Meghan Halley
Assistant Professor (Research) of Pediatrics (Stanford Center for Biomedical Ethics)
Pediatrics - Center for Biomedical Ethics
Bio
Meghan Halley, PhD, MPH, (she/hers) is an Assistant Professor at the Stanford Center for Biomedical Ethics. A medical anthropoloigst by training, Dr. Halley's research focuses on ethical challenges in research and clinical care for patients with rare and undiagnosed genetic conditions.
Professional Education
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PhD, Case Western Reserve University, Anthropology (2012)
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MPH, Case Western Reserve University, Public Health (2009)
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BA, University of Wisconsin-Madison, Anthropology (2005)
Service, Volunteer and Community Work
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Co-Chair, Patient Education and Empowerment Resource, The Undiagnosed Diseases Network (January 1, 2021 - 12/31/2022)
Location
Cambridge, MA
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Member, Board of Directors, Undiagnosed Diseases Network Foundation (11/1/2021 - Present)
Location
Washington, D.C.
Contact
https://orcid.org/0000-0002-5031-9840All Publications
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Increasing Diversity, Equity, Inclusion, and Accessibility in Rare Disease Clinical Trials.
Pharmaceutical medicine
2024
Abstract
Diversity, equity, inclusion, and accessibility (DEIA) are foundational principles for clinical trials and medical research. In rare diseases clinical research, where numbers of participants are already challenged by rarity itself, maximizing inclusion is of particular importance to clinical trial success, as well as ensuring the generalizability and relevance of the trial results to the people affected by these diseases. In this article, we review the medical and gray literature and cite case examples to provide insights into how DEIA can be proactively integrated into rare diseases clinical research. Here, we particularly focus on genetic diversity. While the rare diseases DEIA literature is nascent, it is accelerating as many patient advocacy groups, professional societies, training and educational organizations, researcher groups, and funders are setting intentional strategies to attain DEIA goals moving forward, and to establish metrics to ensure continued improvement. Successful examples in underserved and underrepresented populations are available that can serve as case studies upon which rare diseases clinical research programs can be built. Rare diseases have historically been innovation drivers in basic, translational, and clinical research, and ultimately, all populations benefit from data diversity in rare diseases populations that deliver novel insights and approaches to how clinical research can be performed.
View details for DOI 10.1007/s40290-024-00529-8
View details for PubMedID 38977611
View details for PubMedCentralID 7587233
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The Undiagnosed Diseases Network: Characteristics of solvable applicants and diagnostic suggestions for non-accepted ones.
Genetics in medicine : official journal of the American College of Medical Genetics
2024: 101203
Abstract
Can certain characteristics identify as solvable some undiagnosed patients who seek extensive evaluation and thorough record review, like by the Undiagnosed Diseases Network (UDN)?The UDN is a national research resource to solve medical mysteries through team science. Applicants provide informed consent to access to their medical records. After review, expert panels assess if applicants meet inclusion and exclusion criteria to select participants. When not accepting applicants, UDN experts may offer suggestions for diagnostic efforts. Using minimal information from initial applications, we compare features in applicants not accepted with those accepted and either solved or still not solved by the UDN. The diagnostic suggestions offered to non-accepted applicants and their clinicians were tallied.Non-accepted applicants were more often female, older at first symptoms and application, and longer in review than accepted applicants. The accepted and successfully diagnosed applicants were younger in ages, shorter in review time, more often non-white, of Hispanic ethnicity, and presenting with nervous system features. Half of non-accepted applicants were given suggestions for further local diagnostic evaluation. A few seemed to have two major diagnoses or a provocative environmental exposure history.Comprehensive UDN record review generates possibly helpful advice.
View details for DOI 10.1016/j.gim.2024.101203
View details for PubMedID 38967101
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Career flexibility for PAs: What makes switching specialties successful?
JAAPA : official journal of the American Academy of Physician Assistants
2024
Abstract
ABSTRACT: Switching specialties is common among physician associates/assistants (PAs) and important in combating burnout. Despite this, little is known about the PA experience with switching specialties. This study sought to identify factors associated with successfully switching specialties using semistructured interviews with PAs and hiring managers. Participants reported that switching specialties was initially challenging for managers and PAs because of insufficient onboarding and unrealistic expectations, but they also reported that they were generally satisfied with long-term outcomes. Our findings suggest that PAs hoping to switch specialties may want to focus on building a professional network, identifying areas where they can highlight relevant experience, and demonstrating their intent to remain in that role. Hiring managers could benefit from considering their hiring practices and tailoring onboarding expectations for new hires.
View details for DOI 10.1097/01.JAA.0000000000000004
View details for PubMedID 38595169
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Moral Engagement and Disengagement in Health Care AI Development.
AJOB empirical bioethics
2024: 1-10
Abstract
Machine learning (ML) is utilized increasingly in health care, and can pose harms to patients, clinicians, health systems, and the public. In response, regulators have proposed an approach that would shift more responsibility to ML developers for mitigating potential harms. To be effective, this approach requires ML developers to recognize, accept, and act on responsibility for mitigating harms. However, little is known regarding the perspectives of developers themselves regarding their obligations to mitigate harms.We conducted 40 semi-structured interviews with developers of ML predictive analytics applications for health care in the United States.Participants varied widely in their perspectives on personal responsibility and included examples of both moral engagement and disengagement, albeit in a variety of forms. While most (70%) of participants made a statement indicative of moral engagement, most of these statements reflected an awareness of moral issues, while only a subset of these included additional elements of engagement such as recognizing responsibility, alignment with personal values, addressing conflicts of interests, and opportunities for action. Further, we identified eight distinct categories of moral disengagement reflecting efforts to minimize potential harms or deflect personal responsibility for preventing or mitigating harms.These findings suggest possible facilitators and barriers to the development of ethical ML that could act by encouraging moral engagement or discouraging moral disengagement. Regulatory approaches that depend on the ability of ML developers to recognize, accept, and act on responsibility for mitigating harms might have limited success without education and guidance for ML developers about the extent of their responsibilities and how to implement them.
View details for DOI 10.1080/23294515.2024.2336906
View details for PubMedID 38588388
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Practical Approaches to Enhancing Fairness, Social Responsibility and the Inclusion of Diverse Viewpoints in Biomedicine.
Pacific Symposium on Biocomputing. Pacific Symposium on Biocomputing
2024; 29: 645-649
Abstract
The following sections are included:Workshop DescriptionLearning ObjectivesPresenter InformationAbout the Workshop OrganizersPresentationsSpeaker Presentations.
View details for PubMedID 38160313
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Not in my AI: Moral engagement and disengagement in health care AI development.
Pacific Symposium on Biocomputing. Pacific Symposium on Biocomputing
2023; 28: 496-506
Abstract
Machine learning predictive analytics (MLPA) are utilized increasingly in health care, but can pose harms to patients, clinicians, health systems, and the public. The dynamic nature of this technology creates unique challenges to evaluating safety and efficacy and minimizing harms. In response, regulators have proposed an approach that would shift more responsibility to MLPA developers for mitigating potential harms. To be effective, this approach requires MLPA developers to recognize, accept, and act on responsibility for mitigating harms. In interviews of 40 MLPA developers of health care applications in the United States, we found that a subset of ML developers made statements reflecting moral disengagement, representing several different potential rationales that could create distance between personal accountability and harms. However, we also found a different subset of ML developers who expressed recognition of their role in creating potential hazards, the moral weight of their design decisions, and a sense of responsibility for mitigating harms. We also found evidence of moral conflict and uncertainty about responsibility for averting harms as an individual developer working in a company. These findings suggest possible facilitators and barriers to the development of ethical ML that could act through encouragement of moral engagement or discouragement of moral disengagement. Regulatory approaches that depend on the ability of ML developers to recognize, accept, and act on responsibility for mitigating harms might have limited success without education and guidance for ML developers about the extent of their responsibilities and how to implement them.
View details for PubMedID 36541003
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Developer Perspectives on Potential Harms of Machine Learning Predictive Analytics in Health Care: Qualitative Analysis.
Journal of medical Internet research
2023; 25: e47609
Abstract
Machine learning predictive analytics (MLPA) is increasingly used in health care to reduce costs and improve efficacy; it also has the potential to harm patients and trust in health care. Academic and regulatory leaders have proposed a variety of principles and guidelines to address the challenges of evaluating the safety of machine learning-based software in the health care context, but accepted practices do not yet exist. However, there appears to be a shift toward process-based regulatory paradigms that rely heavily on self-regulation. At the same time, little research has examined the perspectives about the harms of MLPA developers themselves, whose role will be essential in overcoming the "principles-to-practice" gap.The objective of this study was to understand how MLPA developers of health care products perceived the potential harms of those products and their responses to recognized harms.We interviewed 40 individuals who were developing MLPA tools for health care at 15 US-based organizations, including data scientists, software engineers, and those with mid- and high-level management roles. These 15 organizations were selected to represent a range of organizational types and sizes from the 106 that we previously identified. We asked developers about their perspectives on the potential harms of their work, factors that influence these harms, and their role in mitigation. We used standard qualitative analysis of transcribed interviews to identify themes in the data.We found that MLPA developers recognized a range of potential harms of MLPA to individuals, social groups, and the health care system, such as issues of privacy, bias, and system disruption. They also identified drivers of these harms related to the characteristics of machine learning and specific to the health care and commercial contexts in which the products are developed. MLPA developers also described strategies to respond to these drivers and potentially mitigate the harms. Opportunities included balancing algorithm performance goals with potential harms, emphasizing iterative integration of health care expertise, and fostering shared company values. However, their recognition of their own responsibility to address potential harms varied widely.Even though MLPA developers recognized that their products can harm patients, public, and even health systems, robust procedures to assess the potential for harms and the need for mitigation do not exist. Our findings suggest that, to the extent that new oversight paradigms rely on self-regulation, they will face serious challenges if harms are driven by features that developers consider inescapable in health care and business environments. Furthermore, effective self-regulation will require MLPA developers to accept responsibility for safety and efficacy and know how to act accordingly. Our results suggest that, at the very least, substantial education will be necessary to fill the "principles-to-practice" gap.
View details for DOI 10.2196/47609
View details for PubMedID 37971798
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Genomics Research with Undiagnosed Children: Ethical Challenges at the Boundaries of Research and Clinical Care
JOURNAL OF PEDIATRICS
2023; 261
View details for DOI 10.1018/j.jpeds.2023.113537
View details for Web of Science ID 001029333600001
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Blurred Boundaries: Toward an Expanded Ethics of Research and Clinical Care.
The American journal of bioethics : AJOB
2023; 23 (8): 5-9
View details for DOI 10.1080/15265161.2023.2224148
View details for PubMedID 38410998
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Rare Disease, Advocacy and Justice: Intersecting Disparities in Research and Clinical Care.
The American journal of bioethics : AJOB
2023: 1-10
Abstract
Rare genetic diseases collectively impact millions of individuals in the United States. These patients and their families share many challenges including delayed diagnosis, lack of knowledgeable providers, and limited economic incentives to develop new therapies for small patient groups. As such, rare disease patients and families often must rely on advocacy, including both self-advocacy to access clinical care and public advocacy to advance research. However, these demands raise serious concerns for equity, as both care and research for a given disease can depend on the education, financial resources, and social capital available to the patients in a given community. In this article, we utilize three case examples to illustrate ethical challenges at the intersection of rare diseases, advocacy and justice, including how reliance on advocacy in rare disease may drive unintended consequences for equity. We conclude with a discussion of opportunities for diverse stakeholders to begin to address these challenges.
View details for DOI 10.1080/15265161.2023.2207500
View details for PubMedID 37204146
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Demographic Characteristics Associated With Perceptions of Personal Utility in Genetic and Genomic Testing: A Systematic Review.
JAMA network open
2023; 6 (5): e2310367
Abstract
The expansion of genetic and genomic testing in health care has led to recognition that these tests provide personal as well as clinical utility to patients and families. However, available systematic reviews on this topic have not reported the demographic backgrounds of participants in studies of personal utility, leaving generalizability unclear.To determine the demographic characteristics of participants in studies examining the personal utility of genetic and genomic testing in health care.For this systematic review, we utilized and updated the results of a highly cited 2017 systematic review on the personal utility of genetics and genomics, which identified relevant articles published between January 1, 2003, and August 4, 2016. We also used the original methods to update this bibliography with literature published subsequently up to January 1, 2022. Studies were screened for eligibility by 2 independent reviewers. Eligible studies reported empirical data on the perspectives of patients, family members, and/or the general public in the US on the personal utility of any type of health-related genetic or genomic test. We utilized a standardized codebook to extract study and participant characteristics. We summarized demographic characteristics descriptively across all studies and by subgroup based on study and participant characteristics.We included 52 studies with 13 251 eligible participants. Sex or gender was the most frequently reported demographic characteristic (48 studies [92.3%]), followed by race and ethnicity (40 studies [76.9%]), education (38 studies [73.1%]), and income (26 studies [50.0%]). Across studies, participants disproportionately were women or female (mean [SD], 70.8% [20.5%]), were White (mean [SD], 76.1% [22.0%]), had a college degree or higher (mean [SD], 64.5% [19.9%]), and reported income above the US median (mean [SD], 67.4% [19.2%]). Examination of subgroups of results by study and participant characteristics evidenced only small shifts in demographic characteristics.This systematic review examined the demographic characteristics of individual participants in studies of the personal utility of health-related genetic and genomic testing in the US. The results suggest that participants in these studies were disproportionately White, college-educated women with above-average income. Understanding the perspectives of more diverse individuals regarding the personal utility of genetic and genomic testing may inform barriers to research recruitment and uptake of clinical testing in currently underrepresented populations.
View details for DOI 10.1001/jamanetworkopen.2023.10367
View details for PubMedID 37145601
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Participation in a national diagnostic research study: assessing the patient experience.
Orphanet journal of rare diseases
2023; 18 (1): 73
Abstract
INTRODUCTION: The Undiagnosed Diseases Network (UDN), a clinical research study funded by the National Institutes of Health, aims to provide answers for patients with undiagnosed conditions and generate knowledge about underlying disease mechanisms. UDN evaluations involve collaboration between clinicians and researchers and go beyond what is possible in clinical settings. While medical and research outcomes of UDN evaluations have been explored, this is the first formal assessment of the patient and caregiver experience.METHODS: We invited UDN participants and caregivers to participate in focus groups via email, newsletter, and a private participant Facebook group. We developed focus group questions based on research team expertise, literature focused on patients with rare and undiagnosed conditions, and UDN participant and family member feedback. In March 2021, we conducted, recorded, and transcribed four 60-min focus groups via Zoom. Transcripts were evaluated using a thematic analysis approach.RESULTS: The adult undiagnosed focus group described the UDN evaluation as validating and an avenue for access to medical providers. They also noted that the experience impacted professional choices and helped them rely on others for support. The adult diagnosed focus group described the healthcare system as not set up for rare disease. In the pediatric undiagnosed focus group, caregivers discussed a continued desire for information and gratitude for the UDN evaluation. They also described an ability to rule out information and coming to terms with not having answers. The pediatric diagnosed focus group discussed how the experience helped them focus on management and improved communication. Across focus groups, adults (undiagnosed/diagnosed) noted the comprehensiveness of the evaluation. Undiagnosed focus groups (adult/pediatric) discussed a desire for ongoing communication and care with the UDN. Diagnosed focus groups (adult/pediatric) highlighted the importance of the diagnosis they received in the UDN. The majority of the focus groups noted a positive future orientation after participation.CONCLUSION: Our findings are consistent with prior literature focused on the patient experience of rare and undiagnosed conditions and highlight benefits from comprehensive evaluations, regardless of whether a diagnosis is obtained. Focus group themes also suggest areas for improvement and future research related to the diagnostic odyssey.
View details for DOI 10.1186/s13023-023-02695-5
View details for PubMedID 37032333
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Patient Perspectives of Health System Barriers to Accessing Care for Hidradenitis Suppurativa: A Qualitative Study.
JAMA dermatology
2023
Abstract
Patient-perceived barriers to hidradenitis suppurativa (HS) care are poorly understood. Understanding health care barriers is a critical first step toward improving care for this population.To characterize the health care experiences of people living with HS, including perceived barriers and facilitators to health care access, and to elucidate potential associations among these barriers and facilitators, health care access, and disease activity.In this qualitative study, an inductive thematic analysis was conducted on 45 in-depth, 60- to 90-minute semistructured interviews of 45 people with HS from diverse sociodemographic backgrounds that took place between March and April 2020. Individuals were eligible if they could speak English, were 18 years or older, and were diagnosed with HS. A diagnosis of HS was confirmed through physician diagnosis or through self-reported, affirmative response to the validated screening question, "Do you experience boils in your armpits or groin that recur at least every six months?"Interviews were audio recorded and transcribed verbatim. A modified grounded theory approach was used to develop the codebook, which investigators used for inductive thematic analysis.Among the 45 participants included, the median (IQR) age was 37 (16) years, 33 (73%) were female, and 22 (49%) were White. There were 6 interrelated themes associated with participant-perceived barriers to accessing HS care: (1) bidirectional associations of disease activity and employment, (2) association of employment with health care coverage, (3) association of health care coverage with costs and perceived access to care, (4) association of costs with access to patient-centered care, (5) health care professional attitudes and knowledge influence patient-centered care and perceived access to care and disease activity, and (6) health system characteristics influence patient-centered care and associated costs, perceived access to care, and disease activity.This qualitative study highlights themes that generate a conceptual model for understanding barriers that may act synergistically to limit health care access and influence disease activity. The disease activity of HS may be reduced when cycle elements are optimized. This study also highlights areas for future investigations and potential systems-level changes to improve access to patient-centered HS care.
View details for DOI 10.1001/jamadermatol.2023.0486
View details for PubMedID 37017984
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Formally comparing topic models and human-generated qualitative coding of physician mothers' experiences of workplace discrimination
BIG DATA & SOCIETY
2023; 10 (1)
View details for DOI 10.1177/20539517221149106
View details for Web of Science ID 001061745600001
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What is "Personal" About Personal Experience? A Call to Reflexivity for All.
The American journal of bioethics : AJOB
2023; 23 (1): 39-41
View details for DOI 10.1080/15265161.2022.2146794
View details for PubMedID 36595008
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Perspectives of Rare Disease Social Media Group Participants on Engaging With Genetic Counselors: Mixed Methods Study.
Journal of medical Internet research
2022; 24 (12): e42084
Abstract
Social media provides a potential avenue for genetic counselors to address gaps in access to reliable genetics information for rare disease communities. However, only limited research has examined patient and family attitudes toward engaging with genetic counselors through social media.Our study assessed the attitudes of members of rare disease social media groups toward engaging with genetic counselors through social media, characteristics associated with greater interest, and the benefits and potential pitfalls of various approaches to such engagement.We conducted a mixed methods survey of patients and family members recruited from a systematic sample of rare disease Facebook groups. Patient characteristics and their associations with interest in engagement with genetic counselors were evaluated using univariate and bivariate statistics. Responses to open-ended questions were analyzed using thematic content analysis.In total, 1053 individuals from 103 rare disease groups participated. The median overall interest in engaging with genetic counselors on social media was moderately high at 7.0 (IQR 4.0-9.0, range 0-10). No past experience with a genetic counselor was associated with greater interest in engaging with one through social media (µ=6.5 vs 6.0, P=.04). Participants expressed greatest interest (median 9.0, IQR 5.0-10.0) in engagement models allowing direct communication with genetic counselors, which was corroborated by the majority (n=399, 61.3%) of individuals who responded to open-ended questions explicitly stating their interest in 1-on-1 interactions. When asked what forms of support they would request from genetic counselors through social media, participants desired individualized support and information about how to access services. However, participants also expressed concerns regarding privacy and confidentiality.Patients and family members in rare disease social media groups appear interested in engaging with genetic counselors through social media, particularly for individualized support. This form of engagement on social media is not meant to replace the current structure and content of genetic counseling (GC) services, but genetic counselors could more actively use social media as a communication tool to address gaps in knowledge and awareness about genetics services and gaps in accessible patient information. Although encouraging, concerns regarding privacy and feasibility require further consideration, pointing to the need for professional guidelines in this area.
View details for DOI 10.2196/42084
View details for PubMedID 36542454
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Beyond "Ensuring Understanding": Toward a Patient-Partnered Neuroethics of Brain Device Research.
AJOB neuroscience
2022; 13 (4): 241-244
View details for DOI 10.1080/21507740.2022.2126550
View details for PubMedID 36272165
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Supporting undiagnosed participants when clinical genomics studies end.
Nature genetics
2022
View details for DOI 10.1038/s41588-022-01150-8
View details for PubMedID 35902745
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Impact of Sexual Harassment and Social Support on Burnout in Physician Mothers.
Journal of women's health (2002)
2022
Abstract
Background: Burnout affects >50% of physicians, especially women. This study aimed to examine how negative workplace interactions can predict burnout, and whether positive social interactions can mitigate risk. Materials and Methods: In a study of 1627 physician mothers who responded to a survey by the Physician Moms Group, an online Facebook group, we first examined the association between workplace sexual harassment and burnout. In an embedded experiment, we then measured the causal impact of priming perceived social support and connectedness on the three dimensions of employee burnout. Results: Two-thirds of respondents reported having experienced sexual harassment in the past year. Sexual harassment by patients was associated with 0.27 points higher emotional exhaustion, one dimension of burnout (95% confidence interval [CI] 0.12-0.41), equivalent to the predicted impact of an additional 22 weekly work hours on emotional exhaustion. Sexual harassment by patients was also associated with 0.40 points higher patient depersonalization, another dimension of burnout (95% CI 0.27-0.53). Sexual harassment by colleagues was associated with 0.16 points higher emotional exhaustion (95% CI 0.02-0.30), but not other dimensions of burnout. We found no significant relationship between experiences of sexual harassment and levels of personal accomplishment (the third dimension of burnout) among this sample. Priming physician mothers to reflect on their connectedness with other physician mothers significantly increased their sense of personal accomplishment. The priming intervention did not yield a significant effect on emotional exhaustion or depersonalization. Conclusions: Negative and positive social interactions each affect different dimensions of burnout. Sexual harassment-a pervasive type of negative social interaction-strongly predicts emotional exhaustion and depersonalization. Reflecting on social connectedness-a type of positive social interaction-can improve one's sense of personal accomplishment with an effect similar in magnitude to more intensive in-person interventions, suggesting that social connectedness through online groups merits further consideration as a tool to mitigate burnout.
View details for DOI 10.1089/jwh.2021.0487
View details for PubMedID 35730998
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A call for an integrated approach to improve efficiency, equity and sustainability in rare disease research in the United States.
Nature genetics
2022
View details for DOI 10.1038/s41588-022-01027-w
View details for PubMedID 35256804
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A qualitative exploration of the experiences of itch for adults living with epidermolysis bullosa.
The British journal of dermatology
1800
View details for DOI 10.1111/bjd.21031
View details for PubMedID 35092694
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Perceived utility and disutility of genomic sequencing for pediatric patients: Perspectives from parents with diverse sociodemographic characteristics.
American journal of medical genetics. Part A
1800
Abstract
Given the limited therapeutic options for most rare diseases diagnosed through genomic sequencing (GS) and the proportion of patients who remain undiagnosed even after GS, it is important to characterize a broader range of benefits and potential harms of GS from the perspectives of families with diverse sociodemographic characteristics. We recruited parents of children enrolled in the Undiagnosed Diseases Network. Parents completed an in-depth interview, and we conducted a comparative content analysis of the data. Parents (n=30) were demographically diverse, with 43.3% identifying as Hispanic, 33.3% primarily Spanish-speaking, and widely variable household income and education. Parents reported minimal changes in their child's health status following GS but did report a range of other forms of perceived utility, including improvements in their child's healthcare management and access, in their own psychological well-being, and in disease-specific social connections and research opportunities. Parents who received a diagnosis more frequently perceived utility across all domains; however, disutility also was reported by both those with and without a diagnosis. Impacts depended on multiple mediating factors, including parents' underlying expectations and beliefs, family sociodemographic characteristics, individual disease characteristics, and prior healthcare access. Our study suggests that the perceived utility of GS varies widely among parents and may depend on multiple individual, sociodemographic, and contextual factors that are relevant for pre- and post-GS counseling, for value assessment of GS, and for policymaking related to access to new genomic technologies.
View details for DOI 10.1002/ajmg.a.62619
View details for PubMedID 34981646
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Beyond race: Recruitment of diverse participants in clinical genomics research for rare disease.
Frontiers in genetics
2022; 13: 949422
Abstract
Purpose: Despite recent attention to increasing diversity in clinical genomics research, researchers still struggle to recruit participants from varied sociodemographic backgrounds. We examined the experiences of parents from diverse backgrounds with enrolling their children in clinical genomics research on rare diseases. We explored the barriers and facilitators parents encountered and possible impacts of sociodemographic factors on their access to research. Methods: We utilized semi-structured interviews with parents of children participating in the Undiagnosed Diseases Network. Interview data were analyzed using comparative content analysis. Results: We interviewed 13 Hispanic, 11 non-Hispanic White, four Asian, and two biracial parents. Participants discussed different pathways to clinical genomics research for rare disease as well as how sociodemographic factors shaped families' access. Themes focused on variation in: 1) reliance on providers to access research; 2) cultural norms around health communication; 3) the role of social capital in streamlining access; and 4) the importance of language-concordant research engagement. Conclusion: Our findings suggest that variables beyond race/ethnicity may influence access in clinical genomics research. Future efforts to diversify research participation should consider utilizing varied recruitment strategies to reach participants with diverse sociodemographic characteristics.
View details for DOI 10.3389/fgene.2022.949422
View details for PubMedID 36072659
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From "Ought" to "Is": Surfacing Values in Patient and Family Advocacy in Rare Diseases.
The American journal of bioethics : AJOB
2021; 21 (12): 1-3
View details for DOI 10.1080/15265161.2021.1996801
View details for PubMedID 34806973
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Opportunities and pitfalls of social media research in rare genetic diseases: a systematic review.
Genetics in medicine : official journal of the American College of Medical Genetics
2021
Abstract
PURPOSE: Social media may be particularly valuable in research in rare genetic diseases because of the low numbers of patients and the rare disease community's robust online presence. The goal of this systematic review was to understand how social media is currently used in rare disease research and the characteristics of the participants in these studies.METHODS: We conducted a systematic review of six databases to identify studies published in English between January 2004 and November 2020, of which 120 met inclusion criteria.RESULTS: Most studies were observational (n=114, 95.0%) and cross-sectional (n=107, 89.2%), and more than half (n=69, 57.5%) utilized only surveys. Only 101 rare diseases were included across all studies. Participant demographics, when reported, were predominantly female (70.1% ± 22.5%) and white (85.0% ± 11.0%) adult patients and caregivers.CONCLUSION: Despite its potential benefits in rare disease research, the use of social media is still methodologically limited and the participants reached may not be representative of the rare disease population by gender, race, age, or rare disease type. As scholars explore using social media for rare disease research, careful attention should be paid to representativeness when studying this diverse patient community.
View details for DOI 10.1038/s41436-021-01273-z
View details for PubMedID 34282302
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Barriers and facilitators to mobile health and active surveillance use among older adults with skin disease.
Health expectations : an international journal of public participation in health care and health policy
2021
Abstract
BACKGROUND: The COVID-19 pandemic has accelerated the adoption of telemedicine, including teledermatology. Monitoring skin lesions using teledermatology may become increasingly important for several skin diseases, including low-risk skin cancers. The purpose of this study was to describe the key factors that could serve as barriers or facilitators to skin disease monitoring using mobile health technology (mHealth) in older adults.METHODS: Older adult dermatology patients 65years or older and their caregivers who have seen a dermatologist in the last 18months were interviewed and surveyed between December 2019 and July 2020. The purpose of these interviews was to better understand attitudes, beliefs and behaviours that could serve as barriers and facilitators to the use of mHealth and active surveillance to monitor low-risk skin cancers.RESULTS: A total of 33 interviews leading to 6022 unique excerpts yielded 8 factors, or themes, that could serve as barriers, facilitators or both to mHealth and active surveillance. We propose an integrated conceptual framework that highlights the interaction of these themes at both the patient and provider level, including care environment, support systems and personal values.DISCUSSION AND CONCLUSIONS: These preliminary findings reveal factors influencing patient acceptance of active surveillance in dermatology, such as changes to the patient-provider interaction and alignment with personal values. These factors were also found to influence adoption of mHealth interventions. Given such overlap, it is essential to address barriers and facilitators from both domains when designing a new dermatology active surveillance approach with novel mHealth technology.PATIENT OR PUBLIC CONTRIBUTION: The patients included in this study were participants during the data collection process. Members of the Stanford Healthcare and Denver Tech Dermatology health-care teams aided in the recruitment phase of the data collection process.
View details for DOI 10.1111/hex.13229
View details for PubMedID 34190397
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A Typology of Existing Machine Learning-Based Predictive Analytic Tools Focused on Reducing Costs and Improving Quality in Health Care: Systematic Search and Content Analysis.
Journal of medical Internet research
2021; 23 (6): e26391
Abstract
BACKGROUND: Considerable effort has been devoted to the development of artificial intelligence, including machine learning-based predictive analytics (MLPA) for use in health care settings. The growth of MLPA could be fueled by payment reforms that hold health care organizations responsible for providing high-quality, cost-effective care. Policy analysts, ethicists, and computer scientists have identified unique ethical and regulatory challenges from the use of MLPA in health care. However, little is known about the types of MLPA health care products available on the market today or their stated goals.OBJECTIVE: This study aims to better characterize available MLPA health care products, identifying and characterizing claims about products recently or currently in use in US health care settings that are marketed as tools to improve health care efficiency by improving quality of care while reducing costs.METHODS: We conducted systematic database searches of relevant business news and academic research to identify MLPA products for health care efficiency meeting our inclusion and exclusion criteria. We used content analysis to generate MLPA product categories and characterize the organizations marketing the products.RESULTS: We identified 106 products and characterized them based on publicly available information in terms of the types of predictions made and the size, type, and clinical training of the leadership of the companies marketing them. We identified 5 categories of predictions made by MLPA products based on publicly available product marketing materials: disease onset and progression, treatment, cost and utilization, admissions and readmissions, and decompensation and adverse events.CONCLUSIONS: Our findings provide a foundational reference to inform the analysis of specific ethical and regulatory challenges arising from the use of MLPA to improve health care efficiency.
View details for DOI 10.2196/26391
View details for PubMedID 34156338
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"Doctors can read about it, they can know about it, but they've never lived with it": How parents use social media throughout the diagnostic odyssey.
Journal of genetic counseling
2021
Abstract
Parents of children with undiagnosed conditions struggle to obtain information about how to treat and support their children. It can be particularly challenging to find communities and other parents who share their experiences and can provide emotional and informational support. This study sought to characterize how parents use social media, both throughout the diagnostic odyssey and post-diagnosis, to meet their informational, social, and emotional support needs. We conducted qualitative semi-structured interviews with 14 parents from the Stanford site of the Undiagnosed Diseases Network (UDN), including five whose children had received a diagnosis through study participation. Interview recordings were analyzed using inductive, team-based coding and thematic analysis based in grounded theory using Dedoose qualitative analysis software. Through this process, we identified four key themes related to social media use. First, parents struggled to find the "right" community, often seeking out groups of similar patients based on symptoms or similar conditions. Second, though they found much valuable information through social media about caring for their child, they also struggled to interpret the relevance of the information to their own child's condition. Third, the social support and access to other patients' and families' lived experiences were described as both highly valued and emotionally challenging, particularly in the case of poor outcomes for similar families. Finally, parents expressed the need to balance concerns about their child's privacy with the value of transparency and data sharing for diagnosis. Our results suggest that the needs and experiences of undiagnosed patients and families differ from those with diagnosed diseases and highlight the need for support in best utilizing social media resources at different stages of the diagnostic odyssey.
View details for DOI 10.1002/jgc4.1438
View details for PubMedID 34096130
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Age-Related Differences in Experiences with Social Distancing at the Onset of the COVID-19 Pandemic: A Computational and Content Analytic Investigation of Natural Language.
JMIR human factors
2021
Abstract
BACKGROUND: As COVID-19 poses different levels of threat to people of different ages, health communication regarding prevention measures such as social distancing and isolation may be strengthened by understanding the unique experiences of different age groups.OBJECTIVE: The aim was to examine how people of different ages (1) experienced the impact of the COVID-19 pandemic and (2) their respective rates and reasons for compliance or non-compliance with social distancing and isolation health guidance.METHODS: We fielded a survey on social media (N = 17,287) early in the pandemic to examine the emotional impact of COVID-19 and individuals' rates and reasons for non-compliance with public health guidance, using computational and content analytic methods of linguistic analysis. The majority of our participants (76.5%) were from the United States.RESULTS: Younger (18-31), middle-aged (32-44, 45-64), and older (65+) individuals significantly varied in how they described the impact of COVID-19 on their lives, including their emotional experience, self-focused attention, and topical concerns. Younger individuals were more emotionally negative and self-focused, while middle-aged people were other-focused and concerned with family. The oldest and most at-risk group was most concerned with health-related terms but were also lower in anxiety and higher in the use of emotionally positive terms than the other, less at-risk age groups. While all groups discussed topics such as acquiring essential supplies, they differentially experienced the impact of school closures and limited social interactions. We also found relatively high rates of non-compliance with COVID-19 prevention measures, such as social distancing and self-isolation, with younger people being more likely to be non-compliant than older people, (P < .001). Among the 43% of respondents who did not fully comply with health orders, people differed substantially in the reasons they gave for non-compliance. The most common reason for non-compliance was not being able to afford missing work (57.3%). While work obligations proved challenging for participants across ages, younger people struggled more to find adequate space to self-isolate and manage their mental and physical health; middle-aged people faced more concerns regarding childcare; and older people perceived themselves as able to take sufficient precautions.CONCLUSIONS: Analysis of natural language can provide insight into rapidly developing public health challenges like the COVID-19 pandemic, uncovering individual differences in emotional experiences and health-related behaviors. In this case, our analyses revealed significant differences between different age groups in feelings about and responses to public health orders aimed to mitigate the spread of COVID-19. To improve public compliance with health orders as the pandemic continues, health communication strategies could be made more effective by being tailored to these age-related differences.CLINICALTRIAL:
View details for DOI 10.2196/26043
View details for PubMedID 33914689
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"It seems like COVID-19 now is the only disease present on Earth": living with a rare or undiagnosed disease during the COVID-19 pandemic.
Genetics in medicine : official journal of the American College of Medical Genetics
2021
Abstract
Patients with rare and undiagnosed diseases (RUDs) face significant health challenges, which may be exacerbated during the COVID-19 pandemic. The goal of this study was to identify specific impacts of the pandemic on RUD patients, and targets for improving support and health-care access.We conducted an online survey of RUD patients and their family members from 21 April to 8 June 2020, recruited from 76 Facebook groups for RUDs. Questions assessed patient characteristics and impacts of the pandemic on RUD diagnosis and management.Respondents (n = 413), including 274 RUD patients and 139 family members, were predominantly female and white, though income varied. Impacts of the pandemic included (1) barriers to accessing essential health care, (2) specific impacts of restrictive COVID-19 visitation policies on ability to advocate in health-care settings, (3) uncertainty and fear regarding COVID-19 risk, (4) exacerbated physical and mental health challenges, (5) magnified impacts of reduced educational and therapeutic services, and (6) unexpected positive changes due to the pandemic.There are specific, serious challenges affecting RUD patients and families during the COVID-19 pandemic. There is an urgent need to develop approaches to mitigate these challenges both during and beyond the pandemic.
View details for DOI 10.1038/s41436-020-01069-7
View details for PubMedID 33420343
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The Intersection of Work and Home Challenges Faced by Physician Mothers During the Coronavirus Disease 2019 Pandemic: A Mixed-Methods Analysis.
Journal of women's health (2002)
2021
Abstract
Objectives: The coronavirus disease 2019 (COVID-19) pandemic has presented extreme challenges for health care workers. This study sought to characterize challenges faced by physician mothers, compare differences in challenges by home and work characteristics, and elicit specific needs and potential solutions. Methods: We conducted a mixed-methods online survey of the Physician Moms Group (PMG) and PMG COVID19 Subgroup on Facebook from April 18th to 29th, 2020. We collected structured data on personal and professional characteristics and qualitative data on home and work concerns. We analyzed qualitative data thematically and used bivariate analyses to evaluate variation in themes by frontline status and children's ages. Results: We included 1,806 participants in analysis and identified 10 key themes. The most frequently identified need/solution was for Community and Government Support (n = 545, 47.1%). When comparing frontline and nonfrontline physicians, those on the frontline more frequently raised concerns about Personal Health and Safety (67.8% vs. 48.4%, p < 0.001), Organizational Communication and Relationships (31.8% vs. 23.8%, p < 0.001), and Family Health and Safety (27.2 vs. 16.6, p < 0.001), while nonfrontline physicians more frequently addressed Patient Care and Safety (56.4% vs. 48.2%, p < 0.001) and Financial/Job Security (33.8% vs. 46.9%, p < 0.001). Participants with an elementary school-aged child more frequently raised concerns about Parenting/Homeschooling (44.0% vs. 31.1%, p < 0.001) and Work/Life Balance (28.4 vs. 13.7, p < 0.001), and participants with a preschool-aged child more frequently addressed Access to Childcare (24.0 vs. 7.7, p < 0.001) and Spouse/Partner Relationships (15.8 vs. 9.5, p < 0.001), when compared to those without children in these age groups. Conclusions: The physician workforce is not homogenous. Health care and government leaders need to understand these diverse challenges in order to meet physicians' professional and family needs during the pandemic.
View details for DOI 10.1089/jwh.2020.8964
View details for PubMedID 33761277
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Anxiety Levels Among Physician Mothers During the COVID-19 Pandemic.
The American journal of psychiatry
2021; 178 (2): 203–4
View details for DOI 10.1176/appi.ajp.2020.20071014
View details for PubMedID 33517747
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Caring for the Caregivers - Covid-19 Vaccination for Essential Members of the Health Care Team.
The New England journal of medicine
2021
View details for DOI 10.1056/NEJMpv2101339
View details for PubMedID 33577149
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When All You Have Is Quality of Life - Making Medical Decisions in the Face of Uncertainty.
The New England journal of medicine
2020; 383 (16): 1507–9
View details for DOI 10.1056/NEJMp2001574
View details for PubMedID 33053282
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Barriers and Facilitators to Real-world Implementation of the Diabetes Prevention Program in Large Healthcare Systems: Lifestyle Coach Perspectives.
Journal of general internal medicine
2020
Abstract
BACKGROUND: Group-based lifestyle change programs based on the Diabetes Prevention Program (DPP) are associated with clinically significant weight loss and decreases in cardiometabolic risk factors. However, these benefits depend on successful real-world implementation. Studies have examined implementation in community settings, but less is known about integration in healthcare systems, and particularly in large, multi-site systems with the potential for extended reach.OBJECTIVE: To examine the barriers and facilitators to successful DPP implementation in a large multi-site healthcare system.DESIGN: Semi-structured interviews, based on the RE-AIM framework, were conducted in person for 30-90min each.PARTICIPANTS: Past and present DPP lifestyle coaches in the healthcare system identified using purposive sampling.APPROACH: Thematic analysis of qualitative data to identify key factors influencing the success of DPP implementation. An iterative consensus process was used to model the relationships among factors.KEY RESULTS: We conducted 33 interviews across 20 clinic sites serving 12 counties. Participants described six key factors as potential barriers or facilitators to implementation, including (1) Broader Context, including the surrounding physical and sociodemographic context; (2) Institutional Context, including finances, infrastructure, and personnel; (3) Program Provision, including curriculum, administration, cost, goals, and visibility; (4) Recruitment Process, including screening and referrals; (5) Lifestyle Coaches, including their characteristics, behaviors, and morale; and (6) Cohort, including group attrition/retention and interpersonal dynamics. These factors were both highly interconnected in their impact on implementation and widely variable across sites within the healthcare system, as illustrated in our multi-level conceptual framework.CONCLUSIONS: This study identified key factors that could serve as barriers or facilitators in the implementation of DPP in large healthcare systems, from the perspective of lifestyle coaches. With further examination, the conceptual model presented here may be used for planning and managing the implementation of group-based behavioral interventions in these settings.
View details for DOI 10.1007/s11606-020-05744-y
View details for PubMedID 32291720
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Differing views regarding diet and physical activity: adolescents versus parents' perspectives.
BMC pediatrics
2020; 20 (1): 137
Abstract
BACKGROUND: Today, approximately one in five United States adolescents age 12 to 19years is obese and just over a third are either overweight or obese. This study examines how parents and peers influence diet and physical activity behaviors of older adolescents (14-18years) with overweight or obesity to inform weight management interventions.METHODS: Adolescent participants included 14 to 18-year-olds with a Body Mass Index (BMI) greater than the 85th percentile for their age and sex who were receiving care in a large healthcare system in Northern California. Adolescents and their parents participated in separate focus groups and interviews (if not able to attend focus groups) that were held at the same time in the same location. We used qualitative thematic analysis to identify common themes discussed in the adolescent and parent focus groups as well as paired analysis of adolescent-parent dyads.RESULTS: Participants included 26 adolescents and 27 parents. Adolescent participants were 14 to 18years old. Half were female and the participants were almost evenly distributed across year in school. The majority self-identified as White (56%) and Asian (36%).Three themes were identified which included 1) parents overestimated how supportive they were compared to adolescents' perception 2) parents and adolescents had different views regarding parental influence on adolescent diet and physical activity behaviors 3) parents and adolescents held similar views on peers' influential role on lifestyle behaviors.CONCLUSION: Parents' and adolescents' differing views suggest that alignment of parent and adolescent expectations and behaviors for supporting effective weight management could be incorporated into interventions.
View details for DOI 10.1186/s12887-020-02038-4
View details for PubMedID 32220230
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Association between financial links to indoor tanning industry and conclusions of published studies on indoor tanning: systematic review.
BMJ (Clinical research ed.)
2020; 368: m7
Abstract
OBJECTIVE: To assess whether an association exists between financial links to the indoor tanning industry and conclusions of indoor tanning literature.DESIGN: Systematic review.DATA SOURCES: PubMed, Embase, and Web of Science, up to 15 February 2019.STUDY SELECTION CRITERIA: Articles discussing indoor tanning and health were eligible for inclusion, with no article type restrictions (original research, systematic reviews, review articles, case reports, editorials, commentaries, and letters were all eligible). Basic science studies, articles describing only indoor tanning prevalence, non-English articles, and articles without full text available were excluded.RESULTS: 691 articles were included in analysis, including empiric articles (eg, original articles or systematic reviews) (357/691; 51.7%) and non-empiric articles letters (eg, commentaries, letters, or editorials) (334/691; 48.3%). Overall, 7.2% (50/691) of articles had financial links to the indoor tanning industry; 10.7% (74/691) articles favored indoor tanning, 3.9% (27/691) were neutral, and 85.4% (590/691) were critical of indoor tanning. Among the articles without industry funding, 4.4% (27/620) favored indoor tanning, 3.5% (22/620) were neutral, and 92.1% (571/620) were critical of indoor tanning. Among the articles with financial links to the indoor tanning industry, 78% (39/50) favored indoor tanning, 10% (5/50) were neutral, and 12% (6/50) were critical of indoor tanning. Support from the indoor tanning industry was significantly associated with favoring indoor tanning (risk ratio 14.3, 95% confidence interval 10.0 to 20.4).CONCLUSIONS: Although most articles in the indoor tanning literature are independent of industry funding, articles with financial links to the indoor tanning industry are more likely to favor indoor tanning. Public health practitioners and researchers need to be aware of and account for industry funding when interpreting the evidence related to indoor tanning.SYSTEMATIC REVIEW REGISTRATION: PROSPERO CRD42019123617.
View details for DOI 10.1136/bmj.m7
View details for PubMedID 32019742
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Development of a patient decision aid for the management of superficial basal cell carcinoma (BCC) in adults with a limited life expectancy.
BMC medical informatics and decision making
2020; 20 (1): 81
Abstract
Basal cell carcinoma (BCC) is a slow-growing, rarely lethal skin cancer that affects people 65 years or older. A range of treatment options exist for BCC, but there is little evidence available to guide patients and providers in selecting the best treatment options.This study outlines the development of a patient decision aid (PDA) for low-risk BCC that can be used by patients and providers to assist in shared decision-making.In accordance with the International Patient Decision Aids Standards (IPDAS) Collaboration framework, feedback from focus groups and semi-structured interviews with patients and providers, an initial prototype of the PDA was developed. This was tested using cognitive interviews and iteratively updated.We created eighteen different iterations using feedback from 24 patients and 34 providers. The key issues identified included: 1) Addressing fear of cancer; 2) Communicating risk and uncertainty; 3) Values clarification; and 4) Time lag to benefit.The PDA does not include all possible treatment options and is currently paper based.Our PDA has been specifically adapted and designed to support patients with a limited life expectancy in making decisions about their low risk BCC together with their doctors.
View details for DOI 10.1186/s12911-020-1081-8
View details for PubMedID 32349762
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Identifying barriers to care and research in hidradenitis suppurativa: findings from a patient engagement event.
The British journal of dermatology
2019
View details for DOI 10.1111/bjd.18818
View details for PubMedID 31883104
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Implementation of a group-based diabetes prevention program within a healthcare delivery system.
BMC health services research
2019; 19 (1): 694
Abstract
Group-based Diabetes Prevention Programs (DPP), aligned with recommendations from the Centers for Disease Control and Prevention, promote clinically significant weight loss and reduce cardio-metabolic risks. Studies have examined implementation of the DPP in community settings, but less is known about its integration in healthcare systems. In 2010, a group-based DPP known as the Group Lifestyle Balance (GLB) was implemented within a large healthcare delivery system in Northern California, across three geographically distinct regional administration divisions of the organization within 12 state counties, with varying underlying socio-demographics. The regional divisions implemented the program independently, allowing for natural variation in its real-world integration. We leveraged this natural experiment to qualitatively assess the implementation of a DPP in this healthcare system and, especially, its fidelity to the original GLB curriculum and potential heterogeneity in implementation across clinics and regional divisions.Using purposive sampling, we conducted semi-structured interviews with DPP lifestyle coaches. Data were analyzed using mixed-method techniques, guided by an implementation outcomes framework consisting of eight constructs: acceptability, adoption, appropriateness, cost, feasibility, fidelity, penetration, and sustainability.We conducted 33 interviews at 20 clinics across the three regional administrative divisions. Consistencies in implementation of the program were found across regions in terms of satisfaction with the evidence base (acceptability), referral methods (adoption), eligibility criteria (fidelity), and strategies to increase retention and effectiveness (sustainability). Heterogeneity in implementation across regions were found in all categories, including: the number and frequency of sessions (fidelity); program branding (adoption); lifestyle coach training (adoption), and patient-facing cost (cost). Lifestyle coaches expressed differing attitudes about curriculum content (acceptability) and suitability of educational level (appropriateness). While difficulties with recruitment were common across regions (feasibility), strategies used to address these challenges differed (sustainability).Variation exists in the implementation of the DPP within a large multi-site healthcare system, revealing a dynamic and important tension between retaining fidelity to the original program and tailoring the program to meet the local needs. Moreover, certain challenges across sites may represent opportunities for considering alternative implementation to anticipate these barriers. Further research is needed to explore how differences in implementation domains impact program effectiveness.
View details for DOI 10.1186/s12913-019-4569-0
View details for PubMedID 31615525
View details for PubMedCentralID PMC6792249
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Beyond exploratory: a tailored framework for designing and assessing qualitative health research.
BMJ open
2019; 9 (8): e030123
Abstract
The objective of this commentary is to develop a framework for assessing the rigour of qualitative approaches that identifies and distinguishes between the diverse objectives of qualitative health research, guided by a narrative review of the published literature on qualitative guidelines and standards from peer-reviewed journals and national funding organisations that support health services research, patient-centered outcomes research and other applied health research fields. In this framework, we identify and distinguish three objectives of qualitative studies in applied health research: exploratory, descriptive and comparative. For each objective, we propose methodological standards that may be used to assess and improve rigour across all study phases-from design to reporting. Similar to hierarchies of quality of evidence within quantitative studies, we argue that standards for qualitative rigour differ, appropriately, for studies with different objectives and should be evaluated as such. Distinguishing between different objectives of qualitative health research improves the ability to appreciate variation in qualitative studies and to develop appropriate evaluations of the rigour and success of qualitative studies in meeting their stated objectives. Researchers, funders and journal editors should consider how further developing and adopting the framework for assessing qualitative rigour outlined here may advance the rigour and potential impact of this important mode of inquiry.
View details for DOI 10.1136/bmjopen-2019-030123
View details for PubMedID 31462482
View details for PubMedCentralID PMC6720470
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Motivations among sexual-minority men for starting and stopping indoor tanning.
The British journal of dermatology
2019; 180 (6): 1529-1530
View details for DOI 10.1111/bjd.17684
View details for PubMedID 30671929
View details for PubMedCentralID PMC6615883
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Supporting caregivers during hematopoietic cell transplantation for children with primary immunodeficiency disorders
JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY
2019; 143 (6): 2271–78
Abstract
Caregivers of children with primary immunodeficiency disorders (PIDs) experience significant psychological distress during their child's hematopoietic cell transplantation (HCT) process.This study aims to understand caregiver challenges and identify areas for health care system-level improvements to enhance caregiver well-being.In this mixed-methods study caregivers of children with PIDs were contacted in August to November 2017 through online and electronic mailing lists of rare disease consortiums and foundations. Caregivers were invited to participate in an online survey assessing sociodemographic variables, the child's medical characteristics, psychosocial support use, and the World Health Organization-5 Well-Being Index. Open-ended questions about health care system improvements were included. Descriptive statistics and linear multivariate regression analyses were conducted. A modified content analysis method was used to code responses and identify emergent themes.Among the 80 caregiver respondents, caregivers had a median age of 34 years (range, 23-62 years) and were predominantly female, white, and married with male children given a diagnosis of severe combined immune deficiency. In the adjusted regression model lower caregiver well-being was significantly associated with lower household income and medical complications. Challenges during HCT include maintaining relationships with partners and the child's healthy sibling or siblings, managing self-care, and coping with feelings of uncertainty. Caregivers suggested several organizational-level solutions to enhance psychosocial support, including respite services, online connections to other PID caregivers, and bedside mental health services.Certain high-risk subpopulations of caregivers might need more targeted psychosocial support to reduce the long-term effect of the HCT experience on their well-being. Caregivers suggested several organizational-level solutions for provision of this support.
View details for DOI 10.1016/j.jaci.2018.10.017
View details for Web of Science ID 000470113200030
View details for PubMedID 31178019
View details for PubMedCentralID PMC6559373
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Physician mothers' experience of workplace discrimination: a qualitative analysis
BMJ-BRITISH MEDICAL JOURNAL
2018; 363: k4926
Abstract
To report woman physicians' experiences, in their own words, of discrimination based on their role as a mother.Qualitative analysis of physician mothers' free-text responses to the open question: "We want to hear your story and experience. Please share" included in questions about workplace discrimination. Three analysts iteratively formulated a structured codebook, then applied codes after inter-coder reliability scores indicated high concordance. The relationships among themes and sub-themes were organized into a conceptual model illustrated by exemplary quotes.Respondents to an anonymous, voluntary online survey about the health and wellbeing of physician mothers posted on a Facebook group, the Physician Moms Group, an online community of US physicians who identify as mothers.We analyzed 947 free-text responses. Participants provide diverse and vivid descriptions of experiences of maternal discrimination. Gendered job expectations, financial inequalities (including lower pay than equally qualified colleagues and more unpaid work), limited opportunities for advancement, lack of support during the pregnancy and postpartum period, and challenging work-life balance are some of the key themes identified. In addition, participants' quotes show several potential structural drivers of maternal discrimination and describe the downstream consequences of maternal discrimination on the physician herself, her career, family, and the healthcare system.These findings provide a view of maternal discrimination directly from the perspective of those who experience it. Women physicians report a range of previously uncharacterized ways in which they experience maternal discrimination. While certain aspects of these experiences are consistent with those reported by women across other professions, there are unique aspects of medical training and the medical profession that perpetuate maternal discrimination.
View details for PubMedID 30541926
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Beyond Consent: Building Trusting Relationships With Diverse Populations in Precision Medicine Research
AMERICAN JOURNAL OF BIOETHICS
2018; 18 (4): 3–20
View details for DOI 10.1080/15265161.2018.1431322
View details for Web of Science ID 000429342200006
View details for PubMedID 27996908
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Collecting Practice-level Data in a Changing Physician Office-based Ambulatory Care Environment: A Pilot Study Examining the Physician induction interview Component of the National Ambulatory Medical Care Survey.
Vital and health statistics. Series 2, Data evaluation and methods research
2017: 1-18
Abstract
Objective This report examines ways to improve National Ambulatory Medical Care Survey (NAMCS) data on practice and physician characteristics in multispecialty group practices. Methods From February to April 2013, the National Center for Health Statistics (NCHS) conducted a pilot study to observe the collection of the NAMCS physician interview information component in a large multispecialty group practice. Nine physicians were randomly sampled using standard NAMCS recruitment procedures; eight were eligible and agreed to participate. Using standard protocols, three field representatives conducted NAMCS physician induction interviews (PIIs) while trained ethnographers observed and audio recorded the interviews. Transcripts and field notes were analyzed to identify recurrent issues in the data collection process. Results The majority of the NAMCS items appeared to have been easily answered by the physician respondents. Among the items that appeared to be difficult to answer, three themes emerged: (a) physician respondents demonstrated an inconsistent understanding of "location" in responding to questions; (b) lack of familiarity with administrative matters made certain questions difficult for physicians to answer; and (c) certain primary care‑oriented questions were not relevant to specialty care providers. Conclusions Some PII survey questions were challenging for physicians in a multispecialty practice setting. Improving the design and administration of NAMCS data collection is part of NCHS' continuous quality improvement process.
View details for PubMedID 29148968
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Mapping the Decision-Making Process for Adjuvant Endocrine Therapy for Breast Cancer: The Role of Decisional Resolve
MEDICAL DECISION MAKING
2017; 37 (1): 79–90
Abstract
Studies show adjuvant endocrine therapy increases survival and decreases risk of breast cancer recurrence for hormone receptor-positive tumors. Yet studies also suggest that adherence rates among women taking this therapy may be as low as 50% owing largely to adverse side effects. Despite these rates, research on longitudinal patient decision making regarding this therapy is scant.We sought to map the decision-making process for women considering and initiating adjuvant endocrine therapy, paying particular attention to patterns of uncertainty and decisional change over time.A longitudinal series of semistructured interviews conducted at a multispecialty health care organization in Northern California with 35 newly diagnosed patients eligible for adjuvant endocrine therapy were analyzed. Analysis led to the identification and indexing of 3 new decision-making constructs-decisional phase, decisional direction, and decisional resolve-which were then organized using a visual matrix and examined for patterns characterizing the decision-making process.Our data reveal that most patients do not make a single, discrete decision to take or not take hormone therapy but rather traverse multiple decisional states, characterized by 1) phase, 2) direction, and 3) strength of resolve. Our analysis tracks these decisional states longitudinally using a grayscale-coded matrix. Our data show that decisional resolve wavers not just when considering therapy, as the existing concept of decisional conflict suggests, but even after initiating it, which may signal future decisions to forgo therapy.Adjuvant endocrine therapy, like other chronic care decisions, has a longer decision-making process and implementation period. Thus, theoretical, empirical, and clinical approaches should consider further exploring the new concept and measurement of decisional resolve, as it may help to improve subsequent medication adherence.
View details for DOI 10.1177/0272989X16640488
View details for Web of Science ID 000389609900011
View details for PubMedID 27053528
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The Importance Of Integrating Narrative Into Health Care Decision Making.
Health affairs (Project Hope)
2016; 35 (4): 720-5
Abstract
When making health care decisions, patients and consumers use data but also gather stories from family and friends. When advising patients, clinicians consult the medical evidence but also use professional judgment. These stories and judgments, as well as other forms of narrative, shape decision making but remain poorly understood. Furthermore, qualitative research methods to examine narrative are rarely included in health science research. We illustrate how narratives shape decision making and explain why it is difficult but necessary to integrate qualitative research on narrative into the health sciences. We draw on social-scientific insights on rigorous qualitative research and our ongoing studies of decision making by patients with cancer, and we describe new tools and approaches that link qualitative research findings with the predominantly quantitative health science scholarship. Finally, we highlight the benefits of more fully integrating qualitative research and narrative analysis into the medical evidence base and into evidence-based medical practice.
View details for DOI 10.1377/hlthaff.2015.1373
View details for PubMedID 27044974
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An exploratory mixed-methods crossover study comparing DVD- vs. Web-based patient decision support in three conditions: The importance of patient perspectives
HEALTH EXPECTATIONS
2015; 18 (6): 2880–91
Abstract
The last 15 years have witnessed considerable progress in the development of decision support interventions (DESIs). However, fundamental questions about design and format of delivery remain.An exploratory, randomized mixed-method crossover study was conducted to compare a DVD- and Web-based DESI. Randomized participants used either the Web or the DVD first, followed by the alternative format. Participants completed a questionnaire to assess decision-specific knowledge at baseline and a questionnaire and structured qualitative interview after viewing each format. Tracking software was used to capture Web utilization. Transcripts were analyzed using integrated inductive and deductive approaches. Quantitative data were analyzed using exploratory bivariate and multivariate analyses.Exploratory knowledge analyses suggest that both formats increased knowledge, with limited evidence that the DVD increased knowledge more than the Web. Format preference varied across participants: 44% preferred the Web, 32% preferred the DVD and 24% preferred 'both'. Patient discussions of preferences for DESI information structure and the importance of a patients' stage of a given decision suggest these characteristics may be important factors underlying variation in utilization, format preferences and knowledge outcomes.Our results suggest that both DESI formats effectively increase knowledge. Patients' perceptions of these two formats further suggest that there may be no single 'best' format for all patients. These results have important implications for understanding why different DESI formats might be preferable to and more effective for different patients. Further research is needed to explore the relationship between these factors and DESI utilization outcomes across diverse patient populations.
View details for DOI 10.1111/hex.12273
View details for Web of Science ID 000368250300087
View details for PubMedID 25263630
View details for PubMedCentralID PMC5810733
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Implementation of Patient Decision Support Interventions in Primary Care: The Role of Relational Coordination.
Medical decision making : an international journal of the Society for Medical Decision Making
2015; 35 (8): 987-98
Abstract
The benefits of patient decision support interventions (DESIs) have been well documented. However, DESIs remain difficult to incorporate into clinical practice. Relational coordination (RC) has been shown to improve performance and quality of care in health care settings. This study aims to demonstrate how applying RC theory to DESI implementation could elucidate underlying issues limiting widespread uptake.Five primary care clinics in Northern California participated in a DESI implementation project. We used a deductive thematic approach guided by behaviors outlined in RC theory to analyze qualitative data collected from ethnographic field notes documenting the implementation process and focus groups with health care professionals. We then systematically compared the qualitative findings with quantitative DESI distribution data.Based on DESI distribution rates, clinics were placed into 3 performance categories: high, middle, and low. Qualitative data illustrated how each clinic's performance related to RC behaviors. Consistent with RC theory, the high-performing clinic exhibited frequent, timely, and accurate communication and positive working relationships. The 3 middle-performing clinics exhibited high-quality communication within physician-staff teams but limited communication regarding DESI implementation across the clinic. The lowest-performing clinic was characterized by contentious relationships and inadequate communication.Limitations of the study include nonrandom selection of clinics and limited geographic diversity. In addition, ethnographic data collected documented only DESI implementation practices and not larger staff interactions contributing to RC.These findings suggest that a high level of RC within clinical settings may be a key component and facilitator of successful DESI implementation. Future attempts to integrate DESIs into clinical practice should consider incorporating interventions designed to increase positive RC behaviors as a potential means to improve uptake.
View details for DOI 10.1177/0272989X15602886
View details for PubMedID 26314727
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Redefining Risk and Benefit: Understanding the Decision to Undergo Contralateral Prophylactic Mastectomy
QUALITATIVE HEALTH RESEARCH
2015; 25 (9): 1251–59
Abstract
Rates of contralateral prophylactic mastectomy (CPM) among unilateral breast cancer patients are rapidly increasing; however, there are little data documenting the decision-making process of patients with no known BRCA mutations, who elect this more aggressive treatment. We conducted semistructured interviews with nine newly diagnosed patients who elected CPM over other surgical options. Using grounded theory, we analyzed interview data to identify influential decision-making factors by prevalence and intensity across participants. Decision-making factors included subjective evaluations of risk and benefit, avoidance of future breast cancer surveillance and accompanying worry, and desire to maintain (or improve) breast appearance. Based solely on survival benefit, the decision to undergo CPM might be viewed as unnecessary or even misguided. However, our findings show the importance of psychosocial factors in patients' assessments of risk and benefit, and support the need for additional patient-provider communication regarding these factors.
View details for DOI 10.1177/1049732314557085
View details for Web of Science ID 000359853200008
View details for PubMedID 25371380
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Shared clinician- patient decision- making about treatment of pediatric asthma: what do we know and how can we use it?
CURRENT OPINION IN ALLERGY AND CLINICAL IMMUNOLOGY
2014; 14 (2): 161–67
Abstract
Shared decision-making (SDM) is an emerging field that promises to improve healthcare. We aim to explore the concept of SDM, how it has been studied or applied in the treatment of asthma, and how it might be implemented to improve adherence and outcomes in pediatric asthma.Healthcare providers often fail to involve their patients in clinical decision-making by not presenting all available options, associated risks and benefits, in light of the patient's values, preferences, concerns, lifestyle, and perceived barriers to following various treatment regimens. It has been argued that SDM is preferable to a clinician-controlled approach and may improve patient outcomes (increase satisfaction with care, reduce decisional conflict and decisional regret, improve health-related quality of life, and increase decision-specific knowledge). This may be especially important in managing chronic conditions in which adherence to treatment regimen may increase if the patient was actively involved in the decision-making. In pediatrics, the decision process is further complicated by the clinician-parent(s)-child interaction. We found no studies on how to effectively involve and communicate with children at different developmental levels, or how to coalesce the parent and child's perspective to work as a unit.SDM has the promise to improve satisfaction with disease management, treatment adherence and patient-centered outcomes in pediatric asthma, but further research is needed to determine its effectiveness and to establish guidelines on how to implement SDM in the clinical setting and incorporate the input and preferences of all stakeholders' perspectives.
View details for DOI 10.1097/ACI.0000000000000046
View details for Web of Science ID 000333256200014
View details for PubMedID 24553296
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Beyond barriers: fundamental 'disconnects' underlying the treatment of breast cancer patients' sexual health
CULTURE HEALTH & SEXUALITY
2014; 16 (9): 1169-1180
Abstract
Sexual health concerns represent one of the most frequently experienced and longest-lasting effects of breast cancer treatment, but research suggests that service providers rarely discuss sexual health with their patients. Existing research examining barriers to addressing patients' sexual health concerns has focused on discrete characteristics of the provider-patient interaction without considering the broader context in which these interactions occur. Drawing on the experiences of 21 breast cancer survivors, this paper explores three ways in which fundamental cultural and structural characteristics of the cancer care system in the USA may prevent breast cancer survivors from addressing their sexual health concerns, including: (1) when patients discussed sexual health with their providers, their providers approached sexuality as primarily physical, while participants experienced complex, multidimensional sexual health concerns; (2) specialisation within cancer care services made it difficult for patients to identify the appropriate provider to address their concerns; and (3) the structure of cancer care literally disconnects patients from the healthcare system at the time when sexual side effects commonly emerged. These data suggest that addressing breast cancer survivors' sexual health concerns requires a multifaceted approach to health systems change.
View details for DOI 10.1080/13691058.2014.939227
View details for Web of Science ID 000342208800012
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A conceptual model of the multiple stages of communication necessary to support patient-centered care.
Journal of comparative effectiveness research
2013; 2 (4): 421-33
Abstract
Patient-centered care requires that both healthcare providers and patients have access to comparative effectiveness research (CER), which provides direct comparisons of the risks and benefits of available clinical options. However, insufficient attention has been paid to developing the comprehensive communication systems necessary to ensure that CER reaches patients and healthcare providers. In this review, we propose a model of the multiple stages of CER communication necessary for patient-centered care and review the existing research and gaps in knowledge relevant to each stage. These stages include: promotion of the underlying concepts and value of CER; translation of CER results; dissemination of CER results; and utilization of the results of CER in shared decision-making between patients and providers. A comprehensive approach to CER communication is necessary to ensure that the growing interest in and availability of CER is able to support a more patient-centered model of healthcare.
View details for DOI 10.2217/cer.13.46
View details for PubMedID 24236683
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An effort to spread decision aids in five California primary care practices yielded low distribution, highlighting hurdles.
Health affairs (Project Hope)
2013; 32 (2): 311-20
Abstract
Despite the proven efficacy of decision aids as interventions for increasing patient engagement and facilitating shared decision making, they are not used routinely in clinical care. Findings from a project designed to achieve such integration, conducted at five primary care practices in 2010-12, document low rates of distribution of decision aids to eligible patients due for colorectal cancer screening (9.3 percent) and experiencing back pain (10.7 percent). There were also no lasting increases in distribution rates in response to training sessions and other promotional activities for physicians and clinic staff. The results of focus groups, ethnographic field notes, and surveys suggest that major structural and cultural changes in health care practice and policy are necessary to achieve the levels of use of decision aids and shared decision making in routine practice envisioned in current policy. Among these changes are ongoing incentives for use, physician training, and a team-based practice model in which all care team members bear formal responsibility for the use of decision aids in routine primary care.
View details for DOI 10.1377/hlthaff.2012.1070
View details for PubMedID 23381524
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The impact of "significant others" on breast cancer patients' treatment decision making.
Journal of clinical oncology
2012; 30 (34_): 20-?
Abstract
20 Background: Shared medical decision making (SDM) has been lauded by advocates for its potential to democratize the patient-physician relationship. However, the practice of SDM is still conceived of as largely a dyadic moment that exists between the patient and the physician. Few studies have looked at the role of significant others (spouses, partners, family members and friends) in decision making or considered how discussions and actions outside the consultation room affect a patient's medical decisions. This prospective study investigated the impact of significant others on the decision making deliberations of newly diagnosed breast cancer patients.Forty-one newly diagnosed breast cancer patients were interviewed at four critical time points throughout treatment to explore how they deliberated decisions with both care providers and significant others. Surveys assessing HRQOL, role preferences and treatment satisfaction along with EHR abstraction augmented interview data. Grounded theory analysis was used to identify recurrent themes in the qualitative data, and survey data were analyzed using IBM SPSS Statistics 20.Emergent themes from our analysis identified several factors that patients consider when faced with cancer treatment decisions, including 1) presentation of treatment options 2) patient or significant other conflict/concordance with care team recommendations 3) perceived risk of recurrence and 4) short and long term impact of treatment on daily life. Participants stressed the need for clinicians to view patients beyond diagnosis and recognize their larger care network as influential factors in their decision making.Our interviews highlight how the current healthcare delivery structure rarely acknowledges the circles of care that can exert influence on decision making. Lack of attention to non-clinical others can lead to sub-optimal medical decision making because these influences are not adequately understood by clinicians. Findings from this study suggest the need to enhance clinicians' and researchers' understanding of the influence of others in patients' treatment decision making, enabling them to intervene in these practices.
View details for DOI 10.1200/jco.2012.30.34_suppl.20
View details for PubMedID 28146982
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A regional survey of dentists' preferences for hiring a dental associate.
Journal of the American Dental Association (1939)
2008; 139 (7): 973-9
Abstract
For many new dental school graduates, working as an associate may be an attractive option. To provide practical information for new job-seeking dentists, the authors conducted a survey to assess dentists' preferences with regard to hiring an associate.The authors mailed surveys to a random sample of 3,875 general dentists in four states that asked questions about the characteristics they preferred when hiring an associate. Five hundred seventy-four dentists (14.8 percent) responded to the survey.The results of this survey showed a wide variation in the characteristics dentists prefer when hiring an associate, but interpersonal skills appear to be at least as important as technical skills for new dentists' success.Opportunities for associate positions appear to be available to new dentists who have a range of skills, experience and training. Practical Implications. These results provide new dentists with information regarding the variety of career opportunities available for associates.
View details for DOI 10.14219/jada.archive.2008.0285
View details for PubMedID 18594084