Szatmari Peter, Mérette Chantal, Emond Claudia, Zwaigenbaum Lonnie, Jones Marshall B, Maziade Michel, Roy Marc-Andre, Palmour Roberta
Department of Psychiatry and Behavioural Neurosciences, McMaster University, Hamilton, Ontario, Canada.
Am J Med Genet B Neuropsychiatr Genet. 2008 Jan 5;147B(1):3-9. doi: 10.1002/ajmg.b.30561.
The objective of this article is to decompose the level of functioning phenotype in autism to see if it can be conceptualized as two simpler, but still familial, dimensional phenotypes of language and non-verbal IQ. We assembled 80 sibpairs with either autism, Asperger syndrome or atypical autism. To see whether the familial correlation on language scores was accounted for by the familial correlation on non-verbal IQ, residual language scores were calculated for each member of the sibpair based on a multiple regression equation using their IQ score as an explanatory or independent variable and controlling for the age and gender of the affected individual. These residual scores were then used to calculate intraclass correlations between affected sibs. This process was repeated using IQ as the dependent variable and language as a covariate. Within affected individuals there was a strong relation between non-verbal IQ (as measured by the Leiter performance scale) and language (as measured by the Vineland Communication Scale). In addition, there was familial correlation between sibs on both measures. Evidence of familial aggregation on both non-verbal IQ and language remained even after partialling out the effect of the covariates by regression analysis and by generalized estimating equation. These findings suggest that non-verbal IQ and language in PDD may arise from independent genetic mechanisms. The implications of this finding for linkage analysis and for identifying genetically informative phenotypes are discussed.
本文的目的是剖析自闭症患者的功能表现型水平,以探究其是否可被概念化为语言和非言语智商这两种更简单但仍具有家族性的维度表现型。我们收集了80对患有自闭症、阿斯伯格综合征或非典型自闭症的同胞对。为了确定语言分数的家族相关性是否由非言语智商的家族相关性所致,我们根据多元回归方程,以智商分数作为解释变量或自变量,并控制受影响个体的年龄和性别,为每对同胞对中的成员计算残余语言分数。然后,这些残余分数被用于计算受影响同胞之间的组内相关性。以智商为因变量、语言为协变量重复这一过程。在受影响个体中,非言语智商(由莱特操作量表测量)与语言(由文兰社会成熟量表测量)之间存在很强的相关性。此外,同胞对在这两项测量上均存在家族相关性。即使通过回归分析和广义估计方程剔除协变量的影响后,非言语智商和语言的家族聚集证据依然存在。这些发现表明,广泛性发育障碍中的非言语智商和语言可能源于独立的遗传机制。本文还讨论了这一发现对连锁分析以及识别具有遗传信息的表现型的意义。