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日本常染色体显性多囊肾病患者的下一代测序和多重连接依赖探针扩增突变分析。

Mutation analyses by next-generation sequencing and multiplex ligation-dependent probe amplification in Japanese autosomal dominant polycystic kidney disease patients.

作者信息

Mochizuki Toshio, Teraoka Atsuko, Akagawa Hiroyuki, Makabe Shiho, Akihisa Taro, Sato Masayo, Kataoka Hiroshi, Mitobe Michihiro, Furukawa Toru, Tsuchiya Ken, Nitta Kosaku

机构信息

Department of Medicine, Kidney Center, Tokyo Women's Medical University, Tokyo, Japan.

Clinical Research Division for Polycystic Kidney Disease, Department of Medicine, Kidney Center, Tokyo Women's Medical University, Tokyo, Japan.

出版信息

Clin Exp Nephrol. 2019 Aug;23(8):1022-1030. doi: 10.1007/s10157-019-01736-3. Epub 2019 Apr 15.

Abstract

BACKGROUND

Autosomal dominant polycystic kidney disease (ADPKD), one of the most common hereditary kidney diseases, causes gradual growth of cysts in the kidneys, leading to renal failure. Owing to the advanced technology of next-generation sequencing (NGS), genetic analyses of the causative genes PKD1 and PKD2 have been improved.

METHODS

We performed genetic analyses of 111 Japanese ADPKD patients using hybridization-based NGS and long-range (LR)-PCR-based NGS. Additionally, genetic analyses in exon 1 of PKD1 using Sanger sequencing because of an extremely low coverage of NGS and those using multiplex ligation-dependent probe amplification (MLPA) were performed.

RESULTS

The detection rate using NGS for 111 patients was 86.5%. One mutation in exon 1 of PKD1 and five deletions detected by MLPA were identified. When combined, the total detection rate was 91.9%.

CONCLUSION

Although NGS is useful, we propose the addition of Sanger sequencing for exon 1 of PKD1 and MLPA as indispensable for identifying mutations not detected by NGS.

摘要

背景

常染色体显性遗传性多囊肾病(ADPKD)是最常见的遗传性肾病之一,会导致肾脏囊肿逐渐增大,最终发展为肾衰竭。由于新一代测序(NGS)技术的进步,致病基因PKD1和PKD2的基因分析得到了改进。

方法

我们使用基于杂交的NGS和基于长片段(LR)-PCR的NGS对111例日本ADPKD患者进行了基因分析。此外,由于NGS在外显子1区域覆盖极低,我们使用桑格测序法对PKD1外显子1进行了基因分析,并进行了多重连接依赖探针扩增(MLPA)分析。

结果

111例患者使用NGS的检测率为86.5%。鉴定出PKD1外显子1中的一个突变和MLPA检测到的五个缺失。综合起来,总检测率为91.9%。

结论

尽管NGS很有用,但我们建议增加对PKD1外显子1的桑格测序和MLPA,这对于识别NGS未检测到的突变是必不可少的。

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