D'Alberton A, Reschini E, Motta T, Catania A
Clinica Ostetrica e Ginecologica I, Università di Milano, Italy.
J Endocrinol Invest. 1989 Mar;12(3):193-6. doi: 10.1007/BF03349960.
A 27-year-old phenotypic female presented with primary amenorrhea, severe hypertension, and hypokalemia. At the age of puberty sexual development had not occurred; in particular, sexual hair had not grown. Past history revealed an episode of subarachnoid hemorrhage and several episodes of ventricular tachyarrhythmia. Karyotype was 46, XY. The steroids requiring 17-hydroxylation (cortisol, testosterone, pregnanetriol, 17-ketosteroids, 17-hydroxycorticosteroids) were low, while those not requiring 17-hydroxylation (progesterone, deoxycorticosterone, corticosterone) were high, demonstrating 17-hydroxylase deficiency. The corticosterone/deoxycorticosterone ratio was relatively low, suggesting an associated partial deficiency of 11-hydroxylase.
一名27岁的表型女性出现原发性闭经、严重高血压和低钾血症。青春期时性发育未发生;特别是阴毛未生长。既往史显示有一次蛛网膜下腔出血和几次室性快速心律失常发作。核型为46,XY。需要17-羟化的类固醇(皮质醇、睾酮、孕三醇、17-酮类固醇、17-羟皮质类固醇)水平较低,而不需要17-羟化的类固醇(孕酮、脱氧皮质酮、皮质酮)水平较高,表明存在17-羟化酶缺乏。皮质酮/脱氧皮质酮比值相对较低,提示伴有11-羟化酶部分缺乏。