A new statistical test for age-of-onset anticipation: Application to bipolar disorder

Jian Huang, Veronica Vieland

Research output: Journal article publicationJournal articleAcademic researchpeer-review

26 Citations (Scopus)


The discovery of expanding microsatellite repeat mechanisms for a variety of diseases has spurred renewed interest in testing for genetic anticipation in complex diseases. However, standard statistical methods can be shown to be inappropriate for this purpose. We derive a new statistical test for comparing mean age of onset in parents with mean age of onset in children, based on a random sample of affected parent-child pairs. This test incorporates the right-truncation of the age-of-onset distributions and utilizes age-at-interview information. We illustrate the use of the test by application to bipolar affective disorder data. In this example, the correct p-value is 0.014 whereas the results based on the biased estimates give a p- value of 0.0001 [McInnis et al., 1993]. We also briefly consider the effects of ascertainment bias when parent-child pairs are obtained from samples collected for purposes of linkage analysis.
Original languageEnglish
Pages (from-to)1091-1096
Number of pages6
JournalGenetic Epidemiology
Issue number6
Publication statusPublished - 1 Dec 1997
Externally publishedYes


  • Affected parent-child pair
  • Age-of-onset
  • Ascertainment bias
  • Genetic anticipation
  • Right-truncation
  • Trinucleotide repeat expansions

ASJC Scopus subject areas

  • Epidemiology
  • Genetics(clinical)


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