Institut National de la Santé et de la Recherche Médicale, Unité Mixte de Recherche Scientifique 970, Paris Cardiovascular Research Center, Paris, France.
Hypertension. 2012 Mar;59(3):592-8. doi: 10.1161/HYPERTENSIONAHA.111.186478. Epub 2012 Jan 23.
Primary aldosteronism is the most common form of secondary hypertension. Mutations in the KCNJ5 gene have been described recently in aldosterone-producing adenomas (APAs). The aim of this study was to investigate the prevalence of KCNJ5 mutations in unselected patients with primary aldosteronism and their clinical, biological and molecular correlates. KCNJ5 sequencing was performed on somatic (APA, n=380) and peripheral (APA, n=344; bilateral adrenal hyperplasia, n=174) DNA of patients with primary aldosteronism, collected through the European Network for the Study of Adrenal Tumors. Transcriptome analysis was performed in 102 tumors. Somatic KCNJ5 mutations (p.Gly151Arg or p.Leu168Arg) were found in 34% (129 of 380) of APA. They were significantly more prevalent in females (49%) than males (19%; P<10(-3)) and in younger patients (42.1±1.0 versus 47.6±0.7 years; P<10(-3)) and were associated with higher preoperative aldosterone levels (455±26 versus 376±17 ng/L; P=0.012) but not with therapeutic outcome after surgery. Germline KCNJ5 mutations were found neither in patients with APA nor those with bilateral adrenal hyperplasia. Somatic KCNJ5 mutations were specific for APA, because they were not identified in 25 peritumoral adrenal tissues or 16 cortisol-producing adenomas. Hierarchical clustering of transcriptome profiles showed that APAs with p.Gly151Arg or p.Leu168Arg mutations were indistinguishable from tumors without KCNJ5 mutations. In conclusion, although a large proportion of sporadic APAs harbors somatic KCNJ5 mutations, germline mutations are not similarly causative for bilateral adrenal hyperplasia. KCNJ5 mutation carriers are more likely to be females; younger age and higher aldosterone levels at diagnosis suggest that KCNJ5 mutations may be associated with a more florid phenotype of primary aldosteronism.
原发性醛固酮增多症是最常见的继发性高血压类型。最近在醛固酮分泌腺瘤(APA)中描述了 KCNJ5 基因突变。本研究旨在调查原发性醛固酮增多症患者中非选择性 KCNJ5 突变的流行率及其临床、生物学和分子相关性。通过欧洲肾上腺肿瘤研究网络收集了原发性醛固酮增多症患者的体细胞(APA,n=380)和外周(APA,n=344;双侧肾上腺增生,n=174)DNA,对 KCNJ5 进行测序。对 102 个肿瘤进行了转录组分析。在 380 个 APA 中发现了体细胞 KCNJ5 突变(p.Gly151Arg 或 p.Leu168Arg),占 34%(129 例)。它们在女性(49%)中明显比男性(19%;P<10(-3))和年轻患者(42.1±1.0 岁比 47.6±0.7 岁;P<10(-3))更为常见,并且与术前醛固酮水平较高(455±26 比 376±17 ng/L;P=0.012)相关,但与手术后的治疗效果无关。在 APA 患者和双侧肾上腺增生患者中均未发现胚系 KCNJ5 突变。体细胞 KCNJ5 突变是 APA 的特异性,因为在 25 个肿瘤旁肾上腺组织或 16 个皮质醇分泌腺瘤中未发现这些突变。转录组谱的层次聚类显示,具有 p.Gly151Arg 或 p.Leu168Arg 突变的 APA 与无 KCNJ5 突变的肿瘤无法区分。总之,尽管散发性 APA 中有很大一部分存在体细胞 KCNJ5 突变,但胚系突变并非导致双侧肾上腺增生的同样原因。KCNJ5 突变携带者更可能是女性;年龄较小和诊断时醛固酮水平较高提示 KCNJ5 突变可能与原发性醛固酮增多症的更明显表型有关。