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在中国一个常染色体显性多囊肾病家族中发现了一种新的PKD1突变。

A new PKD1 mutation discovered in a Chinese family with autosomal polycystic kidney disease.

作者信息

Wang Zhendi, Wang Yang, Xiong Jing

机构信息

Department of Urology, Union Hospital, Tongji Medical College, Huazhong University of Science & Technology, Wuhan, China.

出版信息

Kidney Blood Press Res. 2014;39(1):1-8. doi: 10.1159/000355772. Epub 2014 Apr 30.

DOI:10.1159/000355772
PMID:24821069
Abstract

BACKGROUND/AIMS: Autosomal-dominant polycystic kidney disease (ADPKD), a heterogeneous genetic disorder characterized by massive kidney enlargement and progressive chronic kidney disease, is due to abnormal proliferation of renal tubular epithelium. ADPKD is known to be caused by mutations in PKD1 and PKD2 genes.

METHODS

In the present study, the mutation analysis of PKD genes was performed in a new Chinese family with ADPKD using Long-Range (LR) PCR sequencing and targeted next-generation sequencing (targeted DNA-HiSeq).

RESULTS

A unique 28 bp deletion (c.12605_12632del28) in exon 46 of the PKD1 gene was identified in two affected family members by LR PCR method, but not in any unaffected relatives or unrelated controls. Higher accuracy and less missing detection presented in LR PCR method compared with targeted DNA-HiSeq. This mutation c.12605_12632del28 (p.Arg4202ProextX146) resulted in a delayed termination of amino acid code, and was highly speculated pathogenic in this ADPKD family. Moreover, this newly identified frame-shift change was compared to the PKD gene database, but no similar mutation was yet reported.

CONCLUSION

A novel frame-shift mutation, c. 12605_12632del28, in the PKD1 gene was found in a Chinese ADPKD family. All evidence available suggested that it might be the mutation responsible for the disease in that family.

摘要

背景/目的:常染色体显性遗传性多囊肾病(ADPKD)是一种异质性遗传病,其特征为肾脏显著增大和进行性慢性肾病,由肾小管上皮异常增殖引起。已知ADPKD由PKD1和PKD2基因突变所致。

方法

在本研究中,采用长片段(LR)PCR测序和靶向新一代测序(靶向DNA-HiSeq)技术,对一个新的中国ADPKD家系进行PKD基因突变分析。

结果

通过LR PCR方法在两名患病家族成员中鉴定出PKD1基因第46外显子存在一个独特的28 bp缺失(c.12605_12632del28),而在任何未患病亲属或无关对照中均未发现。与靶向DNA-HiSeq相比,LR PCR方法具有更高的准确性和更低的漏检率。该突变c.12605_12632del28(p.Arg4202ProextX146)导致氨基酸编码提前终止,高度推测在这个ADPKD家系中具有致病性。此外,将这个新鉴定的移码变化与PKD基因数据库进行比较,尚未发现类似突变。

结论

在中国一个ADPKD家系中发现了PKD1基因的一个新的移码突变,即c.12605_12632del28。所有现有证据表明,它可能是该家系致病的突变。

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引用本文的文献

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The Clinical Manifestation and Management of Autosomal Dominant Polycystic Kidney Disease in China.中国常染色体显性遗传性多囊肾病的临床表现与治疗
Kidney Dis (Basel). 2016 Oct;2(3):111-119. doi: 10.1159/000449030. Epub 2016 Oct 6.