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A Parametric Model for Analyzing Anticipation in Genetically Predisposed Families

Author

Listed:
  • Larsen Klaus

    (Hvidovre Hospital)

  • Petersen Janne

    (Hvdiovre Hospital)

  • Bernstein Inge

    (Hvidovre Hospital)

  • Nilbert Mef

    (Hvidovre Hospital)

Abstract

Anticipation, i.e. a decreasing age-at-onset in subsequent generations has been observed in a number of genetically triggered diseases. The impact of anticipation is generally studied in affected parent-child pairs. These analyses are restricted to pairs in which both individuals have been affected and are sensitive to right truncation of the data. We propose a normal random effects model that allows for right-censored observations and includes covariates, and draw statistical inference based on the likelihood function.

Suggested Citation

  • Larsen Klaus & Petersen Janne & Bernstein Inge & Nilbert Mef, 2009. "A Parametric Model for Analyzing Anticipation in Genetically Predisposed Families," Statistical Applications in Genetics and Molecular Biology, De Gruyter, vol. 8(1), pages 1-11, June.
    • Handle: RePEc:bpj:sagmbi:v:8:y:2009:i:1:n:26
    DOI: 10.2202/1544-6115.1424
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    References listed on IDEAS

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    1. Daniel Rabinowitz & Qiong Yang, 1999. "Testing for Age-at-Onset Anticipation with Affected Parent-Child Pairs," Biometrics, The International Biometric Society, vol. 55(3), pages 834-838, September.
    2. van der Laan, Mark J., 1996. "Nonparametric Estimation of the Bivariate Survival Function with Truncated Data," Journal of Multivariate Analysis, Elsevier, vol. 58(1), pages 107-131, July.
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    Cited by:

    1. Philip S. Boonstra & Bhramar Mukherjee & Jeremy M. G. Taylor & Mef Nilbert & Victor Moreno & Stephen B. Gruber, 2011. "Bayesian Modeling for Genetic Anticipation in Presence of Mutational Heterogeneity: A Case Study in Lynch Syndrome," Biometrics, The International Biometric Society, vol. 67(4), pages 1627-1637, December.

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