There is little information in the medical literature about ongoing communication with participants enrolled in long-term prospective studies. The results of these focus groups and ongoing consultation with the external CAG and ESAB highlight the need for ongoing consultation with the study population. All groups highlighted the importance of community trust in the Marshfield Clinic and how detrimental it would be to the study to lose that trust. High levels of confidence in the organization conducting the research was found in a study in England to be important in decisions about taking part in a genetic epidemiology study, despite misunderstandings of some of the key concepts of the study [Dixon-Woods et al., 2007
Two recent empirical studies found strong subject desire for research results from genetic studies [Beskow et al., 2008
; Kaufman et al., 2008
]. In a study of forty subjects in North Carolina where subjects were asked to review a consent form document, researchers found that more than half thought it would be very important to receive general news about studies being done through a biorepository, and several thought it would constitute fair exchange for having volunteered in the study[Beskow et al., 2008
]. Nearly two-thirds were not concerned about not receiving personal genetic results. An on-line study of adults across the US assessing opinions about a potential national genetics study found that 3 in 4 people would be less likely to participate in such a study without return of research results [Kaufman et al., 2008
]. These results are similar to several findings related to sharing of research results that we have documented in our PMRP cohort. We found previously, in a 10% random sample of the PMRP cohort, that 61% of participants were unsure or thought that they would learn, as a result of participating, which conditions/diseases they would develop, despite the fact that the written informed consent document states that they will not receive any personal genetic information from the study [McCarty et al., 2007B
]. From this current focus group study and the feedback we have received from our CAG, it is clear that we need to find a way to provide study information to all study subjects, regardless of whether they opted out of the study newsletter in the initial consent process. Prospectively, we have removed the opt-out from the consent form, because ongoing communication with study subjects in an open-ended biobank study is essential.
In the current focus group and advisory group discussions, the majority of participants and members felt that information about minimal risk protocol changes to enrolled subjects with the option for an opt-out would be sufficient to then apply for a waiver of written informed consent from the IRB. In additional to being impractical to attempt active reconsent for a biobank of 20,000 subjects, anecdotally it was felt that a requirement for an opt-in instead of an opt-out would significantly impact participation in the protocol change. A cross-sectional study of 177 older patients pre- and post-waiver of written informed consent and Health Insurance Portability and Accountability Act (HIPAA) authorization found that there was a substantial increase in participation rates post-waiver for a minimal risk study [Krousel-Wood et al., 2006
]. Protocol changes involving more than minimal risk would require a different process for informing subjects of the change and may require active reconsent; they were not considered as part of this study and process.
The results of a series of focus groups across the US addressing public opinion on informed consent and privacy concerns for biobanking were reported recently [Kaufman et al., 2009
; Murphy et al., 2009
]. Greater than 75% of these participants in focus groups from larger cities across the US felt that they would have more trust in the study and would feel more respected and involved if researchers had to get their permission to use their hypothetical samples in a biobank before each new project [Murphy et al., 2009
]. Twenty-seven percent felt that it would be a waste of time and money, and 265 felt that they would be bothered by this process. Ninety percent of participants expressed concern about privacy, and 60% said that they would participate in a biobank despite these concerns [Kaufman et al., 2009
]. Ninety-two percent would allow sharing of data with academic researchers, and 75% would allow sharing with industry. The participants were nearly equally divided on whether they would prefer to give a one-time only consent with oversight review or would want to provide consent for each project separately. The level of concern expressed by these focus group participants is somewhat higher than expressed by the PMRP focus groups and CAG members. There are likely two reasons for these differences. First, the PMRP focus groups were conducted with biobank participants, while the national sample was conducted with people in hypothetical situations. Participation in the PMRP biobank is slightly less than 50%, and it is likely that people who had levels of concern about privacy and data sharing as outlined in the PMRP informed consent document elected not to participate in the biobank. Those who chose to participate, although they have privacy concerns as expressed in the focus groups, ultimately felt that the potential scientific good outweighed the potential for a privacy breach. The second possible reason for the observed difference is the relationship of the Marshfield Clinic to the community from which people were recruited. As expressed by both the PMRP focus groups members and the CAG, they trust the Marshfield Clinic, and this trust led them to participate in the biobank. In contrast to the national focus groups, respondents were considering an entity whom they knew rather than a hypothetical situation. Consistent across the earlier PMRP focus groups and the recent focus groups was the need for regular communication about studies using the biobank.
The limitations of our approach lie in the generalizability of the results and the fact that a qualitative approach can be difficult to replicate. The effectiveness of advisory boards is very much dependent on the membership. Half of all working age adults in Marshfield are employed by either Marshfield Clinic or St. Joseph’s Hospital so the medical campus is very much a part of the community. Feelings towards health care institutions and the research being conducted there might differ in large metropolitan areas where people seek their health care at different institutions and may not feel the same allegiance that they do in a small community. Nevertheless, we feel that this approach to community engagement and feedback for large scale biobanks may be applicable in other settings.
In summary, ongoing communication with study participants is essential to maintain the trust of the community, especially where study duration is long, with evolving scientific and ethical issues. For the particular issue under consideration, because of the broad consent given initially, the focus group participants, CAG, and ESAB were comfortable with an opt-out model for protocol changes that are minimal risk, such as accessing residual blood samples. Given the duration of the biobank, communication at regular intervals is necessary for all study participants, particularly those who initially elected not to receive study newsletters. This multi-faceted approach to stakeholder communication has been successful for the PMRP biobank and may be a potential model for others to consider.