Kuhnle U, Chow D, Rapaport R, Pang S, Levine L S, New M I
J Clin Endocrinol Metab. 1981 Mar;52(3):534-44. doi: 10.1210/jcem-52-3-534.
In the two clinical syndromes of congenital adrenal hyperplasia due to a 21-hydroxylation defect of adrenal steroidogenesis, the simple virilizing and the salt-wasting forms, the 21-hydroxylase activity was studied considering the zona fasciculata and the zona glomerulosa of the adrenal cortex as two separate glands under different regulation. To test this hypothesis, we stimulated adrenal steroidogenesis by ACTH infusion or dietary sodium restriction in eight patients with congenital adrenal hyperplasia (four patients with the simple virilizing form and four with the salt-wasting form of congenital adrenal hyperplasia) and in six normal children. Both the 17-hydroxy and 17-deoxy pathways of adrenocortical steroid biosynthesis were examined by measuring serum concentrations of 17-hydroxyprogesterone, cortisol, progesterone, deoxycorticosterone, corticosterone, and aldosterone and the excretion of free deoxycorticosterone, 18-hydroxydeoxycorticosterone, corticosterone, 18-hydroxycorticosterone, cortisol, and aldosterone. We considered the steroids 18-hydroxycorticosterone and aldosterone to be primarily of zona glomerulosa origin. These studies indicated that the zona fasciculata of both the salt-wasting and the simple virilizing forms is defective in 21-hydroxylation of 17-hydroxy and 17-deoxy steroids. The zona glomerulosa demonstrated deficient 21-hydroxylation only in the salt-wasting form, whereas in the simple virilizing form, the glomerulosa was spared this defect.
在因肾上腺类固醇生成21-羟化缺陷导致的先天性肾上腺皮质增生的两种临床综合征,即单纯男性化型和失盐型中,将肾上腺皮质的束状带和球状带视为受不同调节的两个独立腺体,对21-羟化酶活性进行了研究。为验证这一假设,我们对8例先天性肾上腺皮质增生患者(4例单纯男性化型先天性肾上腺皮质增生患者和4例失盐型先天性肾上腺皮质增生患者)以及6名正常儿童,通过静脉输注促肾上腺皮质激素(ACTH)或限制饮食中的钠来刺激肾上腺类固醇生成。通过测量血清中17-羟孕酮、皮质醇、孕酮、脱氧皮质酮、皮质酮和醛固酮的浓度以及游离脱氧皮质酮、18-羟脱氧皮质酮、皮质酮、18-羟皮质酮、皮质醇和醛固酮的排泄量,对肾上腺皮质类固醇生物合成的17-羟和17-脱氧途径进行了检测。我们认为类固醇18-羟皮质酮和醛固酮主要源自球状带。这些研究表明,失盐型和单纯男性化型的束状带在17-羟和17-脱氧类固醇的21-羟化方面均存在缺陷。球状带仅在失盐型中表现出21-羟化不足,而在单纯男性化型中,球状带未出现此缺陷。