ArticleJournal of the American Statistical Association2013
Frailty Models for Familial Risk with Application to Breast Cancer.
Article in Journal of the American Statistical Association, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.
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Who cites it
12 citing papers in PubMed.
- Computationally efficient methods for estimating phenome-wide coheritability of multi-type phenotypes using biobank data.Communications biology · 2025Article
- Application of the Joint Frailty Copula Model for Analyzing Time to Relapse and Time to Death of Women with Cervical Cancer.International journal of women's health · 2023Article
- Semiparametric regression analysis of bivariate censored events in a family study of Alzheimer's disease.Biostatistics (Oxford, England) · 2022Article
- Variation in cancer risk among families with genetic susceptibility.Genetic epidemiology · 2021Article
- Practical implementation of frailty models in Mendelian risk prediction.Genetic epidemiology · 2020Article
- Estimating the age at onset distribution of the asymptomatic stage of a genetic disease based on pedigree data.Statistical methods in medical research · 2020Article
- Bayesian and Frequentist Analytical Approaches Using Log-Normal and Gamma Frailty Parametric Models for Breast Cancer Mortality.Computational and mathematical methods in medicine · 2020Article
- SEMIPARAMETRIC TRANSFORMATION MODELS WITH MULTILEVEL RANDOM EFFECTS FOR CORRELATED DISEASE ONSET IN FAMILIES.Statistica Sinica · 2019Article
- Second-Order Estimating Equations for Clustered Current Status Data from Family Studies Using Response-Dependent Sampling.Statistics in biosciences · 2018Article
- Nonparametric Adjustment for Measurement Error in Time-to-Event Data: Application to Risk Prediction Models.Journal of the American Statistical Association · 2018Article
- Can multiple SNP testing in BRCA2 and BRCA1 female carriers be used to improve risk prediction models in conjunction with clinical assessment?BMC medical informatics and decision making · 2014Article
- Calibrated predictions for multivariate competing risks models.Lifetime data analysis · 2014Article
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3 authors.
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Abstract
In evaluating familial risk for disease we have two main statistical tasks: assessing the probability of carrying an inherited genetic mutation conferring higher risk; and predicting the absolute risk of developing diseases over time, for those individuals whose mutation status is known. Despite substantial progress, much remains unknown about the role of genetic and environmental risk factors, about the sources of variation in risk among families that carry high-risk mutations, and about the sources of familial aggregation beyond major Mendelian effects. These sources of heterogeneity contribute substantial variation in risk across families. In this paper we present simple and efficient methods for accounting for this variation in familial risk assessment. Our methods are based on frailty models. We implemented them in the context of generalizing Mendelian models of cancer risk, and compared our approaches to others that do not consider heterogeneity across families. Our extensive simulation study demonstrates that when predicting the risk of developing a disease over time conditional on carrier status, accounting for heterogeneity results in a substantial improvement in the area under the curve of the receiver operating characteristic. On the other hand, the improvement for carriership probability estimation is more limited. We illustrate the utility of the proposed approach through the analysis of BRCA1 and BRCA2 mutation carriers in the Washington Ashkenazi Kin-Cohort Study of Breast Cancer.
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