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Details

Autor(en) / Beteiligte
Titel
The value of genomic sequencing in complex pediatric neurological disorders: a discrete choice experiment
Ist Teil von
  • Genetics in medicine, 2021-01, Vol.23 (1), p.155-162
Ort / Verlag
United States: Elsevier Inc
Erscheinungsjahr
2021
Quelle
EZB-FREE-00999 freely available EZB journals
Beschreibungen/Notizen
  • To estimate the value of genomic sequencing for complex pediatric neurological disorders of suspected genetic origin. 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. 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. 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.
Sprache
Englisch
Identifikatoren
ISSN: 1098-3600
eISSN: 1530-0366
DOI: 10.1038/s41436-020-00949-2
Titel-ID: cdi_proquest_miscellaneous_2437120838

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