Syed F A, Chalew S A
Division of Pediatric Endocrinology, University of Maryland Medical Center, Baltimore, USA.
J Pediatr Endocrinol Metab. 1999 Jan-Feb;12(1):81-3. doi: 10.1515/jpem.1999.12.1.81.
McCune-Albright syndrome (MAS) in girls is characterized by gonadotropin independent precocious puberty (GIPP). This form of GIPP is resistant to therapy with GnRH analogues. As an alternative treatment, we successfully used ketoconazole 200 mg t.i.d. orally in two girls with MAS, GIPP and advanced bone age Ketoconazole led to rapid control of GIPP with cessation of menses and regression of pubertal signs in both patients. Ketoconazole was temporarily interrupted in one patient due to pruritus but later restarted without problem. After 1 year of therapy both patients have remained free of menses, progression of puberty and other side effects. Repeat sonography on ketoconazole revealed continued presence of ovarian cysts. Our preliminary experience indicates the safety and effectiveness of ketoconazole as a therapy for GIPP with potential advantages over previously used modes of treatment. Longer use of ketoconazole to suppress GIPP is required to determine whether this therapy can prolong linear growth with enhancement of final height.
女孩的McCune-Albright综合征(MAS)的特征是促性腺激素非依赖性性早熟(GIPP)。这种形式的GIPP对GnRH类似物治疗有抵抗性。作为一种替代治疗方法,我们成功地对两名患有MAS、GIPP且骨龄超前的女孩口服酮康唑,剂量为每日3次,每次200毫克。酮康唑使GIPP迅速得到控制,两名患者均停经且青春期体征消退。其中一名患者因瘙痒暂时中断了酮康唑治疗,但后来重新开始用药,未出现问题。治疗1年后,两名患者均未再来月经,青春期未进展,也未出现其他副作用。酮康唑治疗后的重复超声检查显示卵巢囊肿持续存在。我们的初步经验表明,酮康唑作为GIPP的治疗方法具有安全性和有效性,与先前使用的治疗方式相比可能具有潜在优势。需要更长时间使用酮康唑来抑制GIPP,以确定这种治疗方法是否可以通过增加最终身高来延长线性生长。