Stein Catherine M, Truitt Barbara, Deng Fenghua, Ciesla Allison Avrich, Qiu Feiyou, Joseph Peronne, Raghavendra Rekha, Fondran Jeremy, Igo Robert P, Tag Jessica, Freebairn Lisa, Taylor H Gerry, Lewis Barbara A, Iyengar Sudha K
Departments of aEpidemiology and Biostatistics bPsychological Sciences cPediatrics dGenetics eCenter for Proteomics and Bioinformatics, Case Western Reserve University, Cleveland, Ohio, USA.
Psychiatr Genet. 2014 Oct;24(5):191-200. doi: 10.1097/YPG.0000000000000045.
Speech sound disorder (SSD) is one of the most common communication disorders, with a prevalence rate of 16% at 3 years of age, and an estimated 3.8% of children still presenting speech difficulties at 6 years of age. Several studies have identified promising associations between communication disorders and genes in brain and neuronal pathways; however, there have been few studies focusing on SSD and its associated endophenotypes. On the basis of the hypothesis that neuronal genes may influence endophenotypes common to communication disorders, we focused on three genes related to brain and central nervous system functioning: the dopamine D2 receptor (DRD2) gene, the arginine-vasopressin receptor 1a (AVPR1A) gene, and the microcephaly-associated protein gene (ASPM).
We examined the association of these genes with key endophenotypes of SSD - phonological memory measured through multisyllabic and nonword repetition, vocabulary measured using the Expressive One Word Picture Vocabulary Test and Peabody Picture Vocabulary Test, and reading decoding measured using the Woodcock Reading Mastery Tests Revised - as well as with the clinical phenotype of SSD. We genotyped tag single nucleotide polymorphisms in these genes and examined 498 individuals from 180 families.
These data show that several single nucleotide polymorphisms in all three genes were associated with phonological memory, vocabulary, and reading decoding, with P less than 0.05. Notably, associations in AVPR1A (rs11832266) were significant after multiple testing correction. Gene-level tests showed that DRD2 was associated with vocabulary, ASPM with vocabulary and reading decoding, and AVPR1A with all three endophenotypes.
Endophenotypes common to SSD, language impairment, and reading disability are all associated with these neuronal pathway genes.
言语声音障碍(SSD)是最常见的交流障碍之一,3岁儿童的患病率为16%,估计6岁时仍有3.8%的儿童存在言语困难。多项研究已确定交流障碍与大脑和神经元通路中的基因之间存在有前景的关联;然而,很少有研究关注SSD及其相关的内表型。基于神经元基因可能影响交流障碍共有的内表型这一假设,我们聚焦于三个与大脑和中枢神经系统功能相关的基因:多巴胺D2受体(DRD2)基因、精氨酸加压素受体1a(AVPR1A)基因和小头畸形相关蛋白基因(ASPM)。
我们研究了这些基因与SSD的关键内表型——通过多音节和非单词重复测量的语音记忆、使用表达性单字图画词汇测试和皮博迪图画词汇测试测量的词汇量、使用修订版伍德库克阅读能力测验测量的阅读解码——以及与SSD临床表型之间的关联。我们对这些基因中的标签单核苷酸多态性进行基因分型,并研究了来自180个家庭的498名个体。
这些数据表明,所有三个基因中的几个单核苷酸多态性与语音记忆、词汇量和阅读解码相关,P值小于0.05。值得注意的是,在进行多重检验校正后,AVPR1A(rs11832266)中的关联具有显著性。基因水平测试表明,DRD2与词汇量相关,ASPM与词汇量和阅读解码相关,AVPR1A与所有三种内表型相关。
SSD、语言障碍和阅读障碍共有的内表型均与这些神经元通路基因相关。