Escobar L F, Weaver D D, Bixler D, Hodes M E, Mitchell M
Am J Dis Child. 1987 Sep;141(9):1021-4. doi: 10.1001/archpedi.1987.04460090098038.
We encountered six female infants with a specific pattern of developmental abnormalities of the urogenital and lower intestinal tracts. The anomalies included ambiguous genitalia, lack of perineal openings, and müllerian and urinary tract anomalies. Each patient had normal female chromosomes and normal adrenal gland function. We believe that this combination of anomalies represents a recognizable and specific sequence that is due to a failure of migration to and/or fusion of the urorectal septum with the cloacal membrane. This, in turn, we postulate, leads to persistence of the cloaca and cloacal membrane and failure of normal differentiation of the external genitalia. Persistence of the cloacal membrane results in absence of the urethral and vaginal openings and an imperforate anus. We propose calling this entity the urorectal septum malformation sequence.
我们遇到了六名患有泌尿生殖系统和下肠道特定发育异常模式的女婴。这些异常包括生殖器模糊、会阴开口缺失以及苗勒管和泌尿道异常。每名患者的女性染色体和肾上腺功能均正常。我们认为,这种异常组合代表了一种可识别的特定序列,这是由于尿直肠隔向泄殖腔膜迁移和/或与之融合失败所致。我们推测,这进而导致泄殖腔和泄殖腔膜持续存在,以及外生殖器正常分化失败。泄殖腔膜持续存在导致尿道和阴道开口缺失以及肛门闭锁。我们建议将这一实体称为尿直肠隔畸形序列。