How do citizens balance the benefits and burdens of newborn screening? A choice experiment. F. A. Miller1, R. Z. Hayeems1,2, Y. Bombard3, C. Cressman1, C. J. Barg1, J. C. Carroll4, B. Wilson5, J. Little5, D. Avard6, J. Allanson7, P. Chakraborty7, Y. Giguere8, D. A. Regier9 1) Institute of Health Policy, Management & Evaluation, University of Toronto, Toronto, Canada; 2) Institute for Clinical and Evaluative Sciences, Toronto, Canada; 3) Yale University, Department of Public Health and Epidemiology; Center for Health Policy and Outcomes, Memorial Sloan Kettering Cancer Center, New York, USA; 4) Department of Family and Community Medicine, Mount Sinai Hospital, University of Toronto, Canada; 5) Department of Epidemiology and Community Medicine, University of Ottawa, Canada; 6) Centre for Genomics and Policy, Department of Human Genetics, McGill University, Montreal, Canada; 7) Department of Genetics, Childrens Hospital of Eastern Ontario, and Department of Pediatrics, University of Ottawa, Canada; 8) Department of Medical Biology, Centre Hospitalier, Universitaire de Quebec, University of Laval, Canada; 9) Canadian Centre for Applied Research in Cancer Control, BC Cancer Agency, Vancouver, Canada.
Introduction: Newborn screening (NBS) programs have expanded in recent years with increased debate about the appropriate balance between their benefits and burdens. How the public values and trades-off the effects of NBS, including those impacting affected infants and their families (clinical, informational) and those imposed on the rest of the population (false positive results, overdiagnosis) can influence NBS policy and uptake. Methods: In January 2013, a bilingual (French and English) Internet survey was administered to a representative sample of Canadians to assess preferences for NBS across 5 attributes using a discrete choice experiment (DCE). Introduced through a detailed training module, the attributes included: clinical benefits of improved health (none, moderate, significant), earlier time to diagnosis (1 week to 4 years), reproductive risk information (available, not), false positive (FP) results (1-40 per affected baby), and overdiagnosed (OD) infants (0-2 per affected baby). Data were analyzed with a mixed logit model to identify preference heterogeneity among respondents. Results: The survey participation rate was 94%; 1220 completed responses met quality criteria (52% completion rate). Respondents prioritized clinical benefits over all other attributes; all respondents positively valued reproductive risk information, while 65% positively valued earlier diagnosis (35% negatively valued this effect). All respondents had a negative preference for FP results, and 98% negatively valued OD (2% positively valued this effect). Nevertheless, respondents were willing to accept large numbers of FP results and some OD infants to achieve moderate clinical benefit for one affected baby (33 FP, 2 OD), and higher numbers to achieve significant clinical benefit (49 FP, 3 OD). Conclusions: We report a novel approach to exploring public preferences for the complex trade-offs obliged by population screening programs. Our findings point to broad public understanding of the negative implications of false positive results and overdiagnosis, along with a willingness to tolerate some burdens in the pursuit of clinical benefits.
You may contact the first author (during and after the meeting) at