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异常表型患者平衡染色体重排的基因组研究。

Genomic Investigation of Balanced Chromosomal Rearrangements in Patients with Abnormal Phenotypes.

作者信息

Simioni Milena, Artiguenave François, Meyer Vincent, Sgardioli Ilária C, Viguetti-Campos Nilma L, Lopes Monlleó Isabella, Maciel-Guerra Andréa T, Steiner Carlos E, Gil-da-Silva-Lopes Vera L

机构信息

Department of Medical Genetics, School of Medical Sciences, University of Campinas (UNICAMP), Campinas, Brazil.

Centre National de Génotypage, Evry, France.

出版信息

Mol Syndromol. 2017 Jun;8(4):187-194. doi: 10.1159/000477084. Epub 2017 Jun 1.

Abstract

Balanced chromosomal rearrangements (BCR) are associated with abnormal phenotypes in approximately 6% of balanced translocations and 9.4% of balanced inversions. Abnormal phenotypes can be caused by disruption of genes at the breakpoints, deletions, or positional effects. Conventional cytogenetic techniques have a limited resolution and do not enable a thorough genetic investigation. Molecular techniques applied to BCR carriers can contribute to the characterization of this type of chromosomal rearrangement and to the phenotype-genotype correlation. Fifteen individuals among 35 with abnormal phenotypes and BCR were selected for further investigation by molecular techniques. Chromosomal rearrangements involved 11 reciprocal translocations, 3 inversions, and 1 balanced insertion. Array genomic hybridization (AGH) was performed and genomic imbalances were detected in 20% of the cases, 1 at a rearrangement breakpoint and 2 further breakpoints in other chromosomes. Alterations were further confirmed by FISH and associated with the phenotype of the carriers. In the analyzed cases not showing genomic imbalances by AGH, next-generation sequencing (NGS), using whole genome libraries, prepared following the Illumina TruSeq DNA PCR-Free protocol (Illumina®) and then sequenced on an Illumina HiSEQ 2000 as 150-bp paired-end reads, was done. The NGS results suggested breakpoints in 7 cases that were similar or near those estimated by karyotyping. The genes overlapping 6 breakpoint regions were analyzed. Follow-up of BCR carriers would improve the knowledge about these chromosomal rearrangements and their consequences.

摘要

平衡染色体重排(BCR)在约6%的平衡易位和9.4%的平衡倒位中与异常表型相关。异常表型可能由断点处基因的破坏、缺失或位置效应引起。传统的细胞遗传学技术分辨率有限,无法进行全面的基因研究。应用于BCR携带者的分子技术有助于对这类染色体重排进行特征描述,并有助于表型-基因型相关性研究。在35例具有异常表型和BCR的个体中,选择了15例通过分子技术进行进一步研究。染色体重排包括11例相互易位、3例倒位和1例平衡插入。进行了阵列基因组杂交(AGH),在20%的病例中检测到基因组不平衡,其中1例在重排断点处,另外2例在其他染色体的进一步断点处。通过荧光原位杂交(FISH)进一步证实了这些改变,并将其与携带者的表型相关联。在AGH未显示基因组不平衡的分析病例中,采用Illumina TruSeq DNA PCR-Free方案(Illumina®)制备全基因组文库,然后在Illumina HiSEQ 2000上进行150碱基对双端测序,进行了二代测序(NGS)。NGS结果表明,7例病例中的断点与核型分析估计的断点相似或相近。对6个断点区域重叠的基因进行了分析。对BCR携带者的随访将增进对这些染色体重排及其后果的了解。

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