A Fetus with Imperforate Anus Developing Pulmonary Hypoplasia Triggered by Transient Urethral Obstruction.

A Fetus with Imperforate Anus Developing Pulmonary Hypoplasia Triggered by Transient Urethral Obstruction.
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DOI:
10.1155/2021/9950578
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发表时间:
2021
影响因子:
--
通讯作者:
Osuga Y
Osuga Y
中科院分区:
其他
文献类型:
--
作者:
Toshimitsu M;Iriyama T;Sayama S;Suzuki K;Kakiuchi S;Ichinose M;Seyama T;Sone K;Kumasawa K;Nagamatsu T;Fujii T;Osuga Y

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肺发育不全是一种罕见的实体胎儿与immunanus。根据胎儿肠道扩张和19周时肠道钙化的存在,胎儿被诊断为高型肛门直肠瘘伴直肠尿道瘘。随着妊娠的进展,胎儿超声检查显示从妊娠28周开始出现肺发育不全、进行性肠扩张和持续性羊水过少,尽管膀胱充满液体,但无输尿管积水或肾积水。为了防止由于肠扩张和羊水过少引起的胸部压迫引起的肺发育不全进一步恶化,在妊娠32周时通过剖宫产分娩了一名男性新生儿。新生儿出现呼吸衰竭,需要全面呼吸支持。虽然导尿管没有通过尿道进入膀胱在出生时,膀胱尿道造影显示瘘和狭窄的下尿路通畅。产前和产后的研究结果强烈表明,结肠中的胎粪可能进入阴茎的尿道,导致尿液流出到羊膜腔的物理阻塞,导致尿液从膀胱通过瘘管流入结肠,从而导致随后的羊水过少和肠道扩张。据我们所知,这是第一个病例报告的胎儿与immunanus发展肺发育不全可能是由于尿道梗阻。
Pulmonary hypoplasia is a rare entity in a fetus with imperforate anus. The fetus was diagnosed with high-type imperforate anus with rectourethral fistula based on the dilated fetal bowel and the presence of bowel calcification at 19 weeks of gestation. As gestation advanced, fetal ultrasonography demonstrated development of pulmonary hypoplasia, progressive bowel dilation, and persistent oligohydramnios from 28 weeks of gestation despite a fluid-filled bladder without hydroureter or hydronephrosis. To prevent further worsening of pulmonary hypoplasia caused by thoracic compression due to bowel dilation and oligohydramnios, a male neonate was delivered by cesarean section at 32 weeks of gestation. The neonate showed respiratory failure requiring full respiratory support. Although a catheter did not pass through the urethra into the bladder at birth, cystourethrography revealed the patency of fistula and stenosed lower urinary tract. Prenatal and postnatal findings strongly suggested that the meconium in the colon might have passed into the urethra in the penis, resulting in the physical blockage of urine outflow to the amniotic space which leads urine flow from the bladder to the colon through the fistula, which resulted in subsequent oligohydramnios and bowel dilation. To the best of our knowledge, this is the first case report of a fetus with imperforate anus developing pulmonary hypoplasia possibly due to urethral obstruction.
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