title: A fully nonparametric estimator of the marginal survival function based on case-control clustered age-at-onset data.
abstract: Consider a popular case-control family study where individuals with a disease under study (case probands) and individuals who do not have the disease (control probands) are randomly sampled from a well-defined population. Possibly right-censored age at onset and disease status are observed for both probands and their relatives. For example, case probands are men diagnosed with prostate cancer, control probands are men free of prostate cancer, and the prostate cancer history of the fathers of the probands is also collected. Inherited genetic susceptibility, shared environment, and common behavior lead to correlation among the outcomes within a family. In this article, a novel nonparametric estimator of the marginal survival function is provided. The estimator is defined in the presence of intra-cluster dependence, and is based on consistent smoothed kernel estimators of conditional survival functions. By simulation, it is shown that the proposed estimator performs very well in terms of bias. The utility of the estimator is illustrated by the analysis of case-control family data of early onset prostate cancer. To our knowledge, this is the first article that provides a fully nonparametric marginal survival estimator based on case-control clustered age-at-onset data.
authors.affiliation: Department of Statistics and Operations Research, School of Mathematical Sciences, Tel Aviv University, Tel Aviv 69978 Israel gorfinem@post.tau.ac.il.
authors.forename: Malka
authors.initials: M
authors.lastname: Gorfine
authors.affiliation: Faculty of Industrial Engineering and Management, Technion - Israel Institute of Technology, Technion City, Haifa 3200003 Israel.
authors.forename: Nadia
authors.initials: N
authors.lastname: Bordo
authors.affiliation: Division of Public Health Sciences, Fred Hutchinson Cancer Research Center, 1100 Fairview Ave. N., P.O. Box 19024, Seattle, WA 98109-1024, USA.
authors.forename: Li
authors.initials: L
authors.lastname: Hsu
mesh_terms: D016022:Case-Control Studies; D003627:Data Interpretation, Statistical; D006801:Humans; D016019:Survival Analysis
keywords: Case–control; Family study; Multivariate survival; Nonparametric estimator; Smoothed kernel estimator