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成年期表现为红细胞增多症的IV型巴特综合征

Adult presentation of Bartter syndrome type IV with erythrocytosis.

作者信息

Heilberg Ita Pfeferman, Tótoli Cláudia, Calado Joaquim Tomaz

机构信息

Universidade Federal de São Paulo, São Paulo, SP, Brazil.

Faculdade de Ciências Médicas, Universidade Nova de Lisboa, Lisboa, Portugal.

出版信息

Einstein (Sao Paulo). 2015 Oct-Dec;13(4):604-6. doi: 10.1590/S1679-45082015RC3013. Epub 2015 Oct 30.

Abstract

Bartter syndrome comprises a group of rare autosomal-recessive salt-losing disorders with distinct phenotypes, but one unifying pathophysiology consisting of severe reductions of sodium reabsorption caused by mutations in five genes expressed in the thick ascending limb of Henle, coupled with increased urinary excretion of potassium and hydrogen, which leads to hypokalemic alkalosis. Bartter syndrome type IV, caused by loss-of-function mutations in barttin, a subunit of chloride channel CLC-Kb expressed in the kidney and inner ear, usually occurs in the antenatal-neonatal period. We report an unusual case of late onset presentation of Bartter syndrome IV and mild phenotype in a 20 years-old man who had hypokalemia, deafness, secondary hyperparathyroidism and erythrocytosis.

摘要

巴特综合征是一组罕见的常染色体隐性失盐性疾病,具有不同的表型,但有一个统一的病理生理学机制,即由在髓袢升支粗段表达的五个基因突变导致钠重吸收严重减少,同时钾和氢的尿排泄增加,进而导致低钾性碱中毒。IV型巴特综合征由肾脏和内耳中表达的氯离子通道CLC-Kb的一个亚基barttin功能丧失性突变引起,通常发生在产前-新生儿期。我们报告了一例不寻常的IV型巴特综合征迟发性病例,该20岁男性具有低钾血症、耳聋、继发性甲状旁腺功能亢进和红细胞增多症,但其表型较轻。

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本文引用的文献

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Disease-causing dysfunctions of barttin in Bartter syndrome type IV.巴特综合征IV型中barttin的致病功能障碍。
J Am Soc Nephrol. 2009 Jan;20(1):145-53. doi: 10.1681/ASN.2008010102. Epub 2008 Sep 5.
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Bartter syndrome: benefits and side effects of long-term treatment.巴特综合征:长期治疗的益处与副作用
Pediatr Nephrol. 2004 Aug;19(8):858-63. doi: 10.1007/s00467-004-1527-8. Epub 2004 Jun 16.
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Phenotypic variability in Bartter syndrome type I.I型巴特综合征的表型变异性。
Pediatr Nephrol. 2000 Sep;14(10-11):940-5. doi: 10.1007/pl00013418.

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