TY - JOUR
T1 - The value of genomic sequencing in complex pediatric neurological disorders
T2 - a discrete choice experiment
AU - Goranitis, Ilias
AU - Best, Stephanie
AU - Stark, Zornitza
AU - Boughtwood, Tiffany
AU - Christodoulou, John
PY - 2020/8/25
Y1 - 2020/8/25
N2 - Purpose: To estimate the value of genomic sequencing for complex pediatric neurological disorders of suspected genetic origin. Methods: A discrete choice experiment (DCE) was undertaken to elicit societal preferences and values. A Bayesian D-efficient and explicit partial profile design was used. The design included 72 choice tasks, split across six blocks, with eight attributes (three overlapping per choice task) and three alternatives. Choice data were analyzed using a panel error component mixed logit model and a latent class model. Preference heterogeneity according to personal socioeconomic, demographic, and attitudinal characteristics was explored using linear and fractional logistic regressions. Results: In total, 820 members of the Australian public were recruited. Statistically significant preferences were identified across all eight DCE attributes. We estimated that society on average would be willing to pay AU$5650 more (95% confidence interval [CI]: AU$5500 to $5800) (US$3955 [95% CI: US$3850 to $4060]) for genomic sequencing relative to standard care. Preference heterogeneity was identified for some personal characteristics. Conclusion: On average, society highly values all diagnostic, process, clinical, and nonclinical components of personal utility. To ensure fair prioritization of genomics, decision makers need to consider the wide range of risks and benefits associated with genomic information.
AB - Purpose: To estimate the value of genomic sequencing for complex pediatric neurological disorders of suspected genetic origin. Methods: A discrete choice experiment (DCE) was undertaken to elicit societal preferences and values. A Bayesian D-efficient and explicit partial profile design was used. The design included 72 choice tasks, split across six blocks, with eight attributes (three overlapping per choice task) and three alternatives. Choice data were analyzed using a panel error component mixed logit model and a latent class model. Preference heterogeneity according to personal socioeconomic, demographic, and attitudinal characteristics was explored using linear and fractional logistic regressions. Results: In total, 820 members of the Australian public were recruited. Statistically significant preferences were identified across all eight DCE attributes. We estimated that society on average would be willing to pay AU$5650 more (95% confidence interval [CI]: AU$5500 to $5800) (US$3955 [95% CI: US$3850 to $4060]) for genomic sequencing relative to standard care. Preference heterogeneity was identified for some personal characteristics. Conclusion: On average, society highly values all diagnostic, process, clinical, and nonclinical components of personal utility. To ensure fair prioritization of genomics, decision makers need to consider the wide range of risks and benefits associated with genomic information.
KW - children
KW - genomics
KW - neurodevelopmental disorders
KW - personal utility
KW - preferences
UR - http://www.scopus.com/inward/record.url?scp=85089726199&partnerID=8YFLogxK
U2 - 10.1038/s41436-020-00949-2
DO - 10.1038/s41436-020-00949-2
M3 - Article
JO - Genetics in medicine : official journal of the American College of Medical Genetics
JF - Genetics in medicine : official journal of the American College of Medical Genetics
SN - 1098-3600
ER -