title: Assessing risk for the Tourette spectrum of disorders among first-degree relatives of probands with Tourette syndrome.
abstract: Previous studies have indicated that genetic investigations of Tourette syndrome (TS) should focus on a phenotype that includes not only TS, but chronic tics (CT) and obsessive-compulsive disorder (OCD) as well. These studies have shown that sex may play a role in determining which of the disorders in the TS spectrum is expressed in a susceptible individual. Female relatives of TS probands far more often express OCD, while male relatives more often express TS or CT. Data from the Yale Family Study of TS were used to model risk to first-degree relatives of probands with TS for a variety of TS disease phenotypes. Risk to relatives was modeled using multivariate Cox regression analysis, a method appropriate for assessing risk when there is correlation among disease onsets. This is the first known application of this method to family data. The study identified two proband characteristics that increase the risk for disease onset among both male and female relatives for all TS spectrum disorders, lending credence to the hypothesis that TS spectrum disorders share a common etiology. These were a relatively younger age-at-onset, and no experience of simple motor tics. The predictive ability of two additional factors varied by both sex and disease phenotype. These characteristics, i.e., proband onset with compulsive tics, and proband onset with range, appear to increase risk primarily in female relatives, and for the OCD part of the spectrum.
authors.affiliation: Department of Epidemiology, Harvard Program in Psychiatric Epidemiology and Biostatistics, Harvard School of Public Health, Boston, Massachusetts, USA.
authors.forename: S L
authors.initials: SL
authors.lastname: Santangelo
authors.forename: D L
authors.initials: DL
authors.lastname: Pauls
authors.forename: P W
authors.initials: PW
authors.lastname: Lavori
authors.forename: J M
authors.initials: JM
authors.lastname: Goldstein
authors.forename: S V
authors.initials: SV
authors.lastname: Faraone
authors.forename: M T
authors.initials: MT
authors.lastname: Tsuang
mesh_terms: D000328:Adult; D005260:Female; D006801:Humans; D008297:Male; D008957:Models, Genetic; D010641:Phenotype; D018570:Risk Assessment; D005879:Tourette Syndrome / Q000235:genetics* / Q000503:physiopathology