Károlyi L, Ziegler A, Pollak M, Fischbach M, Grzeschik K H, Koch M C, Seyberth H W
Medizinisches Zentrum für Kinderheilkunde, Philipps-Universität, Marburg, Germany.
Pediatr Nephrol. 1996 Oct;10(5):551-4. doi: 10.1007/s004670050158.
In the past the term Bartter's syndrome has been used to describe a spectrum of inherited renal tubular disorders with hypokalemic metabolic alkalosis and overlapping and additional clinical and biochemical features. Pathogenesis remained uncertain until recently Gitelman's syndrome, the hypokalemic-hypomagnesemic variant with hypocalciuria, was linked to the gene encoding the thiazide-sensitive Na-Cl-cotransporter (TSC) located on chromosome 16q. Various mutations in the TSC gene were identified in patients with Gitelman's syndrome. To clarify whether different forms of hypokalemic tubular disorders (HTD) represent variable phenotypes of a common genetic defect, we performed linkage analyses in 17 families with different symptoms of HTD with four highly polymorphic chromosome 16 DNA markers closely linked to the TSC gene. Linkage of Gitelman's syndrome to the TSC locus was confirmed in our families with a maximum two-point Lod score Z = 4.70 (theta = 0.001) for marker locus D16S526. Highly negative LOD scores were obtained at this locus in our families with classic Bartter's syndrome (Z = 9.89, theta = 0.001) and hyperprostaglandin E syndrome (Z = -11.24, theta = 0.001). Our data prove that Gitelman's syndrome is genetically distinct from classic Bartter's syndrome and hyperprostaglandin E syndrome. It remains unknown if classic Bartter's syndrome and hyperprostaglandin E syndrome are caused by a common genetic defect.
过去,巴特综合征(Bartter's syndrome)一词用于描述一系列遗传性肾小管疾病,这些疾病伴有低钾血症性代谢性碱中毒以及重叠和其他临床及生化特征。直到最近,吉特曼综合征(Gitelman's syndrome),即伴有低钙尿症的低钾血症-低镁血症变体,与位于16号染色体q上编码噻嗪类敏感钠-氯协同转运蛋白(TSC)的基因联系起来,其发病机制才得以明确。在吉特曼综合征患者中发现了TSC基因的各种突变。为了阐明低钾性肾小管疾病(HTD)的不同形式是否代表共同遗传缺陷的可变表型,我们对17个有不同HTD症状的家庭进行了连锁分析,使用了与TSC基因紧密连锁的四个高度多态性的16号染色体DNA标记。在我们的家庭中,通过标记位点D16S526确认了吉特曼综合征与TSC基因座的连锁,最大两点Lod分数Z = 4.70(θ = 0.001)。在我们患有经典巴特综合征(Z = 9.89,θ = 0.001)和高前列腺素E综合征(Z = -11.24,θ = 0.001)的家庭中,该位点获得了高度阴性的Lod分数。我们的数据证明,吉特曼综合征在遗传上与经典巴特综合征和高前列腺素E综合征不同。经典巴特综合征和高前列腺素E综合征是否由共同遗传缺陷引起仍不清楚。