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日本吉特曼综合征患者中噻嗪类敏感型钠氯共转运体基因的四个新突变。

Four novel mutations in the thiazide-sensitive Na-Cl co-transporter gene in Japanese patients with Gitelman's syndrome.

作者信息

Maki Nobuki, Komatsuda Atsushi, Wakui Hideki, Ohtani Hiroshi, Kigawa Akihiko, Aiba Namiko, Hamai Keiko, Motegi Mutsuhito, Yamaguchi Akihiko, Imai Hirokazu, Sawada Ken-ichi

机构信息

Third Department of Internal Medicine, Akita University School of Medicine, Akita, Japan.

出版信息

Nephrol Dial Transplant. 2004 Jul;19(7):1761-6. doi: 10.1093/ndt/gfh239. Epub 2004 Apr 6.

Abstract

BACKGROUND

Gitelman's syndrome (GS) is an autosomal recessive disorder resulting from inactivating mutations in the thiazide-sensitive Na-Cl co-transporter (NCCT) gene. To date, almost 90 mutations have been identified. It is possible that there is a population-specific distribution of mutations. In this study, we analysed mutations in the NCCT gene of seven Japanese patients with GS.

METHODS

Peripheral blood mononuclear cells were isolated from patients with GS, their family members and healthy control subjects. A mutation analysis of the NCCT gene was performed completely by direct automated sequencing of polymerase chain reaction-amplified DNA products. In patients with a deletion or splice site mutation, we undertook cDNA sequence analysis.

RESULTS

We identified nine mutations. Five of them [c.185C>T (Thr60Met), c.1712C>T (Ala569Val), c.1930C>T (Arg642Cys), c.2552T>A (Leu849His) and c.1932delC] have been reported in Japanese patients, but not in GS patients from other ethnic groups. The remaining four mutations [c.7A>T (Met1Leu), c.1181_1186+20del26, c.1811_1812delAT and IVS16+1G>A] were novel. In cDNA derived from a patient with c.1181_1186+20del26, a deletion of exon 9 and a frameshift at the start of exon 10 were observed. In cDNA derived from patients with IVS16+1G>A, an additional 96 bp insertion between exons 16 and 17 was observed. Six out of seven patients were compound heterozygotes, and the remaining one carried a single heterozygous mutation.

CONCLUSIONS

We found four novel mutations in the NCCT gene in seven Japanese patients with GS. Moreover, our study suggests that the distribution of mutations in the NCCT gene in Japanese GS patients potentially differs from that in other populations.

摘要

背景

吉特林综合征(GS)是一种常染色体隐性疾病,由噻嗪类敏感型钠氯协同转运体(NCCT)基因的失活突变引起。迄今为止,已鉴定出近90种突变。突变可能存在人群特异性分布。在本研究中,我们分析了7例日本GS患者的NCCT基因中的突变。

方法

从GS患者、其家庭成员和健康对照者中分离外周血单个核细胞。通过对聚合酶链反应扩增的DNA产物进行直接自动测序,对NCCT基因进行完整的突变分析。对于存在缺失或剪接位点突变的患者,我们进行了cDNA序列分析。

结果

我们鉴定出9种突变。其中5种[c.185C>T(Thr60Met)、c.1712C>T(Ala569Val)、c.1930C>T(Arg642Cys)、c.2552T>A(Leu849His)和c.1932delC]已在日本患者中报道,但在其他种族的GS患者中未报道。其余4种突变[c.7A>T(Met1Leu)、c.1181_1186+20del26、c.1811_1812delAT和IVS16+1G>A]是新发现的。在来自c.1181_1186+20del26患者的cDNA中,观察到外显子9缺失和外显子10起始处的移码。在来自IVS16+1G>A患者的cDNA中,观察到外显子16和17之间额外插入了96 bp。7例患者中有6例为复合杂合子,其余1例携带单一杂合突变。

结论

我们在7例日本GS患者的NCCT基因中发现了4种新突变。此外,我们的研究表明,日本GS患者中NCCT基因突变的分布可能与其他人群不同。

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