Bierich J R, Schmidt U
Eur J Pediatr. 1976 Jan 2;121(2):81-7. doi: 10.1007/BF00443063.
In the serum of two infant sisters with a congenital renal salt-losing syndrome, Na was rather low and K considerably increased. Even with Na levels of 126 mval/1, sodium was excreted in the urine. Creatinine and hippurate clearances were normal. Primary disturbances of the steroid metabolism were not detectable; plasma cortisol was normal, aldosterone and renin were compensatorily increased. Treatment with DOCA was unsuccessful. Whereas the first infant died (in another hospital), the second one throve well with high oral substitution of NaCl. There was no pathological findings other than a moderate hyperplasia of the juxtaglomerular apparatus, in a kidney biopsy. Except for minimal activity in the ascending limb of Henle's loop, there was no membrane bound Na, K-ATPase in the microdissected tubules. This finding most probably explains the renal salt loss, as this enzyme is necessary for the transcellular flow of sodium and potassium.
在两名患有先天性肾失盐综合征的婴儿姐妹的血清中,钠含量相当低,钾含量显著升高。即使钠水平为126毫当量/升,尿液中仍有钠排出。肌酐和马尿酸盐清除率正常。未检测到类固醇代谢的原发性紊乱;血浆皮质醇正常,醛固酮和肾素代偿性升高。用去氧皮质酮治疗未成功。第一名婴儿死亡(在另一家医院),而第二名婴儿通过高剂量口服氯化钠替代治疗后茁壮成长。肾活检除了肾小球旁器有中度增生外,没有其他病理发现。在显微解剖的肾小管中,除了亨氏袢升支有轻微活性外,没有膜结合的钠钾ATP酶。这一发现很可能解释了肾失盐现象,因为这种酶是钠和钾跨细胞流动所必需的。