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To retrospectively explore the perspectives of parents of children with severe neurological impairment (SNI), such as those with severe cerebral palsy, epilepsy syndromes, and structural brain differences, on clinician counseling regarding home mechanical ventilation (HMV).Inductive thematic analysis was performed on data from telephone interviews with parents who chose for and against HMV for their child with SNI at three academic children's hospitals across the USA.Twenty-six parents/legal guardians of 24 children were interviewed. Fourteen children had static encephalopathy, 11 received HMV, and 20 were alive at the time of parent interviews. Themes included how HMV related to the child's prognosis, risk of death, and integration with goals of care. Although clinicians voiced uncertainty about how HMV would impact their child, parents felt this was coupled with prescriptive/intimidating examples about the child's end of life and judgments about the child's quality of life.While prognositc uncertainty exists, this study suggests that parents of children with SNI seek clinician counseling about HMV that considers their goals of care and views on their child's quality of life.

CONTEXT: Parents of children with severe neurologic impairment (SNI) often face high-stakes medical decisions when their child is hospitalized. These decisions involve technology/surgery, goals of care/advance care planning, or transitions of care. OBJECTIVE: This study describes the experiences of parents of children with SNI during decision-making. METHODS: Eligible participants were parents facing a decision for a child with SNI admitted to acute or intensive care units at a single tertiary pediatric center. Parents completed 1:1 semi-structured interviews and brief surveys between August 2019-February 2020. Demographic information was extracted from the child’s electronic health record. A team of palliative and complex care researchers with expertise in qualitative methods used thematic content analysis to formulate results. RESULTS: 25 parents participated. The majority had children with congenital/chromosomal SNI conditions (n=13, 65%), >5 subspecialists (n=14, 61%), and chronic technology assistance (n=25, 100%). 68% (n=17) were mothers and 100% identified as being their child’s primary decision-maker. Responses from parents included 3 major themes: 1) our roles and actions; 2) our stresses and challenges; and 3) our meaning and purpose. Responses highlighted the pervasiveness of parental decision-making efforts and parents’ advocacy and vigilance regarding their child’s needs. Despite this, parents often felt unheard and undervalued in the hospital. CONCLUSIONS: During hospitalizations, when parents of children with SNI often face high-stakes medical decisions, interventions are needed to support parents and ensure they feel heard and valued as they navigate their child’s medical needs and system challenges. KEY MESSAGE: This qualitative study of parents of children with SNI facing decision-making in the hospital revealed not only the demands and stresses but also the meaning and purpose parents experience. As parents navigate their crucial roles, tools are needed to empower parents to be heard and to advocate for their child.

OBJECTIVES To explore the family experience of home ventilation through a comparison of anticipated home life changes with subsequent experiences. STUDY DESIGN Guided interviews with parents across three states who chose home ventilation for their child within the last 5 years. PATIENT-SUBJECT SELECTION Purposive sampling of parents who chose home ventilation for their child within the last 5 years. METHODS Interviews were transcribed for qualitative analysis and analyzed for thematic saturation and prevalence of codes. RESULTS Twenty families were interviewed. Families generally reported not considering potential home life changes when facing the decision about home ventilation; instead, they worried most about medical management. These concerns reversed in importance later. Families learned medical management quickly but felt largely unprepared for the extensive changes to their home life, including isolation, altered relationships with extended family and community, effects on siblings, financial strain, and need for physical changes to their house. Families had not anticipated how much they would be affected by home healthcare as a new part of their life. CONCLUSIONS The priorities that families consider during decisions about pediatric home ventilation may not be aligned with the actual home experience of this technology. Given that the success of home ventilation largely rests with the family's care, family expectations for home life adaptations must be augmented, as should postdischarge supports for families with complex home care experiences.

INTRODUCTION: The Cancer Health Assessments Reaching Many study seeks to reduce disparities in genomic care. Two patient advisory committees (PACs) were formed, 1 of English speakers and 1 of Spanish speakers, to vet study processes and materials. Stakeholder engagement in research is relatively new, and we know little about how stakeholders view their engagement. We wanted to learn how patient stakeholders viewed the process, to inform future patient engagement efforts. METHODS: Patients at 2 study sites were invited to serve on 2 PACs. We used an iterative engagement process to solicit and incorporate patient feedback. Much of the PAC feedback on study materials and processes was incorporated. Using surveys and exit interviews, we evaluated stakeholders’ experiences as PAC members. RESULTS: Nearly all PAC members felt satisfied and included in the study decisions, but surveys and exit interviews suggested the need to improve communications. DISCUSSION: Although most believed their feedback was used, and most felt included in study decisions, some said they did not know whether their opinions were used to modify materials or approaches. This suggests the need to explain to patient stakeholders the extent to which their feedback was used and to inform them about the impact that other stakeholders, such as institutional review boards, have on decisions. CONCLUSION: Our evaluation highlights the value of dedicating resources to stakeholder engagement. Although gathering patient feedback on study materials and processes introduced time constraints and complexity to our study, adaptations to materials and processes furthered study goals.

BACKGROUND AND OBJECTIVES: Successful clinical integration of genomic sequencing (GS) requires evidence of its utility. While GS potentially has benefits (utilities) or harms (disutilities) across multiple domains of life for both patients and their families, there is yet no empirically informed conceptual model of these effects. Our objective was to develop an empirically informed conceptual model of perceived utility of GS that captures utilities and disutilities for patients and their families across diverse backgrounds. METHODS: We took a patient-centered approach in which we began with a review of existing literature followed by collection of primary interview data. We conducted semi-structured interviews to explore types of utility in a clinically and sociopolitically diverse sample of 60 adults from seven Clinical Sequencing Evidence-Generating Research (CSER) consortium projects. Interviewees had either personally received, or were parents of a child who had received, GS results. Qualitative data was analysed using thematic analysis. Findings from interviews were integrated with existing literature on clinical and personal utility to form the basis of an initial conceptual model that was refined based on expert review and feedback. RESULTS: Five key utility types that have been previously identified in qualitative literature held up as primary domains of utility and disutility in our diverse sample. Interview data were used to specify and organize subdomains of an initial conceptual model. After expert refinement, the five primary domains included in the final model are: clinical, emotional, behavioral, cognitive, and social, and several sub-domains are specified within each. CONCLUSION: We present an empirically-informed conceptual model of perceived utility of GS. This model can be used to guide development of instruments for patient-centered outcome measurement that capture the range of relevant utilities and disutilities and inform clinical implementation of GS.

BACKGROUND: Clinical genomic implementation studies pose challenges for informed consent. Consent forms often include complex language and concepts, which can be a barrier to diverse enrollment, and these studies often blur traditional research-clinical boundaries. There is a move toward self-directed, web-based research enrollment, but more evidence is needed about how these enrollment approaches work in practice. In this study, we developed and evaluated a literacy-focused, web-based consent approach to support enrollment of diverse participants in an ongoing clinical genomic implementation study. METHODS: As part of the Cancer Health Assessments Reaching Many (CHARM) study, we developed a web-based consent approach that featured plain language, multimedia, and separate descriptions of clinical care and research activities. CHARM offered clinical exome sequencing to individuals at high risk of hereditary cancer. We interviewed CHARM participants about their reactions to the consent approach. We audio recorded, transcribed, and coded interviews using a deductively and inductively derived codebook. We reviewed coded excerpts as a team to identify overarching themes. RESULTS: We conducted 32 interviews, including 12 (38%) in Spanish. Most (69%) enrolled without assistance from study staff, usually on a mobile phone. Those who completed enrollment in one day spent an average of 12 minutes on the consent portion. Interviewees found the information simple to read but comprehensive, were neutral to positive about the multimedia support, and identified increased access to testing in the study as the key difference from clinical care. CONCLUSIONS: This study showed that interviewees found our literacy-focused, web-based consent approach acceptable; did not distinguish the consent materials from other online study processes; and valued getting access to testing in the study. Overall, conducting empirical bioethics research in an ongoing clinical trial was useful to demonstrate the acceptability of our novel consent approach but posed practical challenges.

This study aimed to evaluate the laboratory-related outcomes of participants who were offered genomic testing based on cancer family history risk assessment tools.Patients from clinics that serve populations with access barriers, who are screened at risk for a hereditary cancer syndrome based on adapted family history collection tools (the Breast Cancer Genetics Referral Screening Tool and PREMMOf all the participants, 87% successfully returned a saliva kit. Overall, 5% had a pathogenic/likely pathogenic cancer risk variant and 1% had a secondary finding. Almost all (14/15, 93%) participants completed recommended consultations with nongenetics providers after an average of 17 months. The recommended actions (eg, breast magnetic resonance imaging) were completed by 17 of 25 participants. Participant personal history of cancer and PREMMThis accessible model of hereditary cancer risk assessment and genetic testing yielded results that were often acted upon by patients and physicians.

Rationale Deciding about pediatric home ventilation is exceptionally challenging for parents. Understanding the decision-making needs of parents who made different choices for their children could inform clinician counseling that better supports parents' diverse values and goals. Objectives To determine how clinicians can meet the decisional needs of parents considering home ventilation using a balanced sample of families who chose for or against intervention. Methods We conducted semi-structured interviews of parents who chose for or against home ventilation for their child within the previous five years. Parents were recruited from three academic centers across the United States. Interviews focused on parent-clinician communication during decision-making and how clinicians made the process easier or more difficult. Qualitative analysis was used to generate themes and identify key results. Results Thirty-eight parents were interviewed; 20 chose for and 18 chose against home ventilation. Five themes described their perspectives on how clinicians can facilitate high-quality decision-making: demonstrating dedication to families, effectively managing the medical team, introducing the concept of home ventilation with intention, facilitating meaningful conversation about the treatment options, and supporting and respecting the family's decision. Conclusions High quality decision-making around home ventilation depends on individual clinician actions and the complex operations of large academic settings. Strong working relationships with parents, collaborative alliances with colleagues, and appropriate delivery of key content can help meet the needs of parents considering invasive breathing supports for their children. This article is protected by copyright. All rights reserved.

Health care workers face serious mental health challenges as a result of ongoing work stress. The COVID-19 pandemic exacerbated that stress, resulting in high rates of anxiety, depression, and burnout. To date, few evidence-based programs targeting mental health outcomes in health care workers have been described.To assess the feasibility, acceptability, and preliminary outcomes of a skills-based coaching program designed to reduce stress and build resilience.A pilot cohort study was conducted between September 2020 and April 2021 using preprogram and postprogram assessments and a mixed-methods analysis. Duration of follow-up was 7 weeks. The coaching program was delivered via video conferencing. Participants were health care workers and staff from a large urban health system.The Promoting Resilience in Stress Management (PRISM) program, a manualized, skills-based coaching program originally developed for adolescents and young adults with serious/chronic illness, was adapted to support health care workers and staff ("PRISM at Work"). It included 6 weekly 1-hour group sessions.Feasibility was defined a priori as 70% completion rates. Acceptability was defined quantitatively (satisfaction scores) and qualitatively (open-ended questions regarding experience with program). Preliminary outcomes were assessed with preprogram and post program assessments of self-reported resilience, stress, anxiety, and burnout using validated instruments. Descriptive statistics summarized demographic variables and feasibility and acceptability. Linear mixed effects regression models examined preliminary outcomes, controlling for relevant covariates.A total of 153 participants (median [SD] age, 40.6 [10.1] years; 142 [92%] were female; 128 [84%] identified as having White race; 81 [53%] were in patient-facing roles) enrolled. Of the 132 health care workers who provided follow-up surveys, 120 (91%) had completed the program, and 116 (88%) reported being satisfied. Answers to open-ended questions suggested that participants wanted more PRISM either with longer or additional sessions. Participant-reported resilience (β = 1.74; 95% CI, 1.00-2.48), stress (β = -2.40; 95% CI, -3.28 to -1.51), anxiety (β = -2.04; 95% CI, -2.74 to -1.34), and burnout-exhaustion (β = -0.37; 95% CI, -0.56 to -0.18) improved after the program.Results of this study suggest that PRISM at Work may have utility for health care workers and staff in that the program was found to be feasible, acceptable, and associated with improved outcomes.

Objective To understand what considerations drive family decisions for, and against, pediatric home ventilation. Study design Qualitative interviews with parents of children who faced a decision about home ventilation in the previous 5 years at 3 geographically dispersed institutions. Results In total, 38 families (42 parents) were interviewed; 20 families opted for pediatric home ventilation, and 18 families opted against. Approximately one-quarter of children had isolated chronic lung disease; the remainder had medical complexity that was expected to remain static or decline. Parent perspectives about home ventilation generally reflected whether the child was early, or later, in their disease trajectory. Early on, parents often interpreted prognostic uncertainty as hope and saw home ventilation as a tool permitting time for improvement. For families of children later in their disease course, often already with home technology and home nursing, home ventilation held less possibility for meaningful improvement. Nearly all families experienced the decision as very emotionally distressing. Reflecting back, most families described feeling satisfied with whatever decision they made. Conclusions The 2 principal groups of families in our cohort—those with children whose respiratory insufficiency might improve, and those with children facing chronic decline—warrant targeted counseling approaches about initiating home ventilation. The distressing nature of this decision should be anticipated and family supports reinforced.

Background DSM265 is a selective inhibitor of Plasmodium dihydroorotate dehydrogenase that fully protected against controlled human malarial infection (CHMI) by direct venous inoculation of Plasmodium falciparum sporozoites when administered 1 day before challenge and provided partial protection when administered 7 days before challenge. Methods A double-blinded, randomized, placebo-controlled trial was performed to assess safety, tolerability, pharmacokinetics, and efficacy of 1 oral dose of 400 mg of DSM265 before CHMI. Three cohorts were studied, with DSM265 administered 3 or 7 days before direct venous inoculation of sporozoites or 7 days before 5 bites from infected mosquitoes. Results DSM265-related adverse events consisted of mild-to-moderate headache and gastrointestinal symptoms. DSM265 concentrations were consistent with pharmacokinetic models (mean area under the curve extrapolated to infinity, 1707 µg*h/mL). Placebo-treated participants became positive by quantitative reverse transcription-polymerase chain reaction (qRT-PCR) and were treated 7-10 days after CHMI. Among DSM265-treated subjects, 2 of 6 in each cohort were sterilely protected. DSM265-treated recipients had longer times to development of parasitemia than placebo-treated participants (P < .004). Conclusions This was the first CHMI study of a novel antimalarial compound to compare direct venous inoculation of sporozoites and mosquito bites. Times to qRT-PCR positivity and treatment were comparable for both routes. DSM265 given 3 or 7 days before CHMI was safe and well tolerated but sterilely protected only one third of participants.

Purpose This study describes challenges faced while incorporating sometimes conflicting stakeholder feedback into study design and development of patient-facing materials for a translational genomics study aiming to reduce health disparities among diverse populations. Methods We conducted an ethnographic analysis of study documents including summaries of patient advisory committee meetings and interviews, reflective field notes written by study team members, and correspondence with our institutional review board (IRB). Through this analysis, we identified cross-cutting challenges for incorporating stakeholder feedback into development of our recruitment, risk assessment, and informed consent processes and materials. Results Our analysis revealed three key challenges: (1) balancing precision and simplicity in the design of study materials, (2) providing clinical care within the research context, and (3) emphasizing potential study benefits versus risks and limitations. Conclusion While involving patient stakeholders in study design and materials development can increase inclusivity and responsiveness to patient needs, patient feedback may conflict with that of content area experts on the research team and IRBs who are tasked with overseeing the research. Our analysis highlights the need for further empirical research about ethical challenges when incorporating patient feedback into study design, and for dialogue with genomic researchers and IRB representatives about these issues.

Controlled human malaria infection (CHMI) studies deliberately infect healthy participants with malaria to test interventions faster and more efficiently. Some argue the study design and high payments offered raise ethical concerns about participants’ understanding of risks and undue inducement. We conducted baseline and exit interviews with 16 CHMI study participants to explore these concerns. Participants described themes including decision-making tension with friends and family, mixed motivations for participating, low study risks but high burdens, fair compensation, sacrificing values, deceiving researchers, and perceived benefits. Our findings do not support concerns that high payments limit understanding of study risks, but suggest participants may lack appreciation of study burdens, withhold information or engage in deception, and experience conflict with others regarding study participation.