Monticone S, Tetti M, Burrello J, Buffolo F, De Giovanni R, Veglio F, Williams T A, Mulatero P
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.
J Hum Hypertens. 2017 Dec;31(12):776-781. doi: 10.1038/jhh.2017.34. Epub 2017 Apr 27.
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.
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。