Stein Catherine M, Schick James H, Gerry Taylor H, Shriberg Lawrence D, Millard Christopher, Kundtz-Kluge Amy, Russo Karlie, Minich Nori, Hansen Amy, Freebairn Lisa A, Elston Robert C, Lewis Barbara A, Iyengar Sudha K
Department of Epidemiology and Biostatistics, Rammelkamp Center for Research, Case Western Reserve University, Cleveland, OH 44109, USA.
Am J Hum Genet. 2004 Feb;74(2):283-97. doi: 10.1086/381562. Epub 2004 Jan 20.
Speech-sound disorder (SSD) is a complex behavioral disorder characterized by speech-sound production errors associated with deficits in articulation, phonological processes, and cognitive linguistic processes. SSD is prevalent in childhood and is comorbid with disorders of language, spelling, and reading disability, or dyslexia. Previous research suggests that developmental problems in domains associated with speech and language acquisition place a child at risk for dyslexia. Recent genetic studies have identified several candidate regions for dyslexia, including one on chromosome 3 segregating in a large Finnish pedigree. To explore common genetic influences on SSD and reading, we examined linkage for several quantitative traits to markers in the pericentrometric region of chromosome 3 in 77 families ascertained through a child with SSD. The quantitative scores measured several processes underlying speech-sound production, including phonological memory, phonological representation, articulation, receptive and expressive vocabulary, and reading decoding and comprehension skills. Model-free linkage analysis was followed by identification of sib pairs with linkage and construction of core shared haplotypes. In our multipoint analyses, measures of phonological memory demonstrated the strongest linkage (marker D3S2465, P=5.6 x 10(-5), and marker D3S3716, P=6.8 x 10(-4)). Tests for single-word decoding also demonstrated linkage (real word reading: marker D3S2465, P=.004; nonsense word reading: marker D3S1595, P=.005). The minimum shared haplotype in sib pairs with similar trait values spans 4.9 cM and is bounded by markers D3S3049 and D3S3045. Our results suggest that domains common to SSD and dyslexia are pleiotropically influenced by a putative quantitative trait locus on chromosome 3.
语音障碍(SSD)是一种复杂的行为障碍,其特征在于与发音、语音过程和认知语言过程缺陷相关的语音产生错误。SSD在儿童期很常见,并且与语言、拼写障碍以及阅读障碍或诵读困难共病。先前的研究表明,与语音和语言习得相关领域的发育问题使儿童有患诵读困难的风险。最近的基因研究已经确定了几个诵读困难的候选区域,包括在一个大型芬兰家系中分离的位于3号染色体上的一个区域。为了探究对SSD和阅读的共同遗传影响,我们在通过患有SSD的儿童确定的77个家庭中,检查了几个数量性状与3号染色体着丝粒周围区域标记的连锁关系。这些数量得分测量了语音产生背后的几个过程,包括语音记忆、语音表征、发音、接受性和表达性词汇,以及阅读解码和理解技能。在无模型连锁分析之后,识别出具有连锁关系的同胞对并构建核心共享单倍型。在我们的多点分析中,语音记忆测量显示出最强的连锁关系(标记D3S2465,P = 5.6 x 10^(-5),标记D3S3716,P = 6.8 x 10^(-4))。单字解码测试也显示出连锁关系(实词阅读:标记D3S2465,P =.004;假词阅读:标记D3S1595,P =.005)。具有相似性状值的同胞对中的最小共享单倍型跨度为4.9厘摩,由标记D3S3049和D3S3045界定。我们的结果表明,SSD和诵读困难共有的领域受到3号染色体上一个假定的数量性状基因座的多效性影响。