腹腔镜胃十二指肠吻合（Billroth I）治疗Carmi综合征II型幽门闭锁1例

IF 0.2 Q4 PEDIATRICS

Journal of Pediatric Surgery Case Reports Pub Date : 2025-09-23 DOI:10.1016/j.epsc.2025.103113

Joel Cazares , Jorge Colín-Garnica , Eduardo de la Rosa-Bustamante , Rubi Beatriz Martínez-Carmona , Arturo Guillen-Cárdenas , Jorge Alberto Cantú-Reyes

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引用次数: 0

摘要

幽门闭锁占所有先天性肠梗阻的不到1%，估计发病率为1:10万活产。它与大疱性表皮松解症有关，称为Carmi综合征，虽然罕见，但文献记载充分。微创技术可用于复杂新生儿疾病的外科治疗。病例报告一名男婴在妊娠35周时顺产给一名16岁的母亲，据报道父母之间有血缘关系。出生时，患者表现为广泛的大疱性皮肤病变，累及四肢、胸部和面部，同时伴有阳性的Nikolsky征，这些发现强烈怀疑为大疱性表皮松解症。出生体重2000克，不需要复苏操作。开始肠内喂养后，患者出现非胆汁性呕吐。腹部x线片显示单个胃泡，远端无气体。造影证实胃出口梗阻。第三天的内窥镜检查显示完全幽门阻塞和粘膜脆弱。行腹腔镜检查，确认II型幽门闭锁。腹腔镜胃十二指肠造口术（Billroth I）成功进行，无术中并发症。术后造影证实吻合口通畅。不幸的是，由于广泛的皮肤受累，患者发生败血症，并于术后第三天死亡。结论微创胃十二指肠吻合术（Billroth I）治疗新生儿幽门闭锁在技术上是可行的。

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Laparoscopic gastroduodenal anastomosis (Billroth I) for the management of type II pyloric atresia in Carmi syndrome: a case report

Introduction

Pyloric atresia accounts for less than 1 % of all congenital intestinal obstructions, with an estimated incidence of 1:100,000 live births. Its association with epidermolysis bullosa, known as Carmi syndrome, is rare but well documented. Minimally invasive techniques can be employed in the surgical management of complex neonatal conditions.

Case report

A male neonate was delivered vaginally at 35 weeks of gestation to a 16-year-old mother, with reported consanguinity between the parents. At birth, he presented with widespread bullous skin lesions involving the extremities, thorax, and face, along with a positive Nikolsky sign, findings that raised strong suspicion for epidermolysis bullosa. Birth weight was 2000 g, and no resuscitative maneuvers were required.

Following the initiation of enteral feeding, the patient developed non-bilious vomiting. Abdominal radiography showed a single gastric bubble with absence of distal gas. A contrast study confirmed gastric outlet obstruction. Endoscopy on day three revealed complete pyloric occlusion and mucosal fragility. A laparoscopic procedure was performed, confirming type II pyloric atresia. A Laparoscopic gastroduodenostomy (Billroth I) was successfully carried out without intraoperative complications. Postoperative contrast imaging confirmed adequate anastomotic patency. Unfortunately, the patient developed sepsis due to extensive skin involvement and died on postoperative day three.