Eicher J D, Powers N R, Miller L L, Akshoomoff N, Amaral D G, Bloss C S, Libiger O, Schork N J, Darst B F, Casey B J, Chang L, Ernst T, Frazier J, Kaufmann W E, Keating B, Kenet T, Kennedy D, Mostofsky S, Murray S S, Sowell E R, Bartsch H, Kuperman J M, Brown T T, Hagler D J, Dale A M, Jernigan T L, St Pourcain B, Davey Smith G, Ring S M, Gruen J R
Department of Genetics, Yale University, New Haven, CT, USA.
Genes Brain Behav. 2013 Nov;12(8):792-801. doi: 10.1111/gbb.12085. Epub 2013 Oct 9.
Written and verbal languages are neurobehavioral traits vital to the development of communication skills. Unfortunately, disorders involving these traits-specifically reading disability (RD) and language impairment (LI)-are common and prevent affected individuals from developing adequate communication skills, leaving them at risk for adverse academic, socioeconomic and psychiatric outcomes. Both RD and LI are complex traits that frequently co-occur, leading us to hypothesize that these disorders share genetic etiologies. To test this, we performed a genome-wide association study on individuals affected with both RD and LI in the Avon Longitudinal Study of Parents and Children. The strongest associations were seen with markers in ZNF385D (OR = 1.81, P = 5.45 × 10(-7) ) and COL4A2 (OR = 1.71, P = 7.59 × 10(-7) ). Markers within NDST4 showed the strongest associations with LI individually (OR = 1.827, P = 1.40 × 10(-7) ). We replicated association of ZNF385D using receptive vocabulary measures in the Pediatric Imaging Neurocognitive Genetics study (P = 0.00245). We then used diffusion tensor imaging fiber tract volume data on 16 fiber tracts to examine the implications of replicated markers. ZNF385D was a predictor of overall fiber tract volumes in both hemispheres, as well as global brain volume. Here, we present evidence for ZNF385D as a candidate gene for RD and LI. The implication of transcription factor ZNF385D in RD and LI underscores the importance of transcriptional regulation in the development of higher order neurocognitive traits. Further study is necessary to discern target genes of ZNF385D and how it functions within neural development of fluent language.
书面语言和口头语言是对沟通技能发展至关重要的神经行为特征。不幸的是,涉及这些特征的障碍——特别是阅读障碍(RD)和语言障碍(LI)——很常见,会阻碍受影响个体发展足够的沟通技能,使他们面临学业、社会经济和精神方面不良后果的风险。RD和LI都是复杂的特征,经常同时出现,这使我们推测这些障碍有共同的遗传病因。为了验证这一点,我们在雅芳亲子纵向研究中对同时患有RD和LI的个体进行了全基因组关联研究。在ZNF385D(比值比=1.81,P=5.45×10⁻⁷)和COL4A2(比值比=1.71,P=7.59×10⁻⁷)的标记中观察到最强的关联。NDST4内的标记单独与LI显示出最强的关联(比值比=1.827,P=1.40×10⁻⁷)。我们在儿科影像神经认知遗传学研究中使用接受性词汇测量方法重复了ZNF385D的关联(P=0.00245)。然后,我们使用16条纤维束的扩散张量成像纤维束体积数据来研究重复标记的影响。ZNF385D是两个半球总体纤维束体积以及全脑体积的预测指标。在此,我们提供证据表明ZNF385D是RD和LI的候选基因。转录因子ZNF385D在RD和LI中的作用强调了转录调控在高阶神经认知特征发展中的重要性。需要进一步研究以识别ZNF385D的靶基因及其在流利语言神经发育中的作用机制。