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诊断表现为正常血镁的非典型 Gitelman 综合征时存在的问题。

Problems in diagnosing atypical Gitelman's syndrome presenting with normomagnesaemia.

机构信息

Department of Medicine II, Hokkaido University Graduate School of Medicine, Sapporo, Japan.

出版信息

Clin Endocrinol (Oxf). 2010 Feb;72(2):272-6. doi: 10.1111/j.1365-2265.2009.03649.x. Epub 2009 Jun 8.

Abstract

OBJECTIVE

Gitelman's syndrome, recognized as a variant of Bartter's syndrome, is characterized by hypokalaemic metabolic alkalosis in combination with hypomagnesaemia and hypocalciuria. Overlapping biochemical features in Gitelman's syndrome and Bartter's syndrome has been observed. Here, we investigated the clinical, biochemical, and genetic characteristics of five, chronic, nonhypertensive and hypokalaemic Japanese patients.

METHODS

Serum and urinary electrolytes, plasma renin activity and plasma aldosterone concentration were measured in five patients (four males and one female) with hypokalaemia. Renal clearance tests were performed and distal fractional chloride reabsorption calculated. Finally, mutational analysis of the thiazide-sensitive Na-Cl co-transporter gene was performed.

RESULTS

Symptoms in patients varied from mild (muscle weakness and numbness) to severe (tetany and foot paralysis). All patients were normotensive or hypotensive, and all had hypokalaemia, hypocalciuria, and hyperreninaemic hyperaldosteronism. However, two male patients had normomagnesaemia, while the remainder was hypomagnesaemic. Renal clearance tests showed that the administration of furosemide decreased distal fractional chloride reabsorption, while thiazide ingestion failed to decrease it. Genetic analysis identified six thiazide-sensitive Na-Cl co-transporter gene mutations, including two novel ones. Therefore, on the basis of the confirmatory renal clearance tests and mutational analysis, a diagnosis of Gitelman's syndrome was made in these patients.

CONCLUSIONS

Two of the five patients diagnosed with Gitelman's syndrome were normomagnesaemic, which is uncommon in this syndrome. Our study indicates that renal clearance tests and mutation analysis can play an important role in diagnosing Gitelman's syndrome more precisely.

摘要

目的

吉特曼综合征是巴特综合征的一种变异型,其特征是低钾性代谢性碱中毒合并低镁血症和低钙尿症。吉特曼综合征和巴特综合征存在重叠的生化特征。在此,我们研究了 5 例慢性、非高血压和低钾血症的日本患者的临床、生化和遗传特征。

方法

对 5 例低钾血症患者(4 名男性和 1 名女性)进行了血清和尿电解质、血浆肾素活性和血浆醛固酮浓度检测。进行了肾清除率试验,并计算了远端氯离子重吸收分数。最后,对噻嗪类敏感的 Na-Cl 共转运蛋白基因进行了突变分析。

结果

患者的症状从轻度(肌肉无力和麻木)到重度(抽搐和足瘫痪)不等。所有患者均为血压正常或低血压,均有低钾血症、低钙尿症和高肾素性醛固酮血症。然而,2 名男性患者的镁浓度正常,而其余患者的镁浓度较低。肾清除率试验表明,呋塞米的给予降低了远端氯离子重吸收分数,而噻嗪的给予未能降低其重吸收分数。基因分析确定了 6 个噻嗪类敏感的 Na-Cl 共转运蛋白基因突变,包括 2 个新的基因突变。因此,基于明确的肾清除率试验和突变分析,这些患者被诊断为吉特曼综合征。

结论

5 例被诊断为吉特曼综合征的患者中有 2 例的镁浓度正常,这在该综合征中较为罕见。我们的研究表明,肾清除率试验和突变分析在更准确地诊断吉特曼综合征方面可发挥重要作用。

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