School of Medicine, Loma Linda University, Loma Linda, CA, USA.
Division of Pediatric Surgery, Loma Linda University Children's Hospital, Loma Linda, CA, USA.
Am J Case Rep. 2024 Feb 27;25:e942203. doi: 10.12659/AJCR.942203.
BACKGROUND Cloaca malformations result from a disproportion of apoptosis, cell growth, and maturation. The range of cloacal malformations is extensive and diverse, with a lack of a straightforward classification system. Cloacal dysgenesis sequence (CDS), also known as urorectal septum malformation sequence, is a rare cloaca variant described as the absence of a perineal orifice. Prenatal magnetic resonance imaging and antenatal ultrasounds may reveal a cloacal malformation; however, many patients are not diagnosed with cloacal malformation until birth. CASE REPORT We present a case of a female neonate delivered by a 23-year-old G2P1T1A0L0 mother who had received comprehensive prenatal care. During pregnancy, bilateral multicystic dysplastic kidneys were identified prenatally, leading to the in utero placement of a vesicoamniotic shunt. The physical exam revealed a distended abdomen with reduced abdominal musculature and laxity, ascites, a vesicoamniotic shunt in place, absent urethra, ambiguous genitalia with no vaginal opening, no perineal opening, and clubfoot. Abdominal radiograph showed findings consistent with significant abdominal ascites. An exploratory laparotomy was performed that included diverting colostomy, mucous fistula creation, tube vaginostomy, removal of the vesicoamniotic shunt, and suprapubic tube placement. The patient recovered well from this operation with no complications. CONCLUSIONS CDS is an uncommon condition in pediatric patients, and although sonographic findings can reveal urinary tract abnormalities, prenatal imaging might not always identify CDS. Our case underscores the uniqueness of the case and the significance of early detection and immediate medical and surgical intervention.
泄殖腔畸形是由于细胞凋亡、细胞生长和成熟之间的比例失调引起的。泄殖腔畸形的范围广泛且多样,缺乏直接的分类系统。泄殖腔发育不良序列(CDS),也称为尿直肠隔畸形序列,是一种罕见的泄殖腔变体,表现为无会阴孔。产前磁共振成像和产前超声可能会发现泄殖腔畸形;然而,许多患者直到出生后才被诊断为泄殖腔畸形。
我们报告了一例由 23 岁 G2P1T1A0L0 母亲分娩的女性新生儿,该母亲接受了全面的产前护理。在怀孕期间,双侧多囊性发育不良肾脏在产前被发现,导致宫内放置了膀胱羊膜分流管。体格检查显示腹部膨隆,腹肌减少且松弛,有腹水,膀胱羊膜分流管在位,无尿道,外生殖器模糊,无阴道开口,无会阴开口,以及马蹄足。腹部 X 光片显示符合大量腹部腹水的发现。进行了剖腹探查术,包括结肠造口术、黏液瘘管形成术、管阴道造口术、移除膀胱羊膜分流管和耻骨上管放置术。患者术后恢复良好,无并发症。
CDS 在儿科患者中较为罕见,尽管超声检查结果可以显示泌尿道异常,但产前影像学检查可能并不总是能识别出 CDS。我们的病例强调了该病例的独特性,以及早期发现和立即进行医学和手术干预的重要性。