Political views and organizational distrust affect rural residents' willingness to share personal data for COVID-19 contact tracing: A cross-sectional survey study.

Background: We aimed to examine the attitudes of Pennsylvania rural residents toward data sharing in the setting of the COVID-19 pandemic. Specifically, we were interested in better understanding their willingness to provide personal information for contact tracing to public health staff investigating COVID-19 cases, as well as their concerns. We used a validated scale to describe the influence of distrust of healthcare organizations on their attitudes. Methods: We mailed 4000 surveys to rural residents identified from the electronic medical record of a healthcare system in central Pennsylvania. Data were entered into a REDCap database and analyzed using descriptive summaries, and both binomial and multivariable logistic regression. Results: Binomial logistic regression showed that both distrust in healthcare organizations and political values influence respondents' willingness to share information with contact tracers as well as their concerns about sharing personal data. When our multivariable model was applied, political values remained and were consistently associated with willingness to share and concerns about sharing their data. Conclusion: This study is a first step in eliciting rural residents' willingness to share personal data for contact tracing by public health officials. Understanding and addressing rural residents' willingness to share personal data and their concerns about sharing those data will help public health officials identify effective strategies for managing COVID-19 and future pandemics in rural communities. By involving community members at the ground level, public health staff can ensure residents' buy-in for the need to collect their personal data, thereby helping to mitigate the public health crises.

Current landscape of research ethics consultation services: National survey results.

Introduction: The goal of a research ethics consultation service (RECS) is to assist relevant parties in navigating the ethical issues they encounter in conduct of research. The goal of this survey was to describe the current landscape of research ethics consultation and document if and how it has changed over the last decade. Methods: The survey instrument was based on the survey previously circulated. We included a number of survey domains from the previous survey with the goal of direct comparison of outcomes. The survey was sent to 57 RECS in the USA and Canada. Results: Forty-nine surveys were completed for an overall response rate of 86%. With the passing of 10 years, the volume of consults received by RECS surveyed has increased. The number of consults received by a subset of RECS remains low. RECS continues to receive requests for consults from a wide range of stakeholders. About a quarter of RECS surveyed actively evaluate their services, primarily through satisfaction surveys routinely shared with requestors. The number of RECS evaluating their services has increased. We identified a group of eight key competencies respondents find as key to providing RECS. Conclusions: The findings from our survey demonstrate that there have been growth and development of RECS since 2010. Further developing evaluation and competency guidelines will help existing RECS continue to grow and facilitate newly established RECS maturation. Both will allow RECS personnel to better serve their institutions and add value to the research conducted.

Mining the Data: Exploring Rural Patients' Attitudes about the Use of Their Personal Information in Research.

BACKGROUND: This study examines rural patients' perceived importance of knowing or being consulted about researchers' access and use of their personal data (identifiable and de-identified health information, and identifiable and de-identified non-health information) across five scenarios. This study also examines their views on stewardship or governance of their personal information by researchers in their healthcare systems. METHODS: We conducted a survey by mail. Data were analyzed using descriptive statistics. Multivariable regression analyses were conducted across each scenario and type of personal data with the same variables included in each model. RESULTS: The majority of participants said it was "very important/absolutely essential" to know the purpose of the study, to be asked every time, and to know the policies governing researcher access and use of their identifiable health information. Just over two-thirds of respondents thought it "very important/absolutely essential" to know who serves on the data governance committee and to have a community member serve. Distrust in healthcare organizations was positively correlated with the scenarios while willingness to give permission to donate leftover biological specimens was negatively correlated. CONCLUSION: Our study findings indicate that the type of personal information being accessed and used generally matters to 1,407 patients living in rural Pennsylvania. We also demonstrate that knowing their healthcare organizations' governance policies and practices for managing their personal data is important to many rural Pennsylvania patients. Biomedical researchers need to recognize and attend to those differences as much as possible in order to expand opportunities for and participation in research by residents of these rural communities.Supplemental data for this article is available online at.

Exploring public concerns for sharing and governance of personal health information: a focus group study.

OBJECTIVE: Researchers are increasingly collecting large amounts of deidentified data about individuals to address important health-related challenges and answer fundamental questions. Current US federal regulations permit researchers to use already collected and stored deidentified health-related data from a variety of sources without seeking consent from patients. The objective of this study was to investigate public views on the policies and processes institutions have in place for accessing, using, and sharing of data. MATERIALS AND METHODS: We conducted 5 focus groups with individuals living within a 20-mile radius of the local academic medical center. We also held a focus group with undergraduates at a local university. RESULTS: A total of 37 individuals participated, ages 18-76. Most participants were not surprised that researchers accessed and used deidentified personal information for research, and were supportive of this practice. Transparency was important. Participants wanted to know when their data were accessed, for what purpose, and by whom. Some wanted to have some control over the use of their data valuing the chance to opt-out. Finally, participants supported establishment of an advisory council or group with responsibility for deciding what data were used, who was accessing those data, and whether data could be shared. DISCUSSION AND CONCLUSIONS: The trust people have in their local institutions should be considered fragile, and institutions should not take that trust for granted. How institutions choose to govern patients' data and what voices are included in decisions about use and access are critical to maintaining the trust of the public.

Creating a Research Ethics Consultation Service: Issues to Consider.

This article provides pragmatic advice for organizations interested in creating a research ethics consultation service (RECS). A robust RECS has the potential to build capacity among investigators to identify and consider the ethical issues they encounter while conducting their research. Determining whether to establish an RECS should begin with an institutional-needs assessment that includes three key questions: What are the current resources available to research teams to navigate ethical concerns that arise from their research? Is there a demand or perceived need for more resources? Is there institutional support (financial and otherwise) to establish and maintain an RECS? If this results in the decision to establish the consultation service, relevant institutional stakeholders must be identified and consulted, and personnel with the requisite skills recruited. The next step is to establish an RECS and build the infrastructure to process and respond to requests. The RECS's long-term sustainability will depend on a stable source of funding and a mechanism to receive constructive feedback to ensure that the service is meeting the institutional needs it set out to address.

Informed consent: Old and new challenges in the context of the COVID-19 pandemic.

In this paper, we address how the COVID-19 pandemic has impacted informed consent for clinical research through examining experiences within Clinical and Translation Science Award (CTSA) institutions. We begin with a brief overview of informed consent and the challenges that existed prior to COVID-19. Then, we discuss how informed consent processes were modified or changed to address the pandemic, consider what lessons were learned, and present research and policy steps to prepare for future research and public health crises. The experiences and challenges for CTSA institutions offer an important perspective for examining what we have learned about informed consent and determining the next steps for improving the consent process.

"They're Not Going to Do Nothing for Me": Research Participants' Attitudes towards Elective Genetic Counseling.

As applications of genomic sequencing have expanded, offering genetic counseling support to all patients is arguably no longer practical. Additionally, whether individuals desire and value genetic counseling services for genomic screening is unclear. We offered elective genetic counseling to 5110 individuals prior to undergoing sequencing and 2310 participants who received neutral results to assess demand. A total of 0.2% of the study participants accessed genetic counseling services prior to sequencing, and 0.3% reached out after receiving neutral results. We later conducted 50 interviews with participants to understand why they did not access these services. Many interviewees did not recall the availability of genetic counseling and were unfamiliar with the profession. Interviewees described not needing counseling before sequencing because they understood the study and felt that they could cope with any result. Counseling was considered equally unnecessary after learning neutral results. Although the participants had questions about their results, they did not feel that speaking with a genetic counselor would be helpful. Genomic screening efforts that employ opt-in models of genetic counseling may need to clarify the potential value of genetic counseling support from the outset and feature genetic counseling services more prominently in program materials.

At a Moment's Notice: Community Advisory Board Perspectives on Biobank Communication to Supplement Broad Consent.

INTRODUCTION: To address ethical concerns about the of future research authorization, biobanks employing a broad model of consent can design ongoing communication with contributors. Notifying contributors at the time of sample distribution provides one form of communication to supplement broad consent. However, little is known about how community-informed governance might anticipate contributor responses and inform communication efforts. OBJECTIVE: We explored the attitudes of members of a three-site Community Advisory Board (CAB) network. CAB members responded to a hypothetical proposal for notifying biobank contributors at the time of sample distribution to researchers utilizing the biobank. METHODS: We used regularly scheduled CAB meetings to facilitate 3 large-group and 6 small-group discussions. Discussions were audio-recorded, transcribed, and analyzed for thematic content using descriptive thematic analysis. RESULTS: The results challenged our expectation of general support for the proposed communications. While CAB members identified some advantages, they were concerned about several potential harms to biobank contributors and the biobank. The CABs understood biobank communication in terms of an ongoing relationship with the biobank and a personal contribution to research. CONCLUSION: Our findings contribute to the emerging literature on community engagement in biobanking. Additional communication with biobank contributors can serve a variety of value-based objectives to supplement broad consent. Design of communication efforts by biobanks can be improved by CAB members' anticipation of the unintended consequences of additional contact with contributors. CAB members' holistic interpretation of communication efforts suggests that biobank leadership considers all communication options as part of a more comprehensive communications strategy.

Assessing the stability of biobank donor preferences regarding sample use: evidence supporting the value of dynamic consent.

Dynamic consent has been proposed as a strategy for addressing the limitations of traditional, broad consent for biobank participation. Although the argument for dynamic consent has been made on theoretical grounds, empirical studies evaluating the potential utility of dynamic consent are needed to enhance deliberations about the merits of dynamic consent. Few studies have assessed such considerations as whether donor preferences may change over time or if participants would use a dynamic consent mechanism to modify preferences when they change. We administered a 66-item survey to participants in a large DNA biobank. The survey sought to gauge the stability of donor preferences specified at the time of biobank enrollment, specifically the stability of donors' preference regarding posthumous availability of biospecimens to next-of-kin. We received 1164 completed surveys for a response rate of 72%. Forty percent of respondents indicated a preference regarding sample availability on the survey (T2) that was inconsistent with the preference they had expressed when they enrolled in the biobank (T1). Most (94%) individuals with inconsistent preferences regarding sample availability had initially restricted sample availability at T1 but were comfortable with broader availability when asked at the time of the survey (T2). Our findings demonstrate that preferences regarding sample use expressed at the time of enrollment in a DNA biobank may not be reliable indicators of donor preferences over time. These findings lend empirical support to the case for a dynamic consent model in which biobank participants are approached over time to clarify their views regarding sample use.

Parent Perspectives Towards Genetic and Epigenetic Testing for Autism Spectrum Disorder.

Examining community views on genetic/epigenetic research allows collaborative technology development. Parent perspectives toward genetic/epigenetic testing for autism spectrum disorder (ASD) are not well-studied. Parents of children with ASD (n = 131), non-ASD developmental delay (n = 39), and typical development (n = 74) completed surveys assessing genetic/epigenetic knowledge, genetic/epigenetic concerns, motives for research participation, and attitudes/preferences toward ASD testing. Most parents (96%) were interested in saliva-based molecular testing for ASD. Some had concerns about privacy (14%) and insurance-status (10%). None (0%) doubted scientific evidence behind genetic/epigenetic testing. Most reported familiarity with genetics (88%), but few understood differences from epigenetics (19%). Child developmental status impacted insurance concerns (p = 0.01). There is broad parent interest in a genetic/epigenetic test for ASD. It will be crucial to carefully consider and address bioethical issues surrounding this sensitive topic while developing such technology.

Patients' views on variants of uncertain significance across indications.

As genomic sequencing expands into more areas of patient care, an increasing number of patients learn of the variants of uncertain significance (VUSs) that they carry. Understanding the potential psychosocial consequences of the disclosure of a VUS can help inform pre- and post-test counseling discussions. Medical uncertainty in general elicits a variety of responses from patients, particularly in the growing field of medical genetics and genomics. It is important to consider patients' responses to the ambiguous nature of VUSs across different indications and situational contexts. Genetic counselors and other providers ordering genetic testing should be prepared for the possibility of their patients' misinterpretation of such results. Pre-test counseling should include a discussion of the possibility of VUSs and what it would mean for the patient's care and its potential psychosocial impacts. When a VUS is found, post-test counseling should include additional education and a discussion of the variant's implications and medical management recommendations based on the results. These discussions may help temper subjective interpretations, unrealistic views, and decisional regret.

The Responsibility to Recontact Research Participants after Reinterpretation of Genetic and Genomic Research Results.

The evidence base supporting genetic and genomic sequence-variant interpretations is continuously evolving. An inherent consequence is that a variant's clinical significance might be reinterpreted over time as new evidence emerges regarding its pathogenicity or lack thereof. This raises ethical, legal, and financial issues as to whether there is a responsibility to recontact research participants to provide updates on reinterpretations of variants after the initial analysis. There has been discussion concerning the extent of this obligation in the context of both research and clinical care. Although clinical recommendations have begun to emerge, guidance is lacking on the responsibilities of researchers to inform participants of reinterpreted results. To respond, an American Society of Human Genetics (ASHG) workgroup developed this position statement, which was approved by the ASHG Board in November 2018. The workgroup included representatives from the National Society of Genetic Counselors, the Canadian College of Medical Genetics, and the Canadian Association of Genetic Counsellors. The final statement includes twelve position statements that were endorsed or supported by the following organizations: Genetic Alliance, European Society of Human Genetics, Canadian Association of Genetic Counsellors, American Association of Anthropological Genetics, Executive Committee of the American Association of Physical Anthropologists, Canadian College of Medical Genetics, Human Genetics Society of Australasia, and National Society of Genetic Counselors.

Pancreatic cancer and melanoma related perceptions and behaviors following disclosure of CDKN2A variant status as a research result.

PURPOSE: This study examined whether participants who learned research results related to a germline CDKN2A variant known to be associated with increased risk of pancreatic cancer and malignant melanoma would pursue confirmatory testing and cancer screening, share the genetic information with health care providers and family, and change risk perceptions. METHODS: Participants were pancreas research registry enrollees whose biological sample was tested in a research laboratory for the variant. In total, 133 individuals were invited to learn a genetic research result and participate in a study about the disclosure process. Perceived cancer risk, screening intentions, and behaviors were assessed predisclosure, immediately postdisclosure, and six months postdisclosure. RESULTS: Eighty individuals agreed to participate and 63 completed the study. Immediately postdisclosure, carriers reported greater intentions to undergo pancreatic cancer and melanoma screening (p values ≤0.024). Seventy-three percent of carriers (47.5% noncarriers) intended to seek confirmatory testing within six months and 20% (2.5% noncarriers) followed through. All participants shared results with ≥1 family member. More carriers shared results with their health care provider than noncarriers (p = 0.028). CONCLUSION: Recipients of cancer genetic research results may not follow through with recommended behaviors (confirmatory testing, screening), despite stated intentions. The research result disclosure motivated follow-up behaviors among carriers more than noncarriers.

Managing the Unimaginable: Biobank Participant Views on Reconsent for Whole Genome Sequencing of Stored Biospecimens.

Background: DNA biobanks frequently obtain broad permissions from sample donors, who agree to allow their biospecimens to be used for a variety of future purposes. A limitation of this approach is that it may not be possible to discuss or anticipate all potential uses of biospecimens at the time patient consent is obtained. We surveyed biobank participants to clarify their views regarding the need to be informed about research involving whole genome sequencing (WGS). Methods: We invited 1200 participants in the Mayo Clinic Biobank to complete a survey inquiring about their support for WGS; their interest in being recontacted before WGS of their biospecimens; whether they would consent to WGS if asked; and the acceptability of proceeding with WGS if sample donors could not be reached. Results: Six hundred eighty-seven biobank participants returned completed surveys (57% response). More than 96% of biobank participants were supportive of WGS and would give permission for WGS of their sample, if asked. Nonetheless, 61% of biobank participants felt they should be recontacted before WGS was done. Participants were divided regarding the permissibility of conducting WGS if efforts to recontact sample donors were unsuccessful. Discussion: Our findings highlight a potential discrepancy between the broad permissions granted by biobank participants at the time they donated biospecimens and their views about the application of WGS to their samples. Biobank participants appear to value the ability to confirm their commitment to genetic research when the studies in question involve WGS, a technological capacity they may not have anticipated at the time they donated their biospecimens. Efforts to reevaluate biobank participants' views about the acceptability of new technologies may help to ensure alignment of participants' current beliefs and research applications that would have been difficult to anticipate at the time biospecimens were collected.

Enrichment sampling for a multi-site patient survey using electronic health records and census data.

Objective: We describe a stratified sampling design that combines electronic health records (EHRs) and United States Census (USC) data to construct the sampling frame and an algorithm to enrich the sample with individuals belonging to rarer strata. Materials and Methods: This design was developed for a multi-site survey that sought to examine patient concerns about and barriers to participating in research studies, especially among under-studied populations (eg, minorities, low educational attainment). We defined sampling strata by cross-tabulating several socio-demographic variables obtained from EHR and augmented with census-block-level USC data. We oversampled rarer and historically underrepresented subpopulations. Results: The sampling strategy, which included USC-supplemented EHR data, led to a far more diverse sample than would have been expected under random sampling (eg, 3-, 8-, 7-, and 12-fold increase in African Americans, Asians, Hispanics and those with less than a high school degree, respectively). We observed that our EHR data tended to misclassify minority races more often than majority races, and that non-majority races, Latino ethnicity, younger adult age, lower education, and urban/suburban living were each associated with lower response rates to the mailed surveys. Discussion: We observed substantial enrichment from rarer subpopulations. The magnitude of the enrichment depends on the accuracy of the variables that define the sampling strata and the overall response rate. Conclusion: EHR and USC data may be used to define sampling strata that in turn may be used to enrich the final study sample. This design may be of particular interest for studies of rarer and understudied populations.

Why Health Professionals Should Speak Out Against False Beliefs on the Internet.

Broad dissemination and consumption of false or misleading health information, amplified by the internet, poses risks to public health and problems for both the health care enterprise and the government. In this article, we review government power for, and constitutional limits on, regulating health-related speech, particularly on the internet. We suggest that government regulation can only partially address false or misleading health information dissemination. Drawing on the American Medical Association's Code of Medical Ethics, we argue that health care professionals have responsibilities to convey truthful information to patients, peers, and communities. Finally, we suggest that all health care professionals have essential roles in helping patients and fellow citizens obtain reliable, evidence-based health information.

How Should a Research Ethicist Combat False Beliefs and Therapeutic Misconception Risk in Biomedical Research?

Therapeutic misconception can be especially challenging at large research-intensive academic medical centers, where boundaries between clinical care and research can become murky. In early stage clinical trials, for example, physicians often encourage patients to enroll in a drug or an intervention study as part of a treatment plan. As a research ethicist, I have found myself having to temper researchers' enthusiasm to prevent their overemphasizing positive benefits to participants. One strategy I've used is to encourage researchers to collaborate with treating physicians and to continually engage participants in assessing risks and benefits. This strategy has been helpful not only in early stage trials but also in translational genomic studies in which research can be used in part as a means of making costly testing available to patients.

Parents' attitudes toward consent and data sharing in biobanks: A multisite experimental survey.

BACKGROUND: The factors influencing parents' willingness to enroll their children in biobanks are poorly understood. This study sought to assess parents' willingness to enroll their children, and their perceived benefits, concerns, and information needs under different consent and data-sharing scenarios, and to identify factors associated with willingness. METHODS: This large, experimental survey of patients at the 11 eMERGE Network sites used a disproportionate stratified sampling scheme to enrich the sample with historically underrepresented groups. Participants were randomized to receive one of three consent and data-sharing scenarios. RESULTS: In total, 90,000 surveys were mailed and 13,000 individuals responded (15.8% response rate). 5737 respondents were parents of minor children. Overall, 55% (95% confidence interval 50-59%) of parents were willing to enroll their youngest minor child in a hypothetical biobank; willingness did not differ between consent and data-sharing scenarios. Lower educational attainment, higher religiosity, lower trust, worries about privacy, and attitudes about benefits, concerns, and information needs were independently associated with less willingness to allow their child to participate. Of parents who were willing to participate themselves, 25% were not willing to allow their child to participate. Being willing to participate but not willing to allow one's child to participate was independently associated with multiple factors, including race, lower educational attainment, lower annual household income, public health care insurance, and higher religiosity. CONCLUSIONS: Fifty-five percent of parents were willing to allow their youngest minor child to participate in a hypothetical biobank. Building trust, protecting privacy, and addressing attitudes may increase enrollment and diversity in pediatric biobanks.