title: Societal preferences for the return of incidental findings from clinical genomic sequencing: a discrete-choice experiment.
abstract: BACKGROUND
  An important challenge with the application of next-generation sequencing technology is the possibility of uncovering incidental genomic findings. A paucity of evidence on personal utility for incidental findings has hindered clinical guidelines. Our objective was to estimate personal utility for complex information derived from incidental genomic findings.
  
  
  METHODS
  We used a discrete-choice experiment to evaluate participants' personal utility for the following attributes: disease penetrance, disease treatability, disease severity, carrier status and cost. Study participants were drawn from the Canadian public. We analyzed the data with a mixed logit model.
  
  
  RESULTS
  In total, 1200 participants completed our questionnaire (available in English and French). Participants valued receiving information about high-penetrance disorders but expressed disutility for receiving information on low-penetrance disorders. The average willingness to pay was $445 (95% confidence interval [CI] $322-$567) to receive incidental findings in a scenario where clinicians returned information about high-penetrance, medically treatable disorders, but only 66% of participants (95% CI 63%-71%) indicated that they would choose to receive information in that scenario. On average, participants placed an important value ($725, 95% CI $600-$850) on having a choice about what type of findings they would receive, including receipt of information about high-penetrance, treatable disorders or receipt of information about high-penetrance disorders with or without available treatment. The predicted uptake of that scenario was 76% (95% CI 72%-79%).
  
  
  CONCLUSIONS
  Most participants valued receiving incidental findings, but personal utility depended on the type of finding, and not all participants wanted to receive incidental results, regardless of the potential health implications. These results indicate that to maximize benefit, participant-level preferences should inform the decision about whether to return incidental findings.
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont. dregier@bccrc.ca.
authors.forename: Dean A
authors.initials: DA
authors.lastname: Regier
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: Stuart J
authors.initials: SJ
authors.lastname: Peacock
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: Reka
authors.initials: R
authors.lastname: Pataky
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: Kimberly
authors.initials: K
authors.lastname: van der Hoek
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: Gail P
authors.initials: GP
authors.lastname: Jarvik
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: Jeffrey
authors.initials: J
authors.lastname: Hoch
authors.affiliation: Canadian Centre for Applied Research in Cancer Control (Regier, Peacock, Pataky, van der Hoek), Cancer Control Research, BC Cancer Agency, Vancouver, BC; School of Population and Public Health (Regier, Peacock, Pataky), University of British Columbia, Vancouver, BC; Department of Medicine (Medical Genetics) (Jarvik), Department of Genome Sciences (Jarvik) and Pharmaceutical Outcomes Research and Policy Program, Department of Pharmacy (Veenstra), University of Washington, Seattle, Wash.; Pharmacoeconomics Research Unit (Hoch), Cancer Care Ontario, Toronto, Ont.; Canadian Centre for Applied Research in Cancer Control (Hoch), Toronto, Ont.
authors.forename: David
authors.initials: D
authors.lastname: Veenstra
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D002170:Canada; D002755:Choice Behavior; D003430:Cross-Sectional Studies; D030881:Disclosure; D005260:Female; D020022:Genetic Predisposition to Disease / Q000523:psychology*; D005820:Genetic Testing; D016678:Genome; D017048:Health Care Costs; D006579:Heterozygote; D006801:Humans; D033162:Incidental Findings; D008297:Male; D008875:Middle Aged; D057240:Patient Preference; D019683:Penetrance; D017422:Sequence Analysis, DNA; D012720:Severity of Illness Index; D011795:Surveys and Questionnaires; D055815:Young Adult