title: A competing risks model for correlated data based on the subdistribution hazard.
abstract: Family-based follow-up study designs are important in epidemiology as they enable investigations of disease aggregation within families. Such studies are subject to methodological complications since data may include multiple endpoints as well as intra-family correlation. The methods herein are developed for the analysis of age of onset with multiple disease types for family-based follow-up studies. The proposed model expresses the marginalized frailty model in terms of the subdistribution hazards (SDH). As with Pipper and Martinussen's (Scand J Stat 30:509-521, 2003) model, the proposed multivariate SDH model yields marginal interpretations of the regression coefficients while allowing the correlation structure to be specified by a frailty term. Further, the proposed model allows for a direct investigation of the covariate effects on the cumulative incidence function since the SDH is modeled rather than the cause specific hazard. A simulation study suggests that the proposed model generally offers improved performance in terms of bias and efficiency when a sufficient number of events is observed. The proposed model also offers type I error rates close to nominal. The method is applied to a family-based study of breast cancer when death in absence of breast cancer is considered a competing risk.
authors: Department of Epidemiology and Biostatistics, Schulich School of Medicine & Dentistry, The University of Western Ontario, London, ON N6A 5C1, Canada. Stephanie.Dixon@schulich.uwo.ca
authors: Stephanie N
authors: SN
authors: Dixon
authors: Gerarda A
authors: GA
authors: Darlington
authors: Anthony F
authors: AF
authors: Desmond
references: Hum Mutat. 2003 Aug;22(2):121-8
references: 12872252
references: Lifetime Data Anal. 2000 Jun;6(2):141-56
references: 10851839
references: Stat Med. 1993 Apr 30;12(8):737-51
references: 8516591
references: Lifetime Data Anal. 2007 Dec;13(4):471-80
references: 17768681
references: Stat Med. 2005 Jun 15;24(11):1713-23
references: 15724232
references: Stat Med. 2006 Dec 30;25(24):4267-78
references: 16960919
references: Biometrics. 2008 Mar;64(1):172-9
references: 17680835
references: Am J Hum Genet. 1995 Jul;57(1):1-7
references: 7611277
references: Lifetime Data Anal. 1995;1(2):205-20
references: 9385102
references: Biometrics. 1992 Sep;48(3):795-806
references: 1420842
references: Stat Med. 2004 Feb 15;23(3):369-88
references: 14748034
references: J Clin Oncol. 2005 Dec 1;23(34):8629-35
references: 16314625
references: Stat Med. 2007 Nov 10;26(25):4657-78
references: 17348081