Coto Eliecer, Rodriguez Julian, Jeck Nikola, Alvarez Victoria, Stone Rosario, Loris Cesar, Rodriguez Luis M, Fischbach Michel, Seyberth Hannsjörg W, Santos Fernando
Hospital Universitario Central de Asturias, Spain.
Kidney Int. 2004 Jan;65(1):25-9. doi: 10.1111/j.1523-1755.2004.00388.x.
Gitel syndrome is an inherited tubular disorder characterized by metabolic alkalosis, hypokalemia, and hypomagnesemia of renal origin and hypocalciuria. The majority of patients with Gitelman syndrome carry inactivating mutations in the SLC12A3 gene encoding the sodium-chloride cotransporter located in the distal convoluted tubule. The purpose of this study was to investigate the underlying mutation in Gitelman syndrome patients of Gypsy race from different geographic origin.
Twenty Gypsy patients with clinical and biochemical features of Gitelman syndrome were investigated by mutational analysis. The patients belonged to 12 unrelated Gypsy families living in four different European countries. The parents and unaffected siblings of each patient, as well as the DNA of a population of 200 healthy control patients, were also analyzed.
All patients were homozygous for the same splice site mutation, guanine to thymine in the first position of intron 9 of SLC12A3 gene. This mutation was not found in the control population. Parents were heterozygous for the mutation. Despite sharing a common mutation, the clinical manifestations of the syndrome in the patients varied from lack of symptoms in six children to severe growth retardation in four.
Demonstration of a novel point mutation within the SLC12A3 gene in our cohort of Gypsy families with Gitelman syndrome is highly suggestive of a founder effect. This finding will facilitate the identification of the genetic defect in further cases of Gitelman syndrome among the Gypsy population. Our study represents the largest series ever published of patients with Gitelman syndrome having the same underlying mutation, and supports the lack of correlation between genotype and clinical phenotype in this disease.
吉特曼综合征是一种遗传性肾小管疾病,其特征为代谢性碱中毒、低钾血症、源于肾脏的低镁血症和低钙尿症。大多数吉特曼综合征患者在编码位于远曲小管的氯化钠共转运体的SLC12A3基因中携带失活突变。本研究的目的是调查来自不同地理区域的吉普赛族吉特曼综合征患者的潜在突变。
通过突变分析对20例具有吉特曼综合征临床和生化特征的吉普赛患者进行了研究。这些患者来自生活在四个不同欧洲国家的12个不相关的吉普赛家庭。还分析了每位患者的父母和未受影响的兄弟姐妹,以及200名健康对照患者群体的DNA。
所有患者在SLC12A3基因第9内含子第一位的剪接位点突变均为纯合子,即鸟嘌呤突变为胸腺嘧啶。在对照人群中未发现该突变。父母为该突变的杂合子。尽管共享一个共同突变,但该综合征在患者中的临床表现各不相同,从6名儿童无症状到4名儿童严重生长发育迟缓。
在我们这组患有吉特曼综合征的吉普赛家庭中,SLC12A3基因内一个新的点突变的发现强烈提示存在奠基者效应。这一发现将有助于在吉普赛人群中进一步诊断吉特曼综合征时识别遗传缺陷。我们的研究是有史以来发表的具有相同潜在突变的吉特曼综合征患者的最大系列研究,并支持该疾病基因型与临床表型之间缺乏相关性。