Pescovitz O H, Comite F, Cassorla F, Dwyer A J, Poth M A, Sperling M A, Hench K, McNemar A, Skerda M, Loriaux D L
J Clin Endocrinol Metab. 1984 May;58(5):857-61. doi: 10.1210/jcem-58-5-857.
Congenital adrenal hyperplasia (CAH) is a recognized cause of precocious pseudopuberty. Some children with CAH also develop true precocious puberty with early maturation of the hypothalamic-pituitary-gonadal axis. We have seen four such children (three boys and one girl) who had the diagnosis of CAH made between the ages of 3 and 6 yr. These patients were treated with standard doses of hydrocortisone and fludrocortisone. A diagnosis of true precocious puberty was made because of testicular enlargement in the boys, breast development in the girl, progressive pubic hair development, rapid growth, and rapid bone age maturation. Plasma steroid levels were elevated for age, and gonadotropin levels were within the normal pubertal range, both basally and in response to LHRH stimulation. We treated these children with daily sc injections of a LHRH analog (LHRHa) for 6-18 months in addition to the standard hydrocortisone and fludrocortisone therapy for CAH. LHRHa significantly decreased basal plasma LH and FSH, peak LH and FSH responses to native LHRH, and testosterone levels. Testis size decreased in the males, and breast development regressed in the female. LHRHa therapy led to significant decreases in linear growth rate, ulnar growth rate, and rate of bone age advancement. These results suggest that LHRHa is an effective adjunct to hydrocortisone and fludrocortisone in the treatment of true precocious puberty complicating CAH.
先天性肾上腺增生(CAH)是性早熟假性青春期的一个公认病因。一些患有CAH的儿童也会出现真性性早熟,伴有下丘脑 - 垂体 - 性腺轴的过早成熟。我们见过4名这样的儿童(3名男孩和1名女孩),他们在3至6岁之间被诊断为CAH。这些患者接受了标准剂量的氢化可的松和氟氢可的松治疗。由于男孩睾丸增大、女孩乳房发育、阴毛逐渐生长、生长迅速以及骨龄快速成熟,诊断为真性性早熟。血浆类固醇水平随年龄升高,促性腺激素水平在基础状态以及对促黄体生成素释放激素(LHRH)刺激的反应中均处于正常青春期范围内。除了针对CAH的标准氢化可的松和氟氢可的松治疗外,我们还对这些儿童每日皮下注射LHRH类似物(LHRHa)6至18个月。LHRHa显著降低了基础血浆促黄体生成素(LH)和促卵泡生成素(FSH)、对天然LHRH的LH和FSH峰值反应以及睾酮水平。男性睾丸大小减小,女性乳房发育消退。LHRHa治疗导致线性生长速率、尺骨生长速率和骨龄进展速率显著降低。这些结果表明,LHRHa是氢化可的松和氟氢可的松治疗CAH合并真性性早熟的有效辅助药物。