A Marginal Likelihood Approach for Estimating Penetrance from Kin‐Cohort Designs

Nilanjan Chatterjee and Sholom Wacholder

Biometrics, 2001, vol. 57, issue 1, 245-252

Abstract: Summary. The kin‐cohort design is a promising alternative to traditional cohort or case‐control designs for estimating penetrance of an identified rare autosomal mutation. In this design, a suitably selected sample of participants provides genotype and detailed family history information on the disease of interest. To estimate penetrance of the mutation, we consider a marginal likelihood approach that is computationally simple to implement, more flexible than the original analytic approach proposed by Wacholder et al. (1998, American Journal of Epidemiology148, 623–629), and more robust than the likelihood approach considered by Gail et al. (1999, Genetic Epidemiology16, 15–39) to presence of residual familial correlation. We study the trade‐off between robustness and efficiency using simulation experiments. The method is illustrated by analysis of the data from the Washington Ashkenazi Study.

Date: 2001
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