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11β-羟化酶缺乏所致先天性肾上腺皮质增生症的临床变异性

Clinical variability of congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency.

作者信息

Rösler A, Leiberman E, Sack J, Landau H, Benderly A, Moses S W, Cohen T

出版信息

Horm Res. 1982;16(3):133-41. doi: 10.1159/000179494.

Abstract

Studies in 18 Jewish families from Morocco, Tunis, Turkey and Iran revealed 26 patients with congenital adrenal hyperplasia due to 11 beta-hydroxylase deficiency. The clinical expression of androgen excess varied widely in affected females, and range from solely enlarged clitoris in the mildest forms to severely hypertrophied clitoris with penile urethra and fused labial-scrotal folds in the most extreme forms of masculinization. Intermediate degrees of severity were manifested by ambiguous genitalia. There was no correlation between the degree of virilization and the signs of mineralocorticoid excess. Severe volume-induced hypertension leading to vascular accidents and death were also observed in severe as well as in mildly virilized patients, while completely masculinized females were sometime normotensive. Overt hypokalemia was present in 6 patients but was not a constant feature of hypertensives. However, all affected individuals, except for 2 infants, had very low levels of plasma renin activity suggesting that a state of volume expansion was indeed present in the majority of cases, even though changes in blood pressure did not always occur. The clinical expression of this disorder is characterized by a wide range of variability in the signs of both androgen and mineralocorticoid excess, which do not necessarily correlate with the quantity of hormones secreted.

摘要

对来自摩洛哥、突尼斯、土耳其和伊朗的18个犹太家庭的研究发现,有26例患者因11β-羟化酶缺乏而患有先天性肾上腺皮质增生症。受影响女性雄激素过多的临床表现差异很大,从最轻微形式的单纯阴蒂增大到最极端男性化形式的严重肥大阴蒂伴阴茎尿道和阴唇阴囊褶融合。中度严重程度表现为生殖器模糊。男性化程度与盐皮质激素过多的体征之间没有相关性。在重度和轻度男性化患者中也观察到严重的容量性高血压导致血管意外和死亡,而完全男性化的女性有时血压正常。6例患者出现明显低钾血症,但并非高血压患者的恒定特征。然而,除2例婴儿外,所有受影响个体的血浆肾素活性都非常低,这表明即使血压并非总是发生变化,但大多数病例中确实存在容量扩张状态。这种疾病的临床表现特征是雄激素和盐皮质激素过多的体征存在广泛的变异性,且不一定与分泌的激素量相关。

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