Barriers and facilitators to collaborative care implementation within the New York State Collaborative Care Medicaid Program.

BACKGROUND: Since 2015, the New York State Office of Mental Health has provided state primary care clinics with outreach, free training and technical assistance, and the opportunity to bill Medicaid for the Collaborative Care Model (CoCM) as part of its Collaborative Care Medicaid Program. This study aims to describe the characteristics of New York State primary care clinics at each step of CoCM implementation, and the barriers and facilitators to CoCM implementation for the New York State Collaborative Care Medicaid Program. METHODS: In this mixed-methods study, clinics were categorized into RE-AIM (Reach, Effectiveness, Adoption, Implementation, and Maintenance) steps. Clinics were sent a survey, which included questions related to payer mix, funding sources, billing codes used, and patient population demographics. Qualitative interviews were conducted with clinic representatives, focusing on barriers or facilitators clinics experienced affecting their progression to the next RE-AIM step. RESULTS: One thousand ninety-nine surveys were sent to primary care clinics across New York State, with 107 (9.7%) completing a survey. Significant differences were observed among the different RE-AIM steps for multiple demographic variables including primary payer, percentage of patients with a diagnose of depression or anxiety, and percent of behavioral health services that are reimbursed, in addition to others. Three main themes regarding barriers and facilitators to implementing CoCM for New York State Medicaid billing emerged from 31 qualitative interviews: (1) Billing requirements, (2) Reimbursement rates, and (3) Buy-in to CoCM. CONCLUSIONS: Survey data align with what we would expect to see demographically in NYS primary care clinics. Qualitative data indicated that CoCM billing requirements/structure and reimbursement rates were perceived as barriers to providing CoCM, particularly with New York State Medicaid, and that buy-in, which included active involvement from organizational leaders and providers that understand the Collaborative Care model were facilitators. Having dedicated staff to manage billing and data reporting is one way clinics minimize barriers, however, there appeared to be a disconnect between what clinics can bill for and the reimbursed amount several clinics are receiving, illustrating the need for stronger billing workflows and continued refinement of billing options across different payers.

Implementing digital systems to facilitate genetic testing for hereditary cancer syndromes: An observational study of 4 clinical workflows.

PURPOSE: National efforts have prioritized the identification of effective methods for increasing case ascertainment and delivery of evidence-based health care for individuals at elevated risk for hereditary cancers. METHODS: This study examined the uptake of genetic counseling and testing following the use of a digital cancer genetic risk assessment program implemented at 27 health care sites in 10 states using 1 of 4 clinical workflows: (1) traditional referral, (2) point-of-care scheduling, (3) point-of-care counseling/telegenetics, and (4) point-of-care testing. RESULTS: In 2019, 102,542 patients were screened and 33,113 (32%) were identified as at high risk and meeting National Comprehensive Cancer Network genetic testing criteria for hereditary breast and ovarian cancer, Lynch syndrome, or both. Among those identified at high risk, 5147 (16%) proceeded with genetic testing. Genetic counseling uptake was 11% among the sites with workflows that included seeing a genetic counselor before testing, with 88% of patients proceeding with genetic testing after counseling. Uptake of genetic testing across sites varied significantly by clinical workflow (6% referral, 10% point-of-care scheduling, 14% point-of-care counseling/telegenetics, and 35% point-of-care testing, P < .0001). CONCLUSION: Study findings highlight the potential heterogeneity of effectiveness attributable to different care delivery approaches for implementing digital hereditary cancer risk screening programs.

Third-Party Genetic Interpretation Tools: A Mixed-Methods Study of Consumer Motivation and Behavior.

In an effort to meet ethical obligations and/or participant expectations, researchers may consider offering "raw" or uninterpreted genetic data for result return. It is therefore important to understand the motivations, behaviors, and perspectives of individuals who might choose to access raw data before such return becomes routine. In the direct-to-consumer (DTC) context, where raw data are often made available to customers, the use of third-party interpretation tools has raised concerns about genotype accuracy, data privacy, reliability of interpretation, and consumption of limited health care resources. However, relatively little is known about why individuals access raw data or what they do with the information received from third-party interpretation. Accordingly, we conducted a survey on raw data access and third-party tool usage among 1,137 DTC customers recruited through social media. Most survey respondents (89%) reported downloading their raw data. Among downloaders, 94% used at least one tool, most commonly Promethease (63%) or GEDmatch (84%). More than half (56%) used both health-related and non-health-related tools and differed significantly from those who used only one tool type in terms of demographics, participation in research, DTC tests ordered, and testing motivations. Exploratory interviews were conducted with 10 respondents and illustrated how social networking, initial lack of interesting findings, and general curiosity contributed to use of multiple tool types. These results suggest that even when initially motivated by ancestry and genealogy, consumers frequently also pursue health information in a largely unregulated and expanding suite of third-party tools, raising both challenges and opportunities for the professional genetics community.

A Study Examining the Usefulness of a New Measure of Research Engagement.

INTRODUCTION: Engagement of relevant stakeholders' ideas, opinions, and concerns is critical to the success of modern research projects. We have developed a tool to measure stakeholder engagement, called the Research Engagement Survey Tool (REST). The purpose of this paper is to present the implementation and uptake of the stakeholder engagement measure REST among research teams, including the assessment of barriers and facilitating factors for use of the new research engagement measure in practice. METHODS: In this implementation study, project team members participated in baseline and follow-up web-based surveys. Web-based interviews were conducted with a subset of project teams that implemented the REST. On the baseline survey, project teams were asked to provide details about up to three ongoing or recently completed projects, were asked if they agreed with compensation for REST completion, and were asked if they would like to send the survey to stakeholders or would prefer our project team to email their project stakeholders. Follow-up surveys contained questions on reactions to implementing REST and results of REST. RESULTS: Project team members/researchers who completed the baseline survey (n=86) were mostly female (79%) and Non-Hispanic/Latino(a) White (76%). Those who implemented REST were also mostly female (86%) and Non-Hispanic/Latino(a) White (71%), with an average of 11 years in academic research. About 98% of all participants completing the baseline survey had the capacity to survey partners, while 100% of all teams who implemented REST did. A small portion of respondents indicated the time commitment of REST would be a barrier (29% of baseline survey respondents, 10% of those who implemented REST) and indicated workload would be a barrier (31% of baseline survey respondents, 14% of those who implemented REST). DISCUSSION: The data presented here indicate that REST implementation is feasible in a volunteer group of ongoing research projects.

The FamilyTalk randomized controlled trial: patient-reported outcomes in clinical genetic sequencing for colorectal cancer.

As genetics gains favor in clinical oncology, it is important to address patient concerns around confidentiality, privacy, and security of genetic information that might otherwise limit its utilization. We designed a randomized controlled trial to assess the social impact of an online educational tool (FamilyTalk) to increase family communication about colorectal cancer (CRC) risk and screening. Of 208 randomized participants, 149 (71.6%) returned six-month surveys. Overall, there was no difference in CRC screening between the study arms. Privacy and confidentiality concerns about medical and genetic information, reactions to genetic test results, and lifestyle changes did not differ between arms. Participants with pathogenic or likely pathogenic (P/LP) and variant of uncertain significance (VUS) results were more likely than those with negative results to report that the results accurately predicted their disease risks (OR 5.37, p = 0.02 and OR 3.13, p = 0.02, respectively). This trial demonstrated no evidence that FamilyTalk impacted patient-reported outcomes. Low power, due to the limited number of participants with P/LP results in the overall sample, as well as the short follow-up period, could have contributed to the null findings.

Population-based implementation of behavioral health detection and treatment into primary care: early data from New York state.

BACKGROUND: The Collaborative Care Model is a well-established, evidence-based approach to treating depression and other common behavioral health conditions in primary care settings. Despite a robust evidence base, real world implementation of Collaborative Care has been limited and very slow. The goal of this analysis is to better describe and understand the progression of implementation in the largest state-led Collaborative Care program in the nation-the New York State Collaborative Care Medicaid Program. Data are presented using the RE-AIM model, examining the proportion of clinics in each of the model's five steps from 2014 to 2019. METHODS: We used the RE-AIM model to shape our data presentation, focusing on the proportion of clinics moving into each of the five steps of this model over the years of implementation. Data sources included: a New York State Office of Mental Health clinic tracking database, billing applications, quarterly reports, and Medicaid claims. RESULTS: A total of 84% of clinics with which OMH had an initial contact [n = 611clinics (377 FQHCs and 234 non-FQHCs)] received some form of training and technical assistance. Of those, 51% went on to complete a billing application, 41% reported quarterly data at least once, and 20% were able to successfully bill Medicaid. Of clinics that reported data prior to the first quarter of 2019, 79% (n = 130) maintained Collaborative Care for 1 year or more. The receipt of any training and technical assistance was significantly associated with our implementation indices: (completed billing application, data reporting, billing Medicaid, and maintaining Collaborative Care). The average percent of patient improvement for depression and anxiety across 155 clinics that had at least one quarter of data was 44.81%. Training and technical assistance source (Office of Mental Health, another source, or both) and intensity (high/low) were significantly related to implementation indices and were observed in FQHC versus non-FQHC samples. CONCLUSIONS: Offering Collaborative Care training and technical assistance, particularly high intensity training and technical assistance, increases the likelihood of implementation. Other state-wide organizations might consider the provision of training and technical assistance when assisting clinics to implement Collaborative Care.

Patient-reported outcomes in the Translational Breast Cancer Research Consortium.

Members of the Translational Breast Cancer Research Consortium conducted an expert-driven literature review to identify a list of domains and to evaluate potential measures of these domains for inclusion in a list of preferred measures. Measures were included if they were easily available, free of charge, and had acceptable psychometrics based on published peer-reviewed analyses. A total of 22 domains and 52 measures were identified during the selection process. Taken together, these measures form a reliable and validated list of measurement tools that are easily available and used in multiple cancer trials to assess patient-reported outcomes in relevant patients.

A pilot study of a telehealth family-focused melanoma preventive intervention for children with a family history of melanoma.

OBJECTIVE: Melanoma preventive interventions for children with familial risk are critically needed because ultraviolet radiation (UVR) exposure and sunburn occurrence early in life are the primary modifiable risk factors for melanoma. The current study examined the feasibility and acceptability of a new, family-focused telehealth intervention for children with familial risk for melanoma and their parents. The study also explored changes in child sun protection and risk behaviors, sunburn occurrence, and objectively measured UVR exposure. METHODS: This was a prospective study with a single-group design (n = 21 parent-child dyads, children ages 8-17). Dyads were asked to participate in three in-person assessments and three live video teleconference intervention sessions. RESULTS: The intervention was feasibly delivered, and the intervention content was acceptable to parents and children. The intervention was associated with improvements in child use of certain sun protection strategies over time and declines in child UVR exposure. CONCLUSIONS: A telehealth-delivered,family-focused melanoma preventive intervention was feasibly delivered and was acceptable to parent-child dyads. Future melanoma preventive interventions for this at-risk population could incorporate eHealth technologies to facilitate improvements in use of sun protection and monitoring of UVR exposure. This trial was registered with Clinicaltrials.gov, number NCT02846714.

Implementing collaborative care to reduce depression for rural native American/Alaska native people.

BACKGROUND: The purpose of this study was to identify the effects of Collaborative Care on rural Native American and Alaska Native (AI/AN) patients. METHODS: Collaborative Care was implemented in three AI/AN serving clinics. Clinic staff participated in training and coaching designed to facilitate practice change. We followed clinics for 2 years to observe improvements in depression treatment and to examine treatment outcomes for enrolled patients. Collaborative Care elements included universal screening for depression, evidence-based treatment to target, use of behavioral health care managers to deliver the intervention, use of psychiatric consultants to provide caseload consultation, and quality improvement tracking to improve and maintain outcomes. We used t-tests to evaluate the main effects of Collaborative Care and used multiple linear regression to better understand the predictors of success. We also collected qualitative data from members of the Collaborative Care clinical team about their experience. RESULTS: The clinics participated in training and practice coaching to implement Collaborative Care for depressed patients. Depression response (50% or greater reduction in depression symptoms as measured by the PHQ-9) and remission (PHQ-9 score less than 5) rates were equivalent in AI/AN patients as compared with White patients in the same clinics. Significant predictors of positive treatment outcome include only one depression treatment episodes during the study and more follow-up visits per patient. Clinicians were overall positive about their experience and the effect on patient care in their clinic. CONCLUSIONS: This project showed that it is possible to deliver Collaborative Care to AI/AN patients via primary care settings in rural areas.

Women's beliefs about what causes obesity: variation by race/ethnicity and acculturation in a Washington State sample.

Objective: Individuals' beliefs about the causes of multifactorial health conditions (causal attributions) shape how they conceptualize and respond to health threats and are therefore important for health promotion. Studies of racial/ethnic and cultural variation in obesity causal beliefs, however, are scarce. To address this gap, this study described beliefs about the underlying causes of obesity (genetic inheritance, diet, and physical activity) in Hispanic and non-Hispanic White women participating in a longitudinal cohort study in South King County, Washington State (n = 1,002).Design: Analysis of baseline survey data. Self-reported obesity causal beliefs were compared by race/ethnicity and acculturation indicators (survey language and nativity) using marginal effect estimates generated from multinomial logistic regression models.Results: Hispanic women had a higher probability of not believing 'at all' in inheritance and physical activity as causes of obesity - an absolute increase of 33% and 5% over non-Hispanic White women, respectively. Both acculturation indicators were also associated with a higher probability of not believing 'at all' in inheritance as a cause of obesity, though Hispanic women who completed the survey in English and were born in the United States had genetic causal beliefs similar to non-Hispanic White women. Behavioral attributions did not vary by acculturation indicators in Hispanic women.Conclusions: Differences in obesity casual beliefs, particularly genetic attributions, exist and may be important for developing and delivering effective obesity-related health promotion interventions. Identifying the determinants and public health consequences of cultural variation in obesity attributions should be the focus of future research.

Exploring relatives' perceptions of participation, ethics, and communication in a patient-driven study for hereditary cancer variant reclassification.

Effective communication of genetic information within families depends on several factors. Few studies explore intra-familial communication of variant of uncertain significance (VUS) results or active collaboration between family members to classify VUS. Our qualitative study aimed to describe the experiences of individuals asked by family members to participate in the FindMyVariant study, a patient-driven family study which aimed to reclassify a clinically identified familial VUS in a hereditary cancer gene. We collected feedback from 56 individuals from 21 different families through phone interviews and written correspondence, transcribed the interviews, and performed thematic analysis on all text. We describe themes from three main topics: participation, ethical considerations, and study impacts. Participation in the FindMyVariant study, defined as returning a sample for targeted genotyping, was motivated by convenience and a desire to help the family, oneself, and science. Relatives were generally responsive to invitations to participate in FindMyVariant from another family member. Those who declined to participate did so due to concerns about research program confidentiality rather than family dynamics. No major ethical issues arose in response to the patient-driven study structure, and no major changes in stress and anxiety, medical care, or behavior occurred. Participation in patient-driven familial VUS classification studies has a neutral or positive impact on family health communication. While it is important to design studies to minimize familial coercion, intra-familial confidentiality breaches, and misinterpretation of genetic results, these were not major concerns among relatives in this study. Clinicians and laboratories may consider encouraging familial communication about genetic variants using family members as liaisons.

Development of FamilyTalk: an Intervention to Support Communication and Educate Families About Colorectal Cancer Risk.

IFamily members of individuals with colorectal cancer (CRC) may be at increased risk of developing the disease. However, the majority of CRC can be prevented through colonoscopy screening and family members may not be aware if they are recommended to pursue earlier screening because of their family history of CRC. As such, tools must be developed to effectively communicate potential changes to the recommended age for colonoscopy screening and other important CRC-related information to family members. We modified and adapted a successful intervention for families with melanoma to be appropriate for families with CRC to increase communication and screening in family members. The multistep process included the following: (1) developing a paper version of the intervention, (2) piloting the paper version among families with CRC, (3) developing the web-based version, and (4) testing the intervention for usability. Qualitative data was collected and analyzed for pilot testing. Usability testing utilized both qualitative and quantitative data. Patients with CRC liked the paper version and had multiple suggestions, including adding a better introduction, sections on genetics and family history, and clearer communication assistance. The web-based tool was well received and improved upon the linear book format with links, better section instructions, and more proactive communication tools for families. These processes produced materials that satisfied individuals from various families with assistance and support for communicating about CRC. Evaluating the effects of the tools in rigorous research projects is the next step.

Randomized trial of a web-based survivor intervention on melanoma prevention behaviors of first-degree relatives.

OBJECTIVES: Melanoma can be prevented through reducing sun exposure and detected early by increasing examination of skin for lesions. First-degree relatives of melanoma cases have higher risk than the general population and, therefore, could be targets of behavioral interventions through families. We tested the effects of a family-based web delivered intervention to melanoma families on the melanoma risk reduction behaviors of first-degree relatives of melanoma cases. METHODS: A total of 313 families that included one first-degree relative were recruited to join this randomized trial. All intervention families received access to the Suntalk website developed to promote family communication and behavioral risk reduction among families of melanoma survivors. RESULTS: First degree relatives in the intervention arm significantly increased their yearly performance of both skin self examination and thorough provider examination from baseline to 12-month follow-up while the control FDRs decreased their yearly performance of both of those behaviors (p's = 0.006 and 0.005, respectively). Several sun protection behaviors increased significantly in FDRs in the intervention arm but not the control arm, including wearing a covering on the head (p = 0.005), staying in available shade (p = 0.008), and avoiding sun exposure during peak hours (p = 0.010). Some of these changes were mediated by perceptions of risk and other process variables. CONCLUSIONS: A web-based intervention can reduce risk of melanoma through changes in relevant behaviors among first-degree relatives of melanoma survivors. Future research should identify methods for making this type of intervention accessible to more families and for broadening the reach to other types of cancer families. PRACTICE IMPLICATIONS: This program can be provided to melanoma families to improve their sun protection and screening behaviors, at the point of diagnosis.

Outcomes of 92 patient-driven family studies for reclassification of variants of uncertain significance.

PURPOSE: Family studies are an important but underreported source of information for reclassification of variants of uncertain significance (VUS). We evaluated outcomes of a patient-driven framework that offered familial VUS reclassification analysis to any adult with any clinically ascertained VUS from any laboratory in the United States. METHODS: With guidance from FindMyVariant.org, participants recruited their own relatives for study participation. We genotyped relatives, calculated quantitative cosegregation likelihood ratios, and evaluated variant classifications using Tavtigian's unified framework for Bayesian analysis with American College of Medical Genetics and Genomics/Association for Molecular Pathology (ACMG/AMP) criteria. We report participation and VUS reclassification rates from the 50 families enrolled for at least one year and reclassification results for 112 variants from the larger 92-family cohort. RESULTS: For the 50-family cohort, 6.7 relatives per family were invited to participate and 67% of relatives returned samples for genotyping. Sixty-one percent of VUS were reclassified, 84% of which were classified as benign or likely benign. Genotyping relatives identified a de novo variant, phase variants, and relatives with phenotypes highly specific for or incompatible with specific classifications. CONCLUSIONS: Motivated families can contribute to successful VUS reclassification at substantially higher rates than those previously published. Clinical laboratories could consider offering family studies to all patients with VUS.

MAGENTA (Making Genetic testing accessible): a prospective randomized controlled trial comparing online genetic education and telephone genetic counseling for hereditary cancer genetic testing.

BACKGROUND: Studies have consistently indicated that the majority of individuals meeting the US Prevention Services Task Force guidelines for genetic testing have not had genetic counseling or testing. Despite increased availability and lower costs of multiplex cancer gene panels, there remains a gap in genetics services that has not been addressed by the current care delivery models. Lower cost of DNA sequencing with online patient-initiated ordering could increase test availability, but the ideal quantity and delivery method of patient education is not known. We hypothesized that online genetic education and testing with access to board certified genetic counselors could improve access to genetic testing while maintaining test quality and clinical utility. The MAGENTA (MAking GENetic Testing Accessible) trial is a nationwide randomized study designed to compare the effectiveness of online genetic education with pre- and post-test telephone genetic counseling to three potentially more accessible alternative approaches: online genetic education with optional telephone counseling, online genetic education with required pre-test telephone genetic counseling, and online genetic education with required post-test telephone genetic counseling. METHODS: 3000 women nationwide will undergo genetic testing for 19 hereditary cancer genes. This is a randomized four-arm non-inferiority study with equal randomization. The four study arms were selected to independently assess the delivery of genetic information both before and after genetic testing (pre-test and post-test) by either requiring telephone genetic counseling or providing only online education with optional telephone counseling. Patients have post-test telephone counseling when testing positive for a pathogenic inherited mutation in all four arms. Surveys measuring psychological, behavioral and cognitive state are completed online at baseline, 3 months, 12 months and 24 months post-results disclosure. The primary study outcome is cancer-risk distress at 3 months post-result disclosure. DISCUSSION: This trial will assess the use of a genetic service model using online access and electronic education, while evaluating the need for personal pre- and post-test genetic counseling. Data from this study may lead to increased options for delivery of genetic testing and possibly increase access to genetic testing. Identifying more individuals with inherited cancer susceptibility will allow targeted cancer prevention. TRIAL REGISTRATION: Clinicaltrials.gov: NCT02993068 (registered December 14, 2016).

Comparing provider and patient views of issues for low-resourced breast cancer patients.

OBJECTIVE: The purpose of this study was to compare provider and patient views from the same clinical settings on issues raised by low-socioeconomic status (SES) breast cancer survivors. METHODS: We conducted qualitative interviews among two groups: low-SES breast cancer survivors (n = 37) and medical personnel (ie, physicians, nurses, and navigators; n = 8) who interact and serve with these patients from two geographically distinct low-resourced clinical settings. These semistructured qualitative interviews used grounded theory to identify several potential themes, such as finances, resources, and medical care. Transcripts were coded and summarized into themes. RESULTS: We analyzed each type of interview data separately then compared patient and provider perspectives. From these qualitative interviews, we discovered that low-SES breast cancer survivors reported many unmet needs, including transportation, housing, health literacy, and language, among others. Providers reported that many of these needs are served by the extensive network of supports surrounding these patients. CONCLUSIONS: These results illustrate that low-SES breast cancer survivors have unique needs that differ from other breast cancer survivors. Many providers feel that these needs are being met, but patients have more diverse experiences. By better addressing the links between resource needs and low-SES breast cancer survivors, quality of life can be improved.

Patients' perspectives of variants of uncertain significance and strategies for uncertainty management.

Variants of uncertain significance (VUS) are a well-recognized source of uncertainty in genomic medicine. Despite the existence of straightforward clinical management recommendations, patients report feeling anxiety, worry, and uncertainty in response to VUS. We report the first structured analysis of patient perspectives of VUS-related uncertainty in genome sequencing using Han's taxonomy of genomic uncertainty. We conducted in-depth semi-structured interviews with 11 patients to elicit their thoughts regarding implications of the result for themselves and their family members. Patients' primary concern with VUS-related uncertainty involved personal and practical issues as they directly inform health-care decisions. Patients demonstrated good understanding of the epistemic nature of VUS uncertainty-that information about such variant is currently unknown. However, between-provider discordance in explanations of the implication of this uncertainty for patients' diagnosis, prognosis, and therapy was a major contributor to the overall experience of uncertainty. Strategies for uncertainty reduction involved periodically checking back for reclassification and receiving concordant and clear recommendation from providers. Other proactive strategies of uncertainty reduction-such as information seeking and reading the genetic test report-were not helpful. Collectively, these findings offer previously unreported insight into uncertainty management strategies used by patients which have the potential to guide clinical management practices.

Patient goals, motivations, and attitudes in a patient-driven variant reclassification study.

Family studies to reclassify clinically ascertained variants of uncertain significance (VUS) can impact risk assessment, medical management, and psychological outcomes for patients and their families. There are limited avenues for patients and their families to actively participate in VUS reclassification, and access to family studies at most commercial laboratories is restricted by multiple factors. To explore patient attitudes about participation in family studies for VUS reclassification, we conducted semistructured pre- and post-participation telephone interviews with 38 participants in a family-based VUS reclassification study that utilized a patient-driven approach for family ascertainment and recruitment. Participants had VUS from multigene panel testing performed at multiple clinical laboratories for cancer or other disease risk. Inductive thematic analysis of transcribed interviews highlighted four major themes: (a) Participants' study goals were driven by the desire to resolve uncertainty related to the VUS, (b) Participants had mixed reactions to the VUS reclassification outcomes of the study, (c) Personal, public, and familial knowledge increased through study participation and (d) Participants used study participation to actively cope with the uncertainty of a VUS. As personalized genomic medicine becomes more prevalent, clinicians, clinical laboratories, and researchers could consider creating more opportunities for active partnership with patients and families, who are motivated to contribute data to familial VUS studies.

Content validation of a quantitative stakeholder engagement measure.

AIM: Using a stakeholder-engaged approach, this study conducted content validation and item reduction of a quantitative measure of research engagement. METHODS: A five-round modified Delphi process was used to reach consensus on items. Rounds 1-3 and 5 were conducted using web-based surveys. Round 4 consisted of a 2-day, in-person meeting. Delphi panelists received individualized reports outlining individual and aggregate group responses after rounds 1-3. RESULTS: Over the five-round process, items were added, dropped, modified, and moved from one engagement principle to another. The number of items was reduced from 48 to 32, with three to five items corresponding to eight engagement principles. CONCLUSIONS: Research that develops standardized, reliable, and accurate measures to assess stakeholder engagement is essential to understanding the impact of engagement on scientific discovery and the scientific process. Valid quantitative measures to assess stakeholder engagement in research are necessary to assess associations between engagement and research outcomes.

Hereditary cancer gene panel test reports: wide heterogeneity suggests need for standardization.

PURPOSE: Laboratory-generated genomic test reports are used to convey complex, and frequently multivariant or uncertain, information about disease risk to medical genetics professionals as well as to nonspecialist clinicians, patients, and family members. However, few guidelines exist to guide the content and format of genomic test reports, and little is known about variation in current reporting practices. METHODS: We conducted a structured content analysis of hereditary cancer gene panel test reports obtained from 16 United States-based CLIA-certified laboratories, including reports describing a variant of uncertain significance (VUS) only and reports with both a VUS and pathogenic or likely pathogenic (P/LP) test result. RESULTS: Report content and format varied widely across laboratories and between VUS and VUS + P/LP reports from the same laboratory, with regard to the inclusion and visual prominence of key content as well as in terms of overall length and readability. CONCLUSION: Test report heterogeneity is likely to reflect both the lack of comprehensive reporting guidelines and disagreements between laboratories about the salience of specific types of information to test interpretation and use. Future research should explore the impact of reporting differences on clinician interpretation and shared decision making.