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Biostatistics 2:173-181 (2001)
© 2001 Oxford University Press

Referent sampling, family history and relative risk: the role of length-biased sampling

Ori Davidov and Marvin Zelen

Department of Statistics, University of Haifa, Haifa 31905, Israel. davidov{at}stat.haifa.ac.il
Department of Biostatistics, Harvard School of Public Health, and the Dana Farber Cancer Institute, 655 Huntington Avenue, Boston, MA 02115, USA. zelen{at}jimmy.harvard.edu

Familial risk of disease is often assessed using case control studies based on referent databases. A referent database is a collection of family histories of cases typically assembled as a result of one family member being diagnosed with disease. This sampling scheme is equivalent to sampling families proportional to their size. The larger the family, the greater the probability of finding the family in the referent registry. This phenomena is known as length-biased sampling. The consequence of this kind of sampling is to bias the regression estimate associated with family history. The estimate is typically inflated in comparison to what is true for the actual population.

Keywords: Case control study; Family history; Length biased sampling; Referent registries; Relative risk


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