Purpose

Engaging primary care providers (PCPs) in BRCA1/2 testing and results disclosure would increase testing access. The BRCA Founder OutReach (BFOR) study is a prospective study of BRCA1/2 founder mutation screening among individuals of Ashkenazi Jewish descent that sought to involve participants' PCPs in results disclosure. We used quantitative and qualitative methods to evaluate PCPs' perspectives, knowledge, and experience disclosing results in BFOR.

Methods

Among PCPs nominated by BFOR participants to disclose BRCA1/2 results, we assessed the proportion agreeing to disclose. To examine PCP's perspectives, knowledge, and willingness to disclose results, we surveyed 501 nominated PCPs. To examine PCPs' experiences disclosing results in BFOR, we surveyed 101 PCPs and conducted 10 semi-structured interviews.

Results

In the BFOR study overall, PCPs agreed to disclose their patient's results 40.5% of the time. Two hundred thirty-four PCPs (46.7%) responded to the initial survey. Responding PCPs were more likely to agree to disclose patients' results than non-responders (57.3% vs. 28.6%, p<0.001). Among all respondents, most felt very (19.7%) or somewhat (39.1%) qualified to share results. Among PCPs declining to disclose, insufficient knowledge was the most common reason. In multivariable logistic regression, feeling qualified was the only variable significantly associated with agreeing to disclose results (OR 6.53, 95% CI 3.31, 12.88). In post-disclosure surveys (response rate=55%), PCPs reported largely positive experiences. Interview findings suggested that although PCPs valued the study-provided educational materials, they desired better integration of results and decision support into workflows.

Conclusion

Barriers exist to incorporating BRCA1/2 testing into primary care. Most PCPs declined to disclose their patients' BFOR results, although survey respondents were motivated and had positive disclosure experiences. PCP training and integrated decision support could be beneficial.

Trial registration

ClinicalTrials.gov (NCT03351803), November 24, 2017.

Background

Evidence is needed regarding effective incentive strategies to increase clinician survey response rates. Cash cards are increasingly used as survey incentives; they are appealing because of their convenience and because in some cases their value can be reclaimed by investigators if not used. However, their effectiveness in clinician surveys is not known. In this study within the BRCA Founder OutReach (BFOR) study, a clinical trial of population-based BRCA1/2 mutation screening, we compared the use of upfront cash cards requiring email activation versus checks as clinician survey incentives.

Methods

Participants receiving BRCA1/2 testing in the BFOR study could elect to receive their results from their primary care provider (PCP, named by the patient) or from a geneticist associated with the study. In order to understand PCPs' knowledge, attitudes, experiences and willingness to disclose results we mailed paper surveys to the first 501 primary care providers (PCPs) in New York, Boston, Los Angeles and Philadelphia who were nominated by study participants to disclose their BRCA1/2 mutation results obtained through the study. We used alternating assignment stratified by city to assign the first 303 clinicians to receive a $50 up-front incentive as a cash card (N = 155) or check (N = 148). The cash card required PCPs to send an activation email in order to be used. We compared response rates by incentive type, adjusting for PCP characteristics and study site.

Results

In unadjusted analyses, PCPs who received checks were more likely to respond to the survey than those who received cash cards (54.1% versus 41.9%, p = 0.046); this remained true when we adjusted for provider characteristics (OR for checks 1.61, 95% CI 1.01, 2.59). No other clinician characteristics had a statistically significant association with response rates in adjusted analyses. When we included an interaction term for incentive type and city, the favorable impact of checks on response rates was evident only in Los Angeles and Philadelphia.

Conclusions

An up-front cash card incentive requiring email activation may be less effective in eliciting clinician responses than up-front checks. However, the benefit of checks for clinician response rates may depend on clinicians' geographic location.

Trial registration

ClinicalTrials.gov ( NCT03351803 ), November 24, 2017.

2007 Background: NCCN now endorses BRCA founder mutation genetic testing (GT) via longitudinal studies in all Ashkenazi Jewish (AJ) individuals. The BRCA Founder OutReach (BFOR) study offers pre-GT online education with posttest engagement of primary care providers (PCPs). Methods: The study in 4 US cities enrolls those age > 25 with > 1 AJ grandparent. Participants enroll online with chatbot and video education, have GT at local centers, receive results from their PCP or BFOR staff, and are surveyed 12 weeks post disclosure and annually for 5 years. Univariate analyses and multivariable (MV) logistic regression models were used to evaluate characteristics associated with not completing GT, selecting PCP to disclose GT, and positive GT. Results: As of January 2020, 4754 participants consented (77.5% female, median age 51); 37.7% never previously considered GT. Cancer family histories (FHx) were 56.4% low risk (LR), 36.4% high risk (HR), and 7.2% had a familial mutation (FM). To date, 3658 participants (76.9%) completed and 677 (14.2%) did not complete GT; the remainder are pending. Only 34.8% of participants selected PCP to disclose GT, and 42.6% of PCPs agreed. Of the 124 mutation carriers (3.4%) identified, 60.5% had a FM. At the 12-week survey, 65.4% of mutation carriers planned to proceed with recommended screening or scheduled risk reducing surgery; 3.5% of those with negative GT and HR FHx reported further GT. Satisfaction was high (mean 9.58/10, SD 1.12) and unrelated to result (p>.05). Conclusions: A digital model for founder mutation testing engaged those with LR FHx and no prior experience with GT. Older participants were more likely to complete the study. Males were less likely to enroll but more likely to carry mutations. The majority of those who tested positive had a FM. A minority of results were disclosed by PCPs. Continued follow up is needed to determine long term outcomes. [Table: see text]

Purpose

This study aimed to evaluate uptake and follow-up using internet-assisted population genetic testing (GT) for BRCA1/2 Ashkenazi Jewish founder pathogenic variants (AJPVs).

Methods

Across 4 cities in the United States, from December 2017 to March 2020, individuals aged ≥25 years with ≥1 Ashkenazi Jewish grandparent were offered enrollment. Participants consented and enrolled online with chatbot and video education, underwent BRCA1/2 AJPV GT, and chose to receive results from their primary care provider (PCP) or study staff. Surveys were conducted at baseline, at 12 weeks, and annually for 5 years.

Results

A total of 5193 participants enrolled and 4109 (79.1%) were tested (median age = 54, female = 77.1%). Upon enrollment, 35.1% of participants selected a PCP to disclose results, and 40.5% of PCPs agreed. Of those tested, 138 (3.4%) were AJPV heterozygotes of whom 21 (15.2%) had no significant family history of cancer, whereas 86 (62.3%) had a known familial pathogenic variant. At 12 weeks, 85.5% of participants with AJPVs planned increased cancer screening; only 3.7% with negative results and a significant family history reported further testing.

Conclusion

Although continued follow-up is needed, internet-enabled outreach can expand access to targeted GT using a medical model. Observed challenges for population genetic screening efforts include recruitment barriers, improving PCP engagement, and increasing uptake of additional testing when indicated.