• 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

III型家族性醛固酮增多症

Familial hyperaldosteronism type III.

作者信息

Monticone S, Tetti M, Burrello J, Buffolo F, De Giovanni R, Veglio F, Williams T A, Mulatero P

机构信息

Division of Internal Medicine and Hypertension Unit, Department of Medical Sciences, University of Torino, Torino, Italy.

Unit of Internal Medicine and Angiology, Department of Internal Medicine, Riccione Hospital, AUSL, Romagna, Italy.

出版信息

J Hum Hypertens. 2017 Dec;31(12):776-781. doi: 10.1038/jhh.2017.34. Epub 2017 Apr 27.

DOI:10.1038/jhh.2017.34
PMID:28447626
Abstract

Primary aldosteronism is the most common form of endocrine hypertension. This disorder comprises both sporadic and familial forms. Four familial forms of primary aldosteronism (FH-I to FH-IV) have been described. FH-III is caused by germline mutations in KCNJ5, encoding the potassium channel Kir3.4 (also called GIRK4). These mutations alter the selectivity filter of the channel and lead to abnormal ion currents with loss of potassium selectivity, sodium influx and consequent increased intracellular calcium that causes excessive aldosterone biosynthesis. To date, eleven families have been reported, carrying six different mutations. Although the clinical features are variable, FH-III patients often display severe hyperaldosteronism with an early onset, associated with hypokalemia and diabetes insipidus-like symptoms. In most cases FH-III patients are resistant to pharmacological therapy and require bilateral adrenalectomy to control symptoms. In the present manuscript, we review the genetics and pathological basis of FH-III, the diagnostic work-up, clinical features and therapeutic management. Finally, we will describe a new case of FH-III of an Italian patient carrying a Gly151Arg mutation.

摘要

原发性醛固酮增多症是内分泌性高血压最常见的形式。这种疾病包括散发性和家族性两种形式。原发性醛固酮增多症的四种家族形式(FH-I至FH-IV)已被描述。FH-III是由KCNJ5基因的种系突变引起的,该基因编码钾通道Kir3.4(也称为GIRK4)。这些突变改变了通道的选择性过滤器,导致异常离子电流,失去钾选择性,钠内流,进而导致细胞内钙增加,引起醛固酮生物合成过多。迄今为止,已报道了11个家族,携带6种不同的突变。尽管临床特征各不相同,但FH-III患者常表现为严重的醛固酮增多症,起病早,伴有低钾血症和尿崩症样症状。在大多数情况下,FH-III患者对药物治疗耐药,需要双侧肾上腺切除术来控制症状。在本手稿中,我们回顾了FH-III的遗传学和病理基础、诊断检查、临床特征和治疗管理。最后,我们将描述一例携带Gly151Arg突变的意大利FH-III患者的新病例。

相似文献

1
Familial hyperaldosteronism type III.III型家族性醛固酮增多症
J Hum Hypertens. 2017 Dec;31(12):776-781. doi: 10.1038/jhh.2017.34. Epub 2017 Apr 27.
2
Familial hyperaldosteronism type III a novel case and review of literature.家族性醛固酮增多症 III 型:一例新病例及文献复习。
Rev Endocr Metab Disord. 2019 Mar;20(1):27-36. doi: 10.1007/s11154-018-9481-0.
3
KCNJ5 mutations in European families with nonglucocorticoid remediable familial hyperaldosteronism.KCNJ5 突变与欧洲非糖皮质激素可治愈型家族性醛固酮增多症家系相关。
Hypertension. 2012 Feb;59(2):235-40. doi: 10.1161/HYPERTENSIONAHA.111.183996. Epub 2011 Dec 27.
4
Role of KCNJ5 in familial and sporadic primary aldosteronism.KCNJ5 在家族性和散发性原发性醛固酮增多症中的作用。
Nat Rev Endocrinol. 2013 Feb;9(2):104-12. doi: 10.1038/nrendo.2012.230. Epub 2012 Dec 11.
5
Genetics of primary aldosteronism.原发性醛固酮增多症的遗传学
Front Horm Res. 2014;43:70-8. doi: 10.1159/000360870. Epub 2014 Jun 10.
6
A case of severe hyperaldosteronism caused by a de novo mutation affecting a critical salt bridge Kir3.4 residue.一例由影响关键盐桥Kir3.4残基的新发突变引起的严重醛固酮增多症病例。
J Clin Endocrinol Metab. 2015 Jan;100(1):E114-8. doi: 10.1210/jc.2014-3636.
7
a Novel Y152C KCNJ5 mutation responsible for familial hyperaldosteronism type III.一个导致家族性醛固酮增多症 III 型的新型 Y152C KCNJ5 突变。
J Clin Endocrinol Metab. 2013 Nov;98(11):E1861-5. doi: 10.1210/jc.2013-2428. Epub 2013 Sep 13.
8
Somatic and inherited mutations in primary aldosteronism.原发性醛固酮增多症中的体细胞突变和遗传性突变。
J Mol Endocrinol. 2017 Jul;59(1):R47-R63. doi: 10.1530/JME-17-0035. Epub 2017 Apr 11.
9
GENETICS IN ENDOCRINOLOGY: The expanding genetic horizon of primary aldosteronism.内分泌学中的遗传学:原发性醛固酮增多症不断扩展的遗传视野。
Eur J Endocrinol. 2018 Mar;178(3):R101-R111. doi: 10.1530/EJE-17-0946. Epub 2018 Jan 18.
10
New insights into aldosterone-producing adenomas and hereditary aldosteronism: mutations in the K+ channel KCNJ5.醛固酮瘤和遗传性醛固酮增多症的新见解:钾通道 KCNJ5 的突变。
Curr Opin Nephrol Hypertens. 2013 Mar;22(2):141-7. doi: 10.1097/MNH.0b013e32835cecf8.

引用本文的文献

1
Relevance of in Pathologies of Heart Disease.在心脏病病理学中的相关性。
Int J Mol Sci. 2023 Jun 29;24(13):10849. doi: 10.3390/ijms241310849.
2
Who should be screened for primary aldosteronism? A comprehensive review of current evidence.哪些人应该接受原发性醛固酮增多症筛查?当前证据的全面综述。
J Clin Hypertens (Greenwich). 2022 Sep;24(9):1194-1203. doi: 10.1111/jch.14558.
3
Progress on Genetic Basis of Primary Aldosteronism.原发性醛固酮增多症的遗传基础研究进展

本文引用的文献

1
Disordered zonal and cellular CYP11B2 enzyme expression in familial hyperaldosteronism type 3.3型家族性醛固酮增多症中肾上腺带区和细胞的CYP11B2酶表达紊乱。
Mol Cell Endocrinol. 2017 Jan 5;439:74-80. doi: 10.1016/j.mce.2016.10.025. Epub 2016 Oct 25.
2
A Novel Phenotype of Familial Hyperaldosteronism Type III: Concurrence of Aldosteronism and Cushing's Syndrome.家族性III型原发性醛固酮增多症的一种新表型:醛固酮增多症与库欣综合征并存
J Clin Endocrinol Metab. 2016 Nov;101(11):4290-4297. doi: 10.1210/jc.2016-1504. Epub 2016 Jul 12.
3
Functional characterization of two novel germline mutations of the KCNJ5 gene in hypertensive patients without primary aldosteronism but with ACTH-dependent aldosterone hypersecretion.
Biomedicines. 2021 Nov 17;9(11):1708. doi: 10.3390/biomedicines9111708.
4
Transcriptomics, Epigenetics, and Metabolomics of Primary Aldosteronism.原发性醛固酮增多症的转录组学、表观遗传学和代谢组学
Cancers (Basel). 2021 Nov 8;13(21):5582. doi: 10.3390/cancers13215582.
5
Diseases caused by mutations in the Na/K pump α1 gene .由 Na/K 泵 α1 基因突变引起的疾病。
Am J Physiol Cell Physiol. 2021 Aug 1;321(2):C394-C408. doi: 10.1152/ajpcell.00059.2021. Epub 2021 Jul 7.
6
Advances in Genomics Research of Blood Pressure Responses to Dietary Sodium and Potassium Intakes.膳食钠钾摄入与血压反应的基因组学研究进展。
Hypertension. 2021 Jul;78(1):4-15. doi: 10.1161/HYPERTENSIONAHA.121.16509. Epub 2021 May 17.
7
Overview of Monogenic Forms of Hypertension Combined With Hypokalemia.单基因形式高血压合并低钾血症概述
Front Pediatr. 2021 Jan 25;8:543309. doi: 10.3389/fped.2020.543309. eCollection 2020.
8
Mosaicism for KCNJ5 Causing Early-Onset Primary Aldosteronism due to Bilateral Adrenocortical Hyperplasia.KCNJ5 嵌合体导致双侧肾上腺皮质增生性早发性醛固酮增多症。
Am J Hypertens. 2020 Feb 22;33(2):124-130. doi: 10.1093/ajh/hpz172.
9
Primary Aldosteronism: KCNJ5 Mutations and Adrenocortical Cell Growth.原醛症:KCNJ5 突变与肾上腺皮质细胞生长。
Hypertension. 2019 Oct;74(4):809-816. doi: 10.1161/HYPERTENSIONAHA.119.13476. Epub 2019 Aug 26.
10
Familial hyperaldosteronism type III a novel case and review of literature.家族性醛固酮增多症 III 型:一例新病例及文献复习。
Rev Endocr Metab Disord. 2019 Mar;20(1):27-36. doi: 10.1007/s11154-018-9481-0.
KCNJ5基因两个新的种系突变在无原发性醛固酮增多症但有促肾上腺皮质激素依赖性醛固酮分泌过多的高血压患者中的功能特征。
Clin Endocrinol (Oxf). 2016 Dec;85(6):845-851. doi: 10.1111/cen.13132. Epub 2016 Jul 12.
4
Study Heterogeneity and Estimation of Prevalence of Primary Aldosteronism: A Systematic Review and Meta-Regression Analysis.研究原发性醛固酮增多症的异质性和患病率估计:系统评价和荟萃回归分析。
J Clin Endocrinol Metab. 2016 Jul;101(7):2826-35. doi: 10.1210/jc.2016-1472. Epub 2016 May 12.
5
The Management of Primary Aldosteronism: Case Detection, Diagnosis, and Treatment: An Endocrine Society Clinical Practice Guideline.原发性醛固酮增多症的管理:病例检出、诊断和治疗:内分泌学会临床实践指南。
J Clin Endocrinol Metab. 2016 May;101(5):1889-916. doi: 10.1210/jc.2015-4061. Epub 2016 Mar 2.
6
An Update on Familial Hyperaldosteronism.家族性醛固酮增多症的最新进展
Horm Metab Res. 2015 Dec;47(13):941-6. doi: 10.1055/s-0035-1564166. Epub 2015 Oct 7.
7
A case of severe hyperaldosteronism caused by a de novo mutation affecting a critical salt bridge Kir3.4 residue.一例由影响关键盐桥Kir3.4残基的新发突变引起的严重醛固酮增多症病例。
J Clin Endocrinol Metab. 2015 Jan;100(1):E114-8. doi: 10.1210/jc.2014-3636.
8
Understanding primary aldosteronism: impact of next generation sequencing and expression profiling.了解原发性醛固酮增多症:新一代测序和表达谱分析的影响
Mol Cell Endocrinol. 2015 Jan 5;399:311-20. doi: 10.1016/j.mce.2014.09.015. Epub 2014 Sep 18.
9
Discordant genotype-phenotype correlation in familial hyperaldosteronism type III with KCNJ5 gene mutation: a patient report and review of the literature.伴有KCNJ5基因突变的III型家族性醛固酮增多症中基因型与表型的不一致相关性:一例患者报告及文献综述
Horm Res Paediatr. 2014;82(2):138-42. doi: 10.1159/000358197. Epub 2014 May 10.
10
Pharmacology and pathophysiology of mutated KCNJ5 found in adrenal aldosterone-producing adenomas.在肾上腺产生醛固酮的腺瘤中发现的突变 KCNJ5 的药理学和病理生理学。
Endocrinology. 2014 Apr;155(4):1353-62. doi: 10.1210/en.2013-1944. Epub 2014 Feb 7.