Gamba Gerardo
Molecular Physiology Unit, Instituto de Ciencias Médicas y Nutrición Salvador Zubirán, Vasco de Quiroga No. 15, Tlalpan 14000, México City, Mexico.
Am J Physiol Renal Physiol. 2005 Feb;288(2):F245-52. doi: 10.1152/ajprenal.00311.2004.
A recently discovered family of protein kinases is responsible for an autosomal-dominant disease known as Gordon's syndrome or pseudohypoaldosteronism type II (PHA-II) that features hyperkalemia and hyperchloremic metabolic acidosis, accompanied by hypertension and hypercalciuria. Four genes have been described in this kinase family, which has been named WNK, due to the absence of a key lysine in kinase subdomain II (with no K kinases). Two of these genes, WNK1 and WNK4 located in human chromosomes 12 and 17, respectively, are responsible for PHA-II. Immunohystochemical analysis revealed that WNK1 and WNK4 are predominantly expressed in the distal convoluted tubule and collecting duct. The physiological studies have shown that WNK4 downregulates the activity of ion transport pathways expressed in these nephron segments, such as the apical thiazide-sensitive Na+-Cl- cotransporter and apical secretory K+ channel ROMK, as well as upregulates paracellular chloride transport and phosphorylation of tight junction proteins such as claudins. In addition, WNK4 downregulates other Cl- influx pathways such as the basolateral Na+-K+-2Cl- cotransporter and Cl-/HCO3- exchanger. WNK4 mutations behave as a loss of function for the Na+-Cl- cotransporter and a gain of function when it comes to ROMK and claudins. These dual effects of WNK4 mutations fit with proposed mechanisms for developing electrolyte abnormalities and hypertension in PHA-II and point to WNK4 as a multifunctional regulator of diverse ion transporters.
最近发现的一个蛋白激酶家族与一种常染色体显性疾病有关,该疾病被称为戈登综合征或II型假性醛固酮增多症(PHA-II),其特征为高钾血症和高氯性代谢性酸中毒,并伴有高血压和高钙尿症。这个激酶家族中有四个基因已被描述,因其激酶亚结构域II中缺少一个关键赖氨酸(无K激酶),故而被命名为WNK。其中两个基因,WNK1和WNK4分别位于人类染色体12和17上,是PHA-II的致病原因。免疫组织化学分析显示,WNK1和WNK4主要在远曲小管和集合管中表达。生理学研究表明,WNK4下调这些肾单位节段中表达的离子转运途径的活性,如顶端噻嗪类敏感的Na+-Cl-共转运体和顶端分泌性K+通道ROMK,同时上调细胞旁氯化物转运以及紧密连接蛋白(如claudins)的磷酸化。此外,WNK4下调其他Cl-内流途径,如基底外侧Na+-K+-2Cl-共转运体和Cl-/HCO3-交换体。WNK4突变对于Na+-Cl-共转运体表现为功能丧失,而对于ROMK和claudins则表现为功能获得。WNK4突变的这些双重效应符合PHA-II中电解质异常和高血压发生的推测机制,并表明WNK4是多种离子转运体的多功能调节因子。