Lucis O J, Hobkirk R, Hollenberg C H, MacDonald S A, Blahey P
Can Med Assoc J. 1966 Jan 1;94(1):1-7.
Polycystic ovaries were found in a 16-year-old female with congenital absence of vagina, male-like external genitalia, and congenital adrenal hyperplasia. Masculinization was sufficiently severe to cause the patient to be reared as a male. Biochemical studies of ovarian tissue revealed hyperactivity and an imbalance of enzyme systems concerned with steroid-hormone biosynthesis, which led to production of large amounts of androgens. The pathway towards estrogens was preserved but less efficient than normal. Urinary steroid metabolites before and after hysterectomy and bilateral salpingo-oophorectomy revealed an absence of Porter-Silber chromogens and tetrahydrocortisone. Excretion of aldosterone was normal and that of corticosterone slightly higher than normal. The patterns of urinary 17-ketosteroids, pregnanediol, pregnanetriol and pregnanetriolone were similar to those commonly seen in congenital adrenal hyperplasia with steroid 21-hydroxylase deficiency. Urinary estrogens after panhysterectomy were low, being in the post-menopausal range. The pathogenesis of polycystic ovaries and their possible contribution to masculinization are discussed.
在一名16岁女性中发现多囊卵巢,该女性先天性无阴道、外生殖器呈男性样且患有先天性肾上腺增生。男性化程度严重到足以使患者被当作男性抚养。对卵巢组织的生化研究显示与类固醇激素生物合成相关的酶系统活性亢进且失衡,这导致大量雄激素的产生。雌激素生成途径得以保留,但效率低于正常水平。子宫切除及双侧输卵管卵巢切除术前和术后的尿类固醇代谢产物显示缺乏波特 - 西尔伯色原和四氢皮质醇。醛固酮排泄正常,皮质酮排泄略高于正常。尿17 - 酮类固醇、孕二醇、孕三醇和孕三酮的模式与常见的21 - 羟化酶缺乏型先天性肾上腺增生相似。全子宫切除术后尿雌激素水平较低,处于绝经后范围。本文讨论了多囊卵巢的发病机制及其对男性化可能产生的影响。