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由尿调节蛋白突变引起的常染色体显性遗传性肾小管间质性肾病:寻则得之。

Autosomal dominant tubulointerstitial kidney disease caused by uromodulin mutations: seek and you will find.

作者信息

Raffler Gabriele, Zitt Emanuel, Sprenger-Mähr Hannelore, Nagel Mato, Lhotta Karl

机构信息

Department of Nephrology and Dialysis, Academic Teaching Hospital Feldkirch, Carinagasse 47, 6800, Feldkirch, Austria.

Laboratory for Molecular Diagnostics, Center for Nephrology and Metabolic Disorders, Weißwasser, Germany.

出版信息

Wien Klin Wochenschr. 2016 Apr;128(7-8):291-4. doi: 10.1007/s00508-015-0948-7. Epub 2016 Jan 25.

Abstract

BACKGROUND

Uromodulin (UMOD)-associated kidney disease belongs to the group of autosomal dominant interstitial kidney diseases and is caused by mutations in the UMOD gene. Affected patients present with hyperuricemia, gout, and progressive renal failure. The disease is thought to be very rare but is probably underdiagnosed.

METHODS

Two index patients from two families with tubulointerstitial nephropathy and hyperuricemia were examined, including blood and urine chemistry, ultrasound, and mutation analysis of the UMOD gene. In addition, other available family members were studied.

RESULTS

In a 46-year-old female patient with a fractional excretion of uric acid of 3 %, analysis of the UMOD gene revealed a p.W202S missense mutation. The same mutation was found in her 72-year-old father, who suffers from gout and end-stage renal disease. The second index patient was a 47-year-old female with chronic kidney disease and gout for more than 10 years. Her fractional uric acid excretion was 3.5 %. Genetic analysis identified a novel p.H250Q UMOD mutation that was also present in her 12-year-old son, who had normal renal function and uric acid levels.

CONCLUSION

In patients suffering from chronic tubulointerstitial nephropathy, hyperuricemia, and a low fractional excretion of uric acid mutation, analysis of the UMOD gene should be performed to diagnose UMOD-associated kidney disease.

摘要

背景

尿调节蛋白(UMOD)相关肾病属于常染色体显性遗传性间质性肾病,由UMOD基因突变引起。患病患者表现为高尿酸血症、痛风和进行性肾衰竭。该疾病被认为非常罕见,但可能存在诊断不足的情况。

方法

对来自两个患有肾小管间质性肾病和高尿酸血症家庭的两名索引患者进行了检查,包括血液和尿液化学检查、超声检查以及UMOD基因的突变分析。此外,还对其他在世的家庭成员进行了研究。

结果

在一名尿酸排泄分数为3%的46岁女性患者中,UMOD基因分析发现了一个p.W202S错义突变。在她患有痛风和终末期肾病的72岁父亲身上也发现了相同的突变。第二名索引患者是一名患有慢性肾病和痛风超过10年的47岁女性。她的尿酸排泄分数为3.5%。基因分析确定了一个新的p.H250Q UMOD突变,该突变也存在于她12岁的儿子身上,其肾功能和尿酸水平正常。

结论

对于患有慢性肾小管间质性肾病、高尿酸血症且尿酸排泄分数低的患者,应进行UMOD基因分析以诊断UMOD相关肾病。

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