Mapping parents' journey following prenatal diagnosis of CHD: a qualitative study.

OBJECTIVE: To better understand parents' accounts of their prenatal and postnatal experience after prenatal diagnosis of CHD - particularly emotional processing and coping mechanisms - to identify strategies to improve support. METHODS: This single-centre, longitudinal qualitative study included pregnant mothers and their support persons seen in Fetal Cardiology Clinic at Vanderbilt Children's Hospital from May through August 2019 for probable complex CHD. Twenty-seven individuals from 17 families participated in 62 phone interviews during pregnancy and postpartum: 27 conducted after the initial prenatal cardiology consultation, 15 after a follow-up prenatal visit, and 20 after birth. Applied thematic analysis approach was used to code and analyse transcribed interviews. Coding and codebook revisions occurred iteratively; intercoder reliability was >80%. RESULTS: Patients included mothers (16 [59%]), fathers (8 [30%]), and other support persons (3 [11%]). Initial fetal diagnoses included a range of moderate to severe CHD. Prenatally, parents sought to maintain hope while understanding the diagnosis; planning for the future rather than focusing on day-to-day was more common if prognoses were better. Postnatally, with confirmation of prenatal diagnoses, parents' sense of control expanded, and they desired more active engagement in clinical decision making. CONCLUSIONS: To enhance effective communication and support, understanding how parents conceptualise hope in relation to diagnosis and how that may evolve over time is critical. Expectant parents whose child has a significant risk of mortality may demonstrate hope by focusing on positivity. As prognostic uncertainty diminishes postpartum, the parental role on the team may shift, requiring clinicians to provide different support.

Patient Perceptions of Resident Involvement in Surgery: A Qualitative Study Using Surgical Video.

OBJECTIVE: To improve patient-centered perioperative informed consent, this study used real surgical footage to identify key topics which might be discussed with patients regarding resident involvement in surgery. DESIGN: We conducted semi-structured qualitative interviews with 27 participants. The interviews included a video showing an attending and resident performing a procedure together. Questions focused on comfort with resident involvement and preferences regarding preoperative informed consent. Participants also described residents' participation in their own words. SETTING: Participants were recruited from the infusion room of the allergy clinic and the treatment room of the dialysis clinic at a tertiary care facility in Nashville, TN (Vanderbilt University Medical Center). PARTICIPANTS: Adult medical patients were recruited via periodic verbal announcements by the interviewer in the waiting rooms. Purposive sampling was used to increase demographic diversity. Participants with training in the clinical health professions (i.e., physicians, nurses, and medical assistants) were excluded. RESULTS: Before watching the video, roughly half of participants imagined the resident to have a passive, bystander role, while the remaining imagined a more active role. Despite these differences, most participants found the video of attending-resident teamwork to be a reassuring depiction of resident involvement. When asked the best way to describe resident participation depicted in the video to patients, participants emphasized the need to focus on attending supervision, teamwork, reassurance, as well as resident presence, specific activities, and experience. CONCLUSION: Although patients have varying perceptions regarding the role of trainees in surgery, most participants were comfortable with teamwork between an attending and resident, as depicted in the video. Our participants provided multiple practical ways to transparently articulate resident involvement for testing in future research.

Direct-to-consumer genetic testing: Prospective users' attitudes toward information about ancestry and biological relationships.

Direct-to-consumer genetic testing is marketed as a tool to uncover ancestry and kin. Recent studies of actual and potential users have demonstrated that individuals' responses to the use of these tests for these purposes are complex, with privacy, disruptive consequences, potential for misuse, and secondary use by law enforcement cited as potential concerns. We conducted six focus groups with a diverse sample of participants (n = 62) who were aware of but had not used direct-to-consumer genetic tests, in an effort to understand more about what people considering these tests think about the potential value, risks, and benefits of such testing, taking into account use by third parties, such as potential kin and law enforcement. Participants differed widely in the perceived value of direct-to-consumer genetic tests for ancestry and kinship information for their own lives, including the desirability of contact with previously unknown relatives. Some perceived ancestry testing as mere curiosity or entertainment, while others, particularly those who had gaps in their family history, few living relatives, or who were adopted, saw greater value. Concerns about intrusion into one's life by purported kin and control of data were widespread, with many participants expressing concern about secondary uses of data that could harm users or their families. The use of direct-to-consumer genetic tests data for forensic genealogy elicited a particularly wide array of reactions, both spontaneously and in response to specific discussion prompts, mirroring the current public debate about law enforcement access to such data. The themes uncovered through our investigation warrant specific attention in the continued development of the science, policy, and practice of commercial direct-to-consumer genetic testing.

Surgeon Perspectives on Benefits and Downsides of Overlapping Surgery: In-depth, Qualitative Interviews.

OBJECTIVE: The aim of the study was to characterize surgeon perspectives regarding the benefits and downsides of conducting overlapping surgery. BACKGROUND: Although surgeons are key stakeholders in current discussions surrounding overlapping surgery, little has been published regarding their opinions on the practice. Further characterization of surgeon perspectives is needed to guide future studies and policy development regarding overlapping surgery. METHODS: Study information was sent to all members of 3 professional surgical societies. Interested individuals were eligible to participate if they identified as attending surgeons in an academic setting who work with trainees. Purposive selection was used to diversify surgeons interviewed across multiple dimensions, including subspecialty and opinion regarding appropriateness of overlapping surgery. In-depth, qualitative interviews were conducted with participants regarding their opinions on overlapping surgery. RESULTS: The 51 surgeons interviewed identified a wide array of potential benefits and disadvantages of overlapping surgery, some of which have not previously been measured, including downsides to surgeon wellness and patient experience, less surgeon control over procedures, and difficulty in scheduling cases. Interviewees often disagreed as to whether overlapping surgery negatively or positively affects each dimension discussed, particularly regarding the impact on resident training. CONCLUSIONS: The utilization of the novel perspectives presented here will allow for targeted assessment of physician perspectives in future quantitative studies and increase the likelihood that variables measured encompass the range of factors that surgeons find meaningful and relevant. Priority areas of future research should include examining effects of overlapping surgery on surgical training and surgeon wellness.

Thought Leader Comparisons of Risks in Precision Medicine Research.

Biomedical research is increasingly capitalizing on an array of data to illuminate the interplay between "omics," lifestyle, and health. Leveraging this information presents opportunities to advance knowledge but also poses risks to research participants. In interviews with thought leaders, we asked which data type associated with a hypothetical precision medicine research endeavor was riskiest: 42% chose ongoing access to electronic health records, 17% chose genomic analyses of biospecimens, and 15% chose streaming data from mobile devices. Other responses included "It depends" (15%), the three types are equally risky (8%), and the combination of data types together is riskiest (3%). When asked to consider the hypothetical study overall, 60% rated the likelihood of the risks materializing as low, but 20% rated the potential consequences as severe. These results have implications for study design and informed consent, including placing appropriate emphasis on the risks and protections for the full range of data.

Developing model biobanking consent language: what matters to prospective participants?

BACKGROUND: Efforts to improve informed consent have led to calls for providing information a reasonable person would want to have, in a way that facilitates understanding of the reasons why one might or might not want to participate. At the same time, advances in large-scale genomic research have expanded both the opportunities and the risks for participants, families, and communities. To advance the use of effective consent materials that reflect this landscape, we used empirical data to develop model consent language, as well as brief questions to assist people in thinking about their own values relative to participation. METHODS: We conducted in-person interviews to gather preliminary input on these materials from a diverse sample (n = 32) of the general population in Nashville, Tennessee. We asked them to highlight information they found especially reassuring or concerning, their hypothetical willingness to participate, and their opinions about the values questions. RESULTS: Consent information most often highlighted as reassuring included the purpose of the biobank, the existence and composition of a multidisciplinary oversight committee, the importance of participants' privacy and efforts to protect it, and controlled access to a scientific database. Information most often highlighted as concerning included the deposition of data in a publicly accessible database, the risk of unintended access to data, the potential for non-research use of data, and use of medical record information in general. Seventy-five percent of participants indicated initial willingness to participate in the hypothetical biobank; this decreased to 66% as participants more closely considered the information over the course of the interview. A large majority rated the values questions as helpful. CONCLUSIONS: These results are consistent with other research on public perspectives on biobanking and genomic cohort studies, suggesting that our model language effectively captures commonly expressed reasons for and against participation. Our study enriches this literature by connecting specific consent form disclosures with qualitative data regarding what participants found especially reassuring or concerning and why. Interventions that facilitate individuals' closer engagement with consent information may result in participation decisions more closely aligned with their values.

Uncertainty of Prenatally Diagnosed Congenital Heart Disease: A Qualitative Study.

Importance: Parents who receive a prenatal diagnosis of congenital heart disease may experience more short- and long-term stress than those who receive a postnatal diagnosis. To identify potential interventions to ameliorate that stress, the longitudinal emotional experience of parents must first be understood. Objective: To better understand parents' accounts of their own prenatal experience, particularly aspects they found to be stressful or challenging, and to identify strategies to improve support. Design, Setting, and Participants: This qualitative study included analysis of transcripts of audio recorded telephone interviews with pregnant mothers and their support persons, as applicable, who were referred to and seen at the Fetal Cardiology Clinic at Vanderbilt Children's Hospital from May 2019 to August 2019 with an initial likely diagnosis of complex congenital heart disease at any gestational age. Data analysis was conducted from August 2019 to November 2019. Main Outcomes and Measures: An applied thematic analysis approach was used to code and analyze professionally transcribed interviews. Coding and codebook revisions occurred iteratively; intercoder reliability was assessed and confirmed routinely. One author coded all transcripts; a second author independently reviewed one-fifth of the transcripts at fixed intervals to ensure that interrater reliability remained greater than 80%. Results: Twenty-seven individuals from 17 families participated in 42 phone interviews during pregnancy, 27 conducted at the first time point after the initial prenatal cardiology consultation and 15 at the second time point after a follow-up prenatal cardiology visit. Most interviewees were mothers (16 interviewees [59%]; median [interquartile range] age, 30.0 [27.3-34.8] years) or fathers (8 interviewees [30%)], with a few support individuals (3 interviewees [11%]) (median [interquartile range] age of family member or support individual, 30.0 [26.0-42.0] years). Initial fetal diagnoses included a range of severe congenital heart disease. Uncertainty was identified as a pervasive central theme and was related both to concrete questions on scheduling, logistics, or next steps, and long-term unknown variables concerning the definitiveness of the diagnosis or overall prognosis. Practitioners helped families through their framing of uncertainty at various time points including before, during, and after the clinic visit. Conclusions and Relevance: Families walk an uncertain path following a fetal diagnosis of severe congenital heart disease. The challenges faced by the cardiologists caring for them overlap in many ways with those experienced by pediatric palliative care practitioners. Potential future interventions to improve parental support were identified in the areas of expectation setting before the referral visit, communication in clinic, and identity formation after the new diagnosis.

Research Use of Electronic Health Records: Patients' Views on Alternative Approaches to Permission.

Background: The increased use of electronic health records (EHRs) has resulted in new opportunities for research, but also raises concerns regarding privacy, confidentiality, and patient awareness. Because public trust is essential to the success of the research enterprise, patient perspectives are essential to the development and implementation of ethical approaches to the research use of EHRs. Yet, little is known about patients' views and expectations regarding various approaches to seeking permission for research use of their EHR data. Methods: We conducted semi-structured interviews with 120 patients in four counties in diverse regions of the southeastern United States: Appalachia, the Mississippi Delta, and the Piedmont area of North Carolina. We asked participants to consider, from multiple stakeholder perspectives, the advantages and disadvantages of three approaches to notifying patients of, or obtaining permission for, research use of their EHR data; whether they believed it would be acceptable if their healthcare organization used each approach; and which approach would be most appropriate. Results: Nearly all participants said General Notification, Broad Permission, and Categorical Permission would each be acceptable approaches to notification of, or permission for, EHR research. Over half identified Broad Permission as the most appropriate approach. Across all of these discussions, major themes included the importance of clarity, simplicity, and usability of patient-facing materials, as well as the level of transparency, trustworthiness, and respect for patients the approach conveys. Conclusions: Our findings help to inform the development and implementation of ethical approaches to the research use of EHRs by identifying key patient considerations regarding various approaches to permission and suggesting potential actions for healthcare organizations and researchers.

Unregulated Health Research Using Mobile Devices: Ethical Considerations and Policy Recommendations.

Mobile devices with health apps, direct-to-consumer genetic testing, crowd-sourced information, and other data sources have enabled research by new classes of researchers. Independent researchers, citizen scientists, patient-directed researchers, self-experimenters, and others are not covered by federal research regulations because they are not recipients of federal financial assistance or conducting research in anticipation of a submission to the FDA for approval of a new drug or medical device. This article addresses the difficult policy challenge of promoting the welfare and interests of research participants, as well as the public, in the absence of regulatory requirements and without discouraging independent, innovative scientific inquiry. The article recommends a series of measures, including education, consultation, transparency, self-governance, and regulation to strike the appropriate balance.

Expert Perspectives on Oversight for Unregulated mHealth Research: Empirical Data and Commentary.

In qualitative interviews with a diverse group of experts, the vast majority believed unregulated researchers should seek out independent oversight. Reasons included the need for objectivity, protecting app users from research risks, and consistency in standards for the ethical conduct of research. Concerns included burdening minimal risk research and limitations in current systems of oversight. Literature and analysis supports the use of IRBs even when not required by regulations, and the need for evidence-based improvements in IRB processes.

Ethical Considerations in the Conduct of Unregulated mHealth Research: Expert Perspectives.

To assist in resolving ethical questions surrounding unregulated mHealth research, we conducted in-depth qualitative interviews with experts from four key stakeholder groups: patient/research advocates, researchers, regulatory professionals, and mobile app/device developers. They discussed challenges and potential solutions in the context of two hypothetical scenarios involving unregulated mHealth research, including notifications/permissions for research use of mHealth data, data access procedures, new primary data collection, offering individual research results, and data sharing and dissemination.

Protecting Participants in Genomic Research: Understanding the "Web of Protections" Afforded by Federal and State Law.

Researchers now commonly collect biospecimens for genomic analysis together with information from mobile devices and electronic health records. This rich combination of data creates new opportunities for understanding and addressing important health issues, but also intensifies challenges to privacy and confidentiality. Here, we elucidate the "web" of legal protections for precision medicine research by integrating findings from qualitative interviews with structured legal research and applying them to realistic research scenarios involving various privacy threats.

Trust as a Predictor of Patient Perceptions Regarding Overlapping Surgery and Trainee Independence.

OBJECTIVES: To examine opinions on trainee independence and attending presence among a cross-section of the general population and explore how perceptions of trust, past experiences, and demographics interacted with comfort consenting to these surgical scenarios. STUDY DESIGN: Mixed-methods METHODS: Based on prior qualitative analysis, we designed a survey of patient preferences and values that focused on trust in healthcare practitioners and processes, which also included comfort ratings of three surgical scenarios (including overlapping surgery). The survey was administered to a sample from the general public using Mechanical Turk. We identified discreet domains of trust and examined the association of responses to these domains with comfort ratings, prior healthcare experiences, and demographics. RESULTS: We analyzed 225 surveys and identified four patient subgroups based on responses to the surgical scenarios. Subjects that were more comfortable with overlapping surgery were more trusting of trainees and delegation by the attending. Past experiences in healthcare (positive and negative) were associated with multiple domains of trust (in trainees, surgeons, and the healthcare system). Demographics were not predictive of trust responses or comfort ratings. CONCLUSION: Patients express varying degrees of comfort with overlapping surgery, and this is not associated with demographics. Past negative experiences have an impact on trust in the healthcare system overall, and trust in trainees specifically predicts comfort with attending absence from the operating room. Efforts to increase patient comfort with overlapping surgery and surgical training should include strategies to address past negative experiences and foster trust in trainees and the delegation process. LEVEL OF EVIDENCE: IV Laryngoscope, 130:2728-2735, 2020.

EHR phenotyping for research recruitment: Researcher, IRB, and physician perspectives on approaches to contacting patients.

INTRODUCTION: Failure to achieve accrual goals is a common problem in health-related research. Electronic health records represent a promising resource, offering the ability to identify a precisely defined cohort of patients who meet inclusion/exclusion criteria. However, challenges associated with the recruitment process remain and institutional policies vary. METHODS: We interviewed researchers, institutional review board chairs, and primary care physicians in North Carolina and Tennessee. Questions focused on strategies for initiating contact with potentially eligible patients, as well as recruitment letters asking recipients to opt in versus opt out of further communication. RESULTS: When we asked about initiating contact with prospective participants, qualitative themes included trust, credibility, and established relationships; research efficiency and validity; privacy and autonomy; the intersection between research and clinical care; and disruption to physician-researcher and physician-patient relationships. All interviewees said it was acceptable for researchers to contact patients through their physicians; most said it was acceptable for researchers to contact patients directly. Over half chose contact through physicians as more appropriate. Regarding recruitment letters, qualitative themes included the quality of the participant pool; privacy and control; research efficiency and representativeness; and patients' opportunity to make their own decisions. All interviewees said asking recipients to opt in to further communication was acceptable; nearly all said opt out was acceptable. Similar proportions chose each approach as more appropriate. CONCLUSIONS: Comparing these results to our previous research with patients reveals potential differences in stakeholder perspectives. We offer suggestions for developing balanced approaches that respect patients and facilitate the advancement of science.

Patient values regarding overlapping surgery: Identification of distinct patient subgroups.

OBJECTIVES/HYPOTHESIS: To explore patient values associated with their comfort level with surgical trainees and attending surgeon absence from the operating room. STUDY DESIGN: Qualitative interviews with general medical patients. METHODS: We analyzed data from qualitative interviews with patients that included a quantitative rating on a visual analog scale (VAS) of comfort consenting to three surgical scenarios, including overlapping surgery, to identify subgroups of patients based on comfort level. After identifying subgroups, we compared qualitative responses from participants who were generally comfortable with overlapping surgery to those who were uncomfortable to identify themes associated with these perceptions. RESULTS: We identified three subgroups of patients based on the patterns of VAS responses. Participants who were comfortable with overlapping surgery expressed trust in the surgeon and delegation process. Those who were most uncomfortable expressed a strong desire to know who was operating on them, and a desire for control over their surgical process. Subjects uncomfortable with overlapping surgery were also generally not sensitive to tradeoffs (cost, timing). CONCLUSIONS: We identified distinct subgroups of patients based on their comfort level with trainee independence and primary attending availability. By examining the predominant values in these subgroups, we identified potential explanations for patient discomfort with attending absence. Strategies to enhance patients' knowledge about the process of surgery and a sense of control over their own care may improve comfort with trainee participation and overlapping surgery. LEVEL OF EVIDENCE: 6 Laryngoscope, 2019.

Improving Translation and Cultural Appropriateness of Spanish-Language Consent Materials for Biobanks.

A growing proportion of prospective research participants in the United States speak limited or no English. We conducted cognitive interviews with native Spanish speakers to test Spanish-language translations of simplified and traditional biobank consent forms. Comprehension was generally high and did not differ by form. Most of those who received the simplified form felt it contained the right amount of information, compared with fewer than half of those who received the traditional form. Qualitative results allowed us to identify overarching issues related to tone, formality, and voice that may affect prospective participants' trust and willingness to participate. Certain characteristics of written Spanish are seemingly at odds with recommended actions to simplify consent forms; thus, even when significant empirical effort has been expended to develop simplified consent materials in English, additional work is needed to ensure the accuracy, comprehensibility, and cultural-congruence of Spanish-language translations.

Thought Leader Perspectives on Participant Protections in Precision Medicine Research.

Precision medicine research is rapidly taking a lead role in the pursuit of new ways to improve health and prevent disease, but also presents new challenges for protecting human subjects. The extent to which the current "web" of legal protections, including technical data security measures, as well as measures to restrict access or prevent misuse of research data, will protect participants in this context remains largely unknown. Understanding the strength, usefulness, and limitations of this constellation of laws, regulations, and procedures is critical to ensuring not only that participants are protected, but also that their participation decisions are accurately informed. To address these gaps, we conducted in-depth interviews with a diverse group of 60 thought-leaders to explore their perspectives on the protections associated with precision medicine research.

Patient perspectives on use of electronic health records for research recruitment.

BACKGROUND: EHR phenotyping offers the ability to rapidly assemble a precisely defined cohort of patients prescreened for eligibility to participate in health-related research. Even so, stakeholders in the process must still contend with the practical and ethical challenges associated with research recruitment. Patient perspectives on these matters are particularly important given that the success of research recruitment depends on patients' willingness to participate. METHODS: We conducted 15 focus groups (n = 110 participants) in four counties in diverse regions of the southeastern US: Appalachia, the Mississippi Delta, and the Piedmont area of North Carolina. Based on a hypothetical study of a behavioral intervention for type 2 diabetes, we asked about the acceptability and appropriateness of direct investigator versus physician-mediated contact with patients for research recruitment, and whether patients should be asked to opt in or opt out of further contact in response to recruitment letters. RESULTS: For initial contact, nearly all participants said it would be acceptable for researchers to contact patients directly and three-fourths said that it would be acceptable for researchers to contact patients through their physicians. When we asked which would be most appropriate, a substantial majority chose direct contact. Themes that arose in the discussion included trust and transparency, decision-making power, the effect on research, and the effect on patient care. For response expectations, the vast majority of participants said both opt-in and opt-out would be acceptable-typically finding neither especially problematic and noting that both afford patients the opportunity to make their own decisions. CONCLUSIONS: External validity relies heavily on researchers' success enrolling eligible patients and failure to reach accrual targets is a costly and common barrier to advancing scientific knowledge. Our results suggest that patients recognize multiple advantages and disadvantages of different research recruitment strategies and place value on the implications not just for themselves, but also for researchers and healthcare providers. Our findings, including rich qualitative detail, contribute to the body of empirical and ethical literature on improving research recruitment and suggest specific ways forward as well as important areas for future research.

Thought leader perspectives on benefits and harms in precision medicine research.

Precision medicine research is underway to identify targeted approaches to improving health and preventing disease. However, such endeavors raise significant privacy and confidentiality concerns. The objective of this study was to elucidate the potential benefits and harms associated with precision medicine research through in-depth interviews with a diverse group of thought leaders, including primarily U.S.-based experts and scholars in the areas of ethics, genome research, health law, historically-disadvantaged populations, informatics, and participant-centric perspectives, as well as government officials and human subjects protections leaders. The results suggest the prospect of an array of individual and societal benefits, as well as physical, dignitary, group, economic, psychological, and legal harms. Relative to the way risks and harms are commonly described in consent forms for precision medicine research, the thought leaders we interviewed arguably emphasized a somewhat different set of issues. The return of individual research results, harm to socially-identifiable groups, the value-dependent nature of many benefits and harms, and the risks to the research enterprise itself emerged as important cross-cutting themes. Our findings highlight specific challenges that warrant concentrated care during the design, conduct, dissemination, and translation of precision medicine research and in the development of consent materials and processes.