Suppr超能文献

吉特曼综合征中肾脏对钙和镁的处理

Renal calcium and magnesium handling in Gitelman syndrome.

作者信息

Reyes Jeremiah V, Medina Paul Mark B

机构信息

Biological Models Laboratory, Department of Biochemistry and Molecular Biology, College of Medicine, University of the Philippines Manila Ermita, Manila 1000, Philippines.

出版信息

Am J Transl Res. 2022 Jan 15;14(1):1-19. eCollection 2022.

Abstract

Gitelman syndrome (GS) is an autosomal recessive salt-losing tubulopathy caused by biallelic inactivating mutations in the SLC12A3 gene. This gene encodes the thiazide-sensitive sodium-chloride cotransporter (NCC) which is exclusively expressed in the distal convoluted tubules (DCT). GS patients classically present with hypokalemic metabolic alkalosis with hypocalciuria and hypomagnesemia. While hypokalemia and metabolic alkalosis are easily explained by effects of the genotypic defect in GS, the mechanisms by which hypomagnesemia and hypocalciuria develop in GS are poorly understood. In this review, we aim to achieve three major objectives. First, present a concise discussion about current understanding on physiologic calcium and magnesium handling in the DCT. Second, integrate expression data from studies on calciotropic and magnesiotropic proteins relevant to the GS disease state. Lastly, provide insights into the possible mechanisms of calcium-magnesium crosstalk relating to the co-occurrence of hypocalciuria and hypomagnesemia in GS models. Our analyses highlight specific areas of study that are valuable in elucidating possible molecular pathways of hypocalciuria and hypomagnesemia in GS.

摘要

吉特曼综合征(GS)是一种常染色体隐性遗传性失盐性肾小管病,由SLC12A3基因的双等位基因失活突变引起。该基因编码噻嗪类敏感的氯化钠共转运体(NCC),其仅在远曲小管(DCT)中表达。GS患者的典型表现为低钾血症性代谢性碱中毒伴低钙尿症和低镁血症。虽然低钾血症和代谢性碱中毒很容易用GS的基因型缺陷效应来解释,但GS中低镁血症和低钙尿症发生的机制却知之甚少。在本综述中,我们旨在实现三个主要目标。第一,简要讨论目前对远曲小管中钙和镁生理处理的认识。第二,整合与GS疾病状态相关的钙调节和镁调节蛋白研究的表达数据。最后,深入探讨在GS模型中与低钙尿症和低镁血症同时出现相关的钙镁相互作用的可能机制。我们的分析突出了在阐明GS中低钙尿症和低镁血症可能的分子途径方面有价值的特定研究领域。

相似文献

1
Renal calcium and magnesium handling in Gitelman syndrome.
Am J Transl Res. 2022 Jan 15;14(1):1-19. eCollection 2022.
2
[Expert consensus for the diagnosis and treatment of patients with Gitelman syndrome].
Zhonghua Nei Ke Za Zhi. 2017 Sep 1;56(9):712-716. doi: 10.3760/cma.j.issn.0578-1426.2017.09.021.
3
Altered renal distal tubule structure and renal Na(+) and Ca(2+) handling in a mouse model for Gitelman's syndrome.
J Am Soc Nephrol. 2004 Sep;15(9):2276-88. doi: 10.1097/01.ASN.0000138234.18569.63.
5
Mutations in the chloride channel gene, CLCNKB, leading to a mixed Bartter-Gitelman phenotype.
Pediatr Res. 2000 Dec;48(6):754-8. doi: 10.1203/00006450-200012000-00009.
6
Gitelman syndrome with normocalciuria - a case report.
BMC Nephrol. 2022 May 4;23(1):170. doi: 10.1186/s12882-022-02782-y.
8
The genetic spectrum of Gitelman(-like) syndromes.
Curr Opin Nephrol Hypertens. 2022 Sep 1;31(5):508-515. doi: 10.1097/MNH.0000000000000818. Epub 2022 Jul 11.

引用本文的文献

1
2
Gitelman syndrome presenting with lower limb paralysis: a case report.
J Med Case Rep. 2025 Feb 24;19(1):69. doi: 10.1186/s13256-025-05106-4.
3
Pathophysiological role of Na-Cl cotransporter in kidneys, blood pressure, and metabolism.
Hum Cell. 2024 Sep;37(5):1306-1315. doi: 10.1007/s13577-024-01099-2. Epub 2024 Jul 10.
4
Renal Hypokalemia: An Endocrine Perspective.
J Clin Endocrinol Metab. 2024 Jun 17;109(7):1694-1706. doi: 10.1210/clinem/dgae201.
6
Novel compound heterozygous variants of gene in a Chinese patient with Gitelman syndrome: a case report.
Front Genet. 2023 Jun 12;14:1067242. doi: 10.3389/fgene.2023.1067242. eCollection 2023.

本文引用的文献

1
Molecular Mechanisms of Renal Magnesium Reabsorption.
J Am Soc Nephrol. 2021 Sep;32(9):2125-2136. doi: 10.1681/ASN.2021010042. Epub 2021 May 27.
2
Deletion of renal Nedd4-2 abolishes the effect of high K intake on Kir4.1/Kir5.1 and NCC activity in the distal convoluted tubule.
Am J Physiol Renal Physiol. 2021 Jul 1;321(1):F1-F11. doi: 10.1152/ajprenal.00072.2021. Epub 2021 May 24.
3
Deletion of renal Nedd4-2 abolishes the effect of high sodium intake (HS) on Kir4.1, ENaC, and NCC and causes hypokalemia during high HS.
Am J Physiol Renal Physiol. 2021 May 1;320(5):F883-F896. doi: 10.1152/ajprenal.00555.2020. Epub 2021 Apr 5.
4
Targeted Single-Cell RNA-seq Identifies Minority Cell Types of Kidney Distal Nephron.
J Am Soc Nephrol. 2021 Apr;32(4):886-896. doi: 10.1681/ASN.2020101407. Epub 2021 Mar 4.
5
Magnesium and Calcium Homeostasis Depend on KCTD1 Function in the Distal Nephron.
Cell Rep. 2021 Jan 12;34(2):108616. doi: 10.1016/j.celrep.2020.108616.
6
NaCl cotransporter activity and Mg handling by the distal convoluted tubule.
Am J Physiol Renal Physiol. 2020 Dec 1;319(6):F1043-F1053. doi: 10.1152/ajprenal.00463.2020. Epub 2020 Nov 2.
7
Distal convoluted tubule Cl concentration is modulated via K channels and transporters.
Am J Physiol Renal Physiol. 2020 Sep 1;319(3):F534-F540. doi: 10.1152/ajprenal.00284.2020. Epub 2020 Jul 27.
8
Mechanisms coupling sodium and magnesium reabsorption in the distal convoluted tubule of the kidney.
Acta Physiol (Oxf). 2021 Feb;231(2):e13528. doi: 10.1111/apha.13528. Epub 2020 Jul 26.
9
Quantitative Proteomics of All 14 Renal Tubule Segments in Rat.
J Am Soc Nephrol. 2020 Jun;31(6):1255-1266. doi: 10.1681/ASN.2020010071. Epub 2020 May 1.
10
Renal Tubule Nedd4-2 Deficiency Stimulates Kir4.1/Kir5.1 and Thiazide-Sensitive NaCl Cotransporter in Distal Convoluted Tubule.
J Am Soc Nephrol. 2020 Jun;31(6):1226-1242. doi: 10.1681/ASN.2019090923. Epub 2020 Apr 15.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验