Participant Satisfaction With a Preference-Setting Tool for the Return of Individual Research Results in Pediatric Genomic Research

Abstract

The perceived benefit of return of individual research results (IRRs) in accordance to participants' preferences in genomic biobank research is unclear. We developed an online preference-setting tool for return of IRRs based on the preventability and severity of a condition, which included an opt-out option for IRRs for mental illness, developmental disorders, childhood-onset degenerative conditions, and adult-onset conditions. Parents of patients <18 years of age at Boston Children's Hospital were randomized to the hypothetical scenario that their child was enrolled in one of four biobanks with different policies for IRRs to receive (a) "None," (b) "All," (c) "Binary"--choice to receive all or none, and (d) "Granular"--use the preference-setting tool to choose categories of IRRs. Parents were given a hypothetical IRRs report for their child. The survey was sent to 11,391 parents and completed by 2,718. The Granular group was the most satisfied with the process, biobank, and hypothetical IRRs received. The None group was least satisfied and least likely to agree that the biobank was beneficial (p < .001). The response to the statement that the biobank was harmful was not different between groups. Our data suggest that the ability to designate preferences leads to greater satisfaction and may increase biobank participation.

Experience of parents receiving results from a quality‐of‐life study in pediatric advanced cancer: A report from the PediQUEST study

“…The spectrum of emotions elicited highlights the importance of personalizing approaches and developing thoughtful dissemination strategies. Notably, presenting granular options for dissemination has improved parent satisfaction …”

Newborn Sequencing in Genomic Medicine and Public Health

“…48,49 Determining criteria for disclosure of information will be a challenge for clinicians and policymakers and will require development of decisional supports to help parents determine the information they want to learn. 50 52 The benefits of detecting true positives must therefore be balanced against the magnitude of harms.…”

Family health history reporting is sensitive to small changes in wording

“…Detailed methods and primary findings are reported elsewhere. 11,12 Briefly, parents of Boston Children’s Hospital (BCH) patients were invited to participate in a study about receiving genomic research results about their children. The study was designed to explore whether satisfaction with enrollment in a hypothetical biobank was associated with having an ability to designate preferences regarding what results to receive.…”

“…Data from the three randomization arms who were not given instructions to consider extended family are pooled, given no differences in study procedures prior to reporting personal and family history and given no differences in demographics. The sample size was established to test primary study hypotheses about differences in satisfaction, 11 but post-hoc analyses suggest 99% power to detect small differences (i.e., Cohen’s d=0.2 for t-tests, 0.1 for chi-squared tests) by randomization status in analyses comparing randomization arms at a significance level of p=0.01. All statistical tests were two-sided, with significance set at p=0.01 to account for the large number of analyses.…”