Background <p>Gastroschisis is a congenital defect of the abdominal wall with evisceration of bowels and stomach without a covering sac. Sutureless reduction of the abdominal contents can be done gradually using a silo. This condition may be associated with other gastrointestinal problems such as bowel atresia or stenosis, perforation, necrotizing enterocolitis, malrotation, or bowel dysmotility. Pyloric stricture post-sutureless silo reduction is a rare form of complex gastroschisis that has not been reported before in the literature. We discuss the diagnostic challenges and management of this rare case.</p> Case presentation <p>A premature female was born at 35 weeks, with a birth weight of 2.2&#xa0;kg, and had gastroschisis. Evisceration of the stomach, small bowels, and large bowels can be seen via an abdominal wall defect. There was bowel matting at the jejunum and right colon, causing a narrowing effect. Small bowel contents were decompressed proximally into the stomach and aspirated via an orogastric tube. Abdominal contents were successfully reduced using a spring-loaded silo. The abdominal wall defect was covered with the umbilical stump and was allowed to heal spontaneously. Feeding was started after one month and increased to full enteral feedings. She developed recurrent episodes of vomiting and was treated for gastroesophageal reflux disease and bowel dysmotility. Despite optimal medical treatment, clinical observation showed a progressive nature of obstruction. Vomiting episodes became more frequent over time, and total tolerable feeds were progressively less in amount. An upper gastrointestinal contrast study showed holdup of contrast in the stomach with delayed emptying into the small bowel. Esophagogastroscopy confirmed a pinpoint opening at the pylorus. Laparotomy, excision of pyloric stricture, and gastroduodenostomy were done. The child recovered well after surgery and was discharged home.</p> Conclusion <p>Pyloric stricture is a rare sequel following sutureless silo reduction of gastroschisis. The pathophysiology behind this phenomenon remains unknown. Recurrent feeding intolerance in a patient with gastroschisis, not responding to medical treatment, warrants further investigations to rule out mechanical obstruction. Surgical resection and gastroduodenostomy are curative.</p>

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Unveiling complexity: a case report on management of pyloric stricture in complex gastroschisis

  • Siti Nurr Syafeena Rais,
  • Mohd Shahrulsalam Mohd Shah,
  • Nurul Adzimah Kamaruddin,
  • Shung Ken Tan,
  • Tammy Teoh Han Qi,
  • Hooi Hooi Tan,
  • Mohan Arunasalam Nallusamy

摘要

Background

Gastroschisis is a congenital defect of the abdominal wall with evisceration of bowels and stomach without a covering sac. Sutureless reduction of the abdominal contents can be done gradually using a silo. This condition may be associated with other gastrointestinal problems such as bowel atresia or stenosis, perforation, necrotizing enterocolitis, malrotation, or bowel dysmotility. Pyloric stricture post-sutureless silo reduction is a rare form of complex gastroschisis that has not been reported before in the literature. We discuss the diagnostic challenges and management of this rare case.

Case presentation

A premature female was born at 35 weeks, with a birth weight of 2.2 kg, and had gastroschisis. Evisceration of the stomach, small bowels, and large bowels can be seen via an abdominal wall defect. There was bowel matting at the jejunum and right colon, causing a narrowing effect. Small bowel contents were decompressed proximally into the stomach and aspirated via an orogastric tube. Abdominal contents were successfully reduced using a spring-loaded silo. The abdominal wall defect was covered with the umbilical stump and was allowed to heal spontaneously. Feeding was started after one month and increased to full enteral feedings. She developed recurrent episodes of vomiting and was treated for gastroesophageal reflux disease and bowel dysmotility. Despite optimal medical treatment, clinical observation showed a progressive nature of obstruction. Vomiting episodes became more frequent over time, and total tolerable feeds were progressively less in amount. An upper gastrointestinal contrast study showed holdup of contrast in the stomach with delayed emptying into the small bowel. Esophagogastroscopy confirmed a pinpoint opening at the pylorus. Laparotomy, excision of pyloric stricture, and gastroduodenostomy were done. The child recovered well after surgery and was discharged home.

Conclusion

Pyloric stricture is a rare sequel following sutureless silo reduction of gastroschisis. The pathophysiology behind this phenomenon remains unknown. Recurrent feeding intolerance in a patient with gastroschisis, not responding to medical treatment, warrants further investigations to rule out mechanical obstruction. Surgical resection and gastroduodenostomy are curative.