Coimbra, Portugal. The workshop gathered 43 Action members from 21 European countries and was designed as an open space technology (OST) meeting. OST meetings do not require a specific detailed agenda but are rather conducted as open forums where discussions on a few broadly defined topics are encouraged. The
workshop was moderated by a COST Action member with expertise in leading OST events.
To facilitate discussions, a case study (Appendix B) was used requiring workshop participants to anticipate how they would organize the feedback of individual genetic research results to research participants at their home institution, the challenges they expected to encounter, and the types of solutions that would be needed to address these challenges. Notes were taken by appointed participants during the workshop and collected by the working group chairs at the end of the day. The data collected through the survey questionnaire (sent out in July 2014), and notes from the workshop, were gathered and analysed using a qualitative content analysis approach as described later in section 4.2. These data formed the basis for paper I.
4.1.2. Paper II - Literature review
Paper II aimed to investigate the perspectives of policymakers regarding the types of behaviours and practices expected of citizens to support the realization of PM. During recent years, policymakers have published several reports that detail the steps required for the realization of PM, and the kind of behaviours and practices that are expected from patients and citizens to support PM. It was therefore decided that a systematic review of these reports would be useful to gain insight into 1) the types of roles and responsibilities that patients and citizens are envisioned to endorse under PM, 2) the implications of these expectations for citizens, and 3) potential barriers that exist, which may prevent citizens from endorsing these roles. To our knowledge, this review would be the first to focus specifically on how the reports describe new behaviours of patients and citizens under PM. The benefit of conducting such review is that it provides good insight into what policymakers in different countries expect from patients and citizens. An Internet search was conducted using Google and the following search terms: [‘personalized medicine’] and/or [personalised medicine] and/or [‘stratified medicine’] and/or [‘precision medicine’] combined with [report] and [pdf]. In
total, 18 publicly available reports published between 2008 and 2013 and written in English were selected and reviewed to identify sections of text addressing envisaged behaviours and/or practices of patients and/or citizens. These reports are listed in the paper [121]. The sections describing behaviours and/or practices of patients and/or citizens were extracted and compiled into a list of verbatim texts. The data collected were analysed using a qualitative content analysis approach as described later in section 4.2.
4.1.3. Paper III - Interviews
Paper III aimed to investigate the views of patient and interest organizations (PIOs) regarding potential challenges to the realization of PM, and strategies to address these challenges. We decided to engage directly with the PIOs by approaching their leading representatives and inviting them to discuss PM in individual interviews. First, an e-mail invitation was sent to the leading representatives of a small number of PIOs that co-operate with the NCGC. The invitation provided an outline of the study objectives and a description of what participation in the study entailed1. Those who accepted our invitation helped to identify other PIO representatives that might want to participate in the study. Such snowball sampling enabled the recruitment of representatives from 8 PIOs concerned with one specific disease or disease area. Second, the same e-mail invitation was sent to 20 leaders of disease-specific PIOs members of the European Patients’ Forum (EPF), an umbrella organization that works with patient groups and health advocacy organizations across Europe [122]. This further enabled the recruitment of 5 PIO representatives. Between July 2014 and January 2015, semi-structured individual telephone interviews were conducted with 10 organizational leaders (e.g.
CEO, secretary general, and director) and 3 senior managers, representing in total 13 PIOs.
1 In the information provided to representatives, we used the acronym PAOs (patient advocacy organizations) to describe patient groups. Later, after we had conducted the interviews, we changed the acronym to PIOs to reflect more accurately the way the organizations defined themselves.
The PIOs worked within the following areas: cancer (4), hereditary and genetic disorders (3), mental health (1), diabetes (1), psoriasis (1), AIDS (1), lupus (1), and primary immunodeficiencies (1). All participants signed an informed consent form (Appendix C) and the study was approved by the Norwegian Social Science Data Services (Appendix D and E).
The interviews were conducted using an interview guide (Appendix F) which included open-ended questions about the PIOs’ perspectives regarding PM, PM-related activities, perceived challenges with regard to the realization of PM, recommendations for the adoption of PM, and the potential roles the PIOs may play in the realization of PM. The interviews lasted, on average, 40 minutes and were audio recorded and transcribed verbatim in English and Norwegian for data analysis using a qualitative content analysis approach as described in section 4.2.
4.1.4. Paper IV - Oxford workshop
Paper IV aimed to investigate the views of PM researchers regarding how dynamic consent may help address some of the challenges usually encountered in PM-friendly research. Both the NCGC and the COST Action had an interest in investigating dynamic consent approaches.
The Action called for “coordination of analysis of ICT solutions for dynamic consent and research subject participation” and the production of a working paper [19]. It was therefore decided to organize a workshop (the preferred method of consultation in the Action, following the principles of the RRI framework). In October 2015, the Research Ethics working group (WG1) of the Action and the Centre for Health, Law and Emerging Technologies (HeLEX) at the University of Oxford jointly organized an interdisciplinary two-day workshop gathering PM researchers experienced in the use of dynamic consent solutions [123]. The workshop brought together ethicists, lawyers, clinicians, medical researchers, research nurses and research participants to discuss how dynamic consent approaches may facilitate the conduct
of biomedical research. During the workshop, the participants mapped a process flowchart describing main tasks and goals for recruiting, enrolling, and maintaining participants in research studies and identified challenges usually encountered when carrying out these tasks.
Then, they discussed how dynamic consent may facilitate the conduct of these tasks and attainment of the goals. Input from researchers involved in the design of dynamic consent solutions for their projects were central to the discussions. Appointed participants took notes during the workshop, and the workshop organizers gathered these notes, summarised their content, and analysed the data using a content analysis approach as described below.