• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

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.

DOI:10.1093/ndt/gfh239
PMID:15069170
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。

相似文献

1
Four novel mutations in the thiazide-sensitive Na-Cl co-transporter gene in Japanese patients with Gitelman's syndrome.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。
Nephrol Dial Transplant. 2004 Jul;19(7):1761-6. doi: 10.1093/ndt/gfh239. Epub 2004 Apr 6.
2
Novel mutations in the SLC12A3 gene causing Gitelman's syndrome in Swedes.导致瑞典人患吉特曼综合征的SLC12A3基因新突变。
DNA Seq. 2007 Oct;18(5):395-9. doi: 10.1080/10425170701400456.
3
Problems in diagnosing atypical Gitelman's syndrome presenting with normomagnesaemia.诊断表现为正常血镁的非典型 Gitelman 综合征时存在的问题。
Clin Endocrinol (Oxf). 2010 Feb;72(2):272-6. doi: 10.1111/j.1365-2265.2009.03649.x. Epub 2009 Jun 8.
4
Novel NCCT gene mutations as a cause of Gitelman's syndrome and a systematic review of mutant and polymorphic NCCT alleles.新型NCCT基因突变是吉特曼综合征的病因及对NCCT突变和多态性等位基因的系统综述。
Kidney Blood Press Res. 2002;25(6):354-62. doi: 10.1159/000068695.
5
[Gitelman's syndrome: an important differential diagnosis of hypokalemia].吉特林综合征:低钾血症的重要鉴别诊断
Dtsch Med Wochenschr. 2003 May 30;128(22):1225-8. doi: 10.1055/s-2003-39456.
6
Subjects heterozygous for genetic loss of function of the thiazide-sensitive cotransporter have reduced blood pressure.噻嗪类敏感共转运体功能遗传缺失的杂合子受试者血压降低。
Hum Mol Genet. 2008 Feb 1;17(3):413-8. doi: 10.1093/hmg/ddm318. Epub 2007 Nov 1.
7
Analysis of renal tubular electrolyte transporter genes in seven patients with hypokalemic metabolic alkalosis.7例低钾血症性代谢性碱中毒患者肾小管电解质转运体基因分析
Kidney Int. 2003 Sep;64(3):808-16. doi: 10.1046/j.1523-1755.2003.00163.x.
8
Gitelman's syndrome: report of one case.吉特曼综合征:1例报告。
Acta Paediatr Taiwan. 2008 Jan-Feb;49(1):31-4.
9
[Clinical significance of thiazide-sensitive Na-Cl cotransporter gene by mutational analysis].[噻嗪类敏感型钠氯共转运体基因突变分析的临床意义]
Rinsho Byori. 2007 Apr;55(4):338-43.
10
Novel mutations in thiazide-sensitive Na-Cl cotransporter gene of patients with Gitelman's syndrome.吉特曼综合征患者噻嗪类敏感型钠氯共转运体基因的新突变
J Am Soc Nephrol. 2000 Jan;11(1):65-70. doi: 10.1681/ASN.V11165.

引用本文的文献

1
Unique genetic presentation of Gitelman syndrome in a Hispanic patient: Case report.一名西班牙裔患者吉特曼综合征的独特基因表现:病例报告
SAGE Open Med Case Rep. 2025 Jun 20;13:2050313X251351547. doi: 10.1177/2050313X251351547. eCollection 2025.
2
Genetic and Biological Effects of , a Sodium-Chloride Cotransporter, in Gitelman Syndrome and Diabetic Kidney Disease.氯化钠共转运体在吉特曼综合征和糖尿病肾病中的遗传及生物学效应
Front Genet. 2022 May 3;13:799224. doi: 10.3389/fgene.2022.799224. eCollection 2022.
3
Frequent SLC12A3 mutations in Chinese Gitelman syndrome patients: structure and function disorder.
中国吉特曼综合征患者中常见的SLC12A3突变:结构与功能紊乱
Endocr Connect. 2022 Jan 27;11(1):e210262. doi: 10.1530/EC-21-0262.
4
The First Korean Case of Aberrant Skipping of Two Exons Detected by RNA Splicing Analysis.
Case Rep Nephrol Dial. 2021 Jul 19;11(2):210-213. doi: 10.1159/000517139. eCollection 2021 May-Aug.
5
Clinical and Genetic Features in 31 Serial Chinese Children With Gitelman Syndrome.31例中国吉特曼综合征患儿的临床及遗传学特征
Front Pediatr. 2021 Apr 29;9:544925. doi: 10.3389/fped.2021.544925. eCollection 2021.
6
Clinical Characteristics and Gene Mutation Analysis of the Chinese Han Population with Gitelman Syndrome: 3 Case Reports and a Literature Review.中国汉族吉特林综合征患者的临床特征及基因突变分析:3例报告并文献复习
Case Rep Med. 2020 Oct 24;2020:6263721. doi: 10.1155/2020/6263721. eCollection 2020.
7
A Family with Gitelman Syndrome with Asymptomatic Phenotypes while Carrying Reported SLC12A3 Mutations.一个携带已报道的SLC12A3突变但具有无症状表型的吉特曼综合征家族。
Case Rep Nephrol Dial. 2020 Jul 13;10(2):71-78. doi: 10.1159/000507845. eCollection 2020 May-Aug.
8
Two mutations in the thiazide-sensitive NaCl co-transporter gene in a Romanian Gitelman syndrome patient: case report.一名罗马尼亚吉特曼综合征患者的噻嗪类敏感型氯化钠协同转运蛋白基因中的两个突变:病例报告
Ther Clin Risk Manag. 2018 Jan 22;14:149-155. doi: 10.2147/TCRM.S150483. eCollection 2018.
9
A Pedigree with c.179 Cytosine to Threonine Missense Mutation of SLC12A3 Gene Presenting Gitelman's Syndrome.一个携带SLC12A3基因c.179位点胞嘧啶至苏氨酸错义突变的家系,表现为吉特曼综合征。
Electrolyte Blood Press. 2016 Jun;14(1):16-9. doi: 10.5049/EBP.2016.14.1.16. Epub 2016 Jun 30.
10
Analysis of mutations of two Gitelman syndrome family SLC12A3 genes and proposed treatments using Chinese medicine.两个吉特曼综合征家族SLC12A3基因的突变分析及中医药治疗方案
Chin J Integr Med. 2017 Jun;23(6):461-468. doi: 10.1007/s11655-016-2461-x. Epub 2016 Jan 29.