Department of Surgery, University of California, San Francisco, CA 94143, USA.
J Pediatr Surg. 2009 Dec;44(12):e31-3. doi: 10.1016/j.jpedsurg.2009.09.029.
Cloacal dysgenesis sequence (CDS) is a severe hindgut malformation occurring in 1:50,000 to 250,000 live births (Qureshi et al. Prenatal diagnosis of cloacal dysgenesis sequence: differential diagnosis from other forms of fetal obstructive uropathy. Fetal Diagn Ther 1998;13:69-74; Bargaje et al. Cloacal dysgenesis sequence. Ann Diagn Pathol 2008;12:62-66). It is characterized by a smooth perineum with no urethral, vaginal, or anal openings, and lack of labioscrotal development. Typically, the bladder, vagina, and colon each end blindly, although persistent cloaca without perineal orifice can be seen. With no egress for urine, infants have renal insufficiency and pulmonary hypoplasia, usually making CDS lethal (Sahinoglu Z et al. The prenatal diagnosis of cloacal dysgenesis in six cases: can the termination of pregnancy always be the first choice? Prenat Diagn 2004;24:10-16). Reported survivors have had a persistent urachus or have been spared the effects of oligohydramnios by the presence of a twin (Liang X. Cloacal dysgenesis sequence: observations in four patients, including three fetuses of second trimester gestation. Pediatr Dev Pathol 1998;1:281-288). We report a case of long-term survival, currently to 25 months of age, and renal transplantation in a monochorionic, diamniotic twin girl with CDS.
肛膜发育不良序列(CDS)是一种严重的后肠畸形,发病率为 150000 至 250000 活产儿中的 1 例(Qureshi 等人,产前诊断肛膜发育不良序列:与其他类型胎儿梗阻性尿路病的鉴别诊断。产前诊断 1998;13:69-74;Bargaje 等人,肛膜发育不良序列。安诊断病理 2008;12:62-66)。其特征为会阴光滑,无尿道、阴道或肛门开口,且缺乏阴唇阴囊发育。通常情况下,膀胱、阴道和结肠均盲端,尽管也可见无会阴开口的永存肛膜。由于尿液无法排出,婴儿会出现肾功能不全和肺发育不全,通常导致 CDS 致命(Sahinoglu Z 等人,6 例肛膜发育不良的产前诊断:终止妊娠是否总是首选?产前诊断 2004;24:10-16)。据报道,存活者存在永存脐尿管,或由于双胎的存在而免受羊水过少的影响(Liang X.,肛膜发育不良序列:4 例患者,包括 3 例中期妊娠胎儿的观察。儿科发育病理学 1998;1:281-288)。我们报告了一例 25 个月龄单绒毛膜、双羊膜双胎女性 CDS 患者的长期存活和肾移植病例。