Division of Gynecology, Department of Perinatology and Gynecology, Poznan University of Medical Sciences, 60-535 Poznan, Poland.
Int J Mol Sci. 2021 Oct 25;22(21):11495. doi: 10.3390/ijms222111495.
Congenital anomalies of the female reproductive tract that present with primary amenorrhea involve Müllerian aplasia, also known as Mayer-Rokitansky-Küster-Hauser syndrome (MRKHS), and cervical and vaginal anomalies that completely obstruct the reproductive tract. Karyotype abnormalities do not exclude the diagnosis of MRKHS. Familial cases of Müllerian anomalies and associated malformations of the urinary and skeletal systems strongly suggest a complex genetic etiology, but so far, the molecular mechanism in the vast majority of cases remains unknown. Primary amenorrhea may also be the first presentation of complete androgen insensitivity syndrome, steroid 5α-reductase type 2 deficiency, 17β-hydroxysteroid dehydrogenase type 3 deficiency, and Leydig cells hypoplasia type 1; therefore, these disorders should be considered in the differential diagnosis of the congenital absence of the uterus and vagina. The molecular diagnosis in the majority of these cases can be established.
女性生殖道先天畸形表现为原发性闭经,包括 Müllerian 发育不全,也称为 Mayer-Rokitansky-Küster-Hauser 综合征(MRKHS),以及完全阻塞生殖道的宫颈和阴道异常。染色体核型异常不能排除 MRKHS 的诊断。Müllerian 异常和泌尿系统及骨骼系统相关畸形的家族病例强烈提示复杂的遗传病因,但迄今为止,绝大多数病例的分子机制仍不清楚。原发性闭经也可能是完全雄激素不敏感综合征、类固醇 5α-还原酶 2 缺乏症、17β-羟类固醇脱氢酶 3 缺乏症和 Leydig 细胞发育不全 1 型的首发表现;因此,这些疾病应在先天性无子宫和阴道的鉴别诊断中考虑。这些病例中的大多数可以进行分子诊断。