Reyes Jeremiah V, Medina Paul Mark B
Biological Models Laboratory, Department of Biochemistry and Molecular Biology, College of Medicine, University of the Philippines Manila Ermita, Manila 1000, Philippines.
Am J Transl Res. 2022 Jan 15;14(1):1-19. eCollection 2022.
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.
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。