Müller Bent, Boltze Johannes, Czepezauer Ivonne, Hesse Volker, Wilcke Arndt, Kirsten Holger
Fraunhofer Institute for Cell Therapy and Immunology, Leipzig, Germany.
Fraunhofer Research Institution for Marine Biotechnology, Department of Medical Cell Technology, Lübeck, Germany.
Genet Mol Biol. 2018 Jan-Mar;41(1):41-49. doi: 10.1590/1678-4685-GMB-2017-0165. Epub 2018 Feb 19.
An increasing number of genetic variants involved in dyslexia development were discovered during the last years, yet little is known about the molecular functional mechanisms of these SNPs. In this study we investigated whether dyslexia candidate SNPs have a direct, disease-specific effect on local expression levels of the assumed target gene by using a differential allelic expression assay. In total, 12 SNPs previously associated with dyslexia and related phenotypes were suitable for analysis. Transcripts corresponding to four SNPs were sufficiently expressed in 28 cell lines originating from controls and a family affected by dyslexia. We observed a significant effect of rs600753 on expression levels of DYX1C1 in forward and reverse sequencing approaches. The expression level of the rs600753 risk allele was increased in the respective seven cell lines from members of the dyslexia family which might be due to a disturbed transcription factor binding sites. When considering our results in the context of neuroanatomical dyslexia-specific findings, we speculate that this mechanism may be part of the pathomechanisms underlying the dyslexia-specific brain phenotype. Our results suggest that allele-specific DYX1C1 expression levels depend on genetic variants of rs600753 and contribute to dyslexia. However, these results are preliminary and need replication.
在过去几年中,发现了越来越多与诵读困难症发展相关的基因变异,但对于这些单核苷酸多态性(SNP)的分子功能机制却知之甚少。在本研究中,我们通过使用差异等位基因表达分析,研究了诵读困难症候选SNP是否对假定靶基因的局部表达水平具有直接的、疾病特异性的影响。总共有12个先前与诵读困难症及相关表型相关的SNP适合进行分析。对应于四个SNP的转录本在来自对照组和一个受诵读困难症影响的家族的28个细胞系中充分表达。在正向和反向测序方法中,我们观察到rs600753对DYX1C1表达水平有显著影响。在来自诵读困难症家族成员的各自七个细胞系中,rs600753风险等位基因的表达水平有所增加,这可能是由于转录因子结合位点受到干扰所致。结合神经解剖学上诵读困难症特异性的研究结果来考虑我们的结果,我们推测这种机制可能是诵读困难症特异性脑表型潜在发病机制的一部分。我们的结果表明,等位基因特异性的DYX1C1表达水平取决于rs600753的基因变异,并与诵读困难症有关。然而,这些结果是初步的,需要重复验证。