Modeling of successive cancer risks in Lynch syndrome families in the presence of competing risks using copulas

Biometrics. 2017 Mar;73(1):271-282. doi: 10.1111/biom.12561. Epub 2016 Jul 5.


In this article, we propose an association model to estimate the penetrance (risk) of successive cancers in the presence of competing risks. The association between the successive events is modeled via a copula and a proportional hazards model is specified for each competing event. This work is motivated by the analysis of successive cancers for people with Lynch Syndrome in the presence of competing risks. The proposed inference procedure is adapted to handle missing genetic covariates and selection bias, induced by the data collection protocol of the data at hand. The performance of the proposed estimation procedure is evaluated by simulations and its use is illustrated with data from the Colon Cancer Family Registry (Colon CFR).

Keywords: Ascertainment correction; Missing covariates; Penetrance function; Successive competing risks.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Analysis of Variance
  • Bias
  • Colonic Neoplasms
  • Colorectal Neoplasms, Hereditary Nonpolyposis / pathology*
  • Computer Simulation
  • Data Interpretation, Statistical*
  • Genetics
  • Humans
  • Proportional Hazards Models*
  • Registries
  • Risk