Dallago Cristina Micheletto, Abech Denise Dotta, Pereira-Lima Julia Fernanda Semmelmann, Leães Caroline Garcia Soares, Batista Rafael Loch, Trarbach Ericka Barbosa, Oliveira Miriam da Costa
Departamento de Endocrinologia, Complexo Hospitalar Santa Casa de Porto Alegre, Porto Alegre, Brazil.
Pituitary. 2008;11(1):109-12. doi: 10.1007/s11102-007-0043-9.
Kallmann syndrome (KS) is a developmental disease characterized by the association of isolated hypogonadotropic hypogonadism and anosmia/hyposmia. We report an unusual presentation of two females with KS and empty sella. These females, aged at 20 and 29-year-old, presented primary amenorrhea with prepubertal estradiol and low gonadotropin levels. No other significant clinical signs were observed. Empty sella was observed on MRI in both cases. Sequencing of FGFR1 gene, recently implicated in autosomal form of KS, was performed and one splicing mutation (IVS14 + 1G > A) was identified in one patient.
卡尔曼综合征(KS)是一种发育性疾病,其特征为孤立性低促性腺激素性性腺功能减退与嗅觉缺失/嗅觉减退相关。我们报告了两名患有KS和空蝶鞍的女性的不寻常表现。这两名女性年龄分别为20岁和29岁,表现为原发性闭经,青春期前雌二醇水平和促性腺激素水平低。未观察到其他明显的临床体征。两例患者的MRI均显示有空蝶鞍。对最近发现与常染色体形式的KS相关的FGFR1基因进行了测序,在一名患者中鉴定出一个剪接突变(IVS14 + 1G > A)。