遗传学
遗传性球形红细胞增多症
RNA剪接
生物
桑格测序
基因
外显子
外显子跳跃
分子生物学
先证者
质粒
选择性拼接
中国仓鼠卵巢细胞
基因组DNA
突变
核糖核酸
细胞培养
作者
Hongyan Liu,Jiaqiang Huang,Yinghai Jiang,Liangjie Guo,Haijun Xiao,Hongdan Wang
出处
期刊:PubMed
日期:2020-01-10
卷期号:37 (1): 17-20
标识
DOI:10.3760/cma.j.issn.1003-9406.2020.01.005
摘要
To explore the genetic basis of a pedigree affected with hereditary spherocytosis.Peripheral blood samples were collected from 17 members of the pedigree. Genomic DNA of the proband was subjected to next generation sequencing. Candidate variant was validated by co-segregation analysis. pCAS2(c.5798+1G) and pCAS2(c.5798+1A) plasmids were constructed by homologous recombination and transfected into 293T cells. Reverse transcription PCR, TA cloning and Sanger sequencing were used to analyze the effect of candidate variant on splicing. Meanwhile, peripheral blood RNAs were extracted to analyze the effect of candidate variant on splicing in vivo.The proband was found to carry a c.5798+1G>A variant of the SPTB gene. The variant has co-segregated with the phenotype in the pedigree. In vitro and in vivo splicing experiments confirmed that the mutation has significantly affected the splicing, resulting in shift of reading frame and produced a premature termination codon.The novel c.5798+1G>A variant of the SPTB gene probably underlies the pathogenesis of hereditary spherocytosis in this pedigree.
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