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[Chronic intestinal pseudo-obstruction syndrome in infants and associated anomalies]
R Destuynder1, A Menget, O Destuynder
1Services de Pédiatrie, CHU Saint-Jacques, Besançon.
Insights
This study details two infant cases of functional intestinal pseudo-obstruction linked with congenital anomalies. Early diagnosis and surgical intervention are crucial for managing these complex gastrointestinal and urologic conditions.
Area of Science:
- Pediatric Surgery
- Gastroenterology
- Developmental Biology
Background:
- Functional intestinal pseudo-obstruction (FIPO) is a rare motility disorder.
- Infantile FIPO often presents with severe gastrointestinal symptoms and requires complex management.
- Associated congenital anomalies can complicate diagnosis and treatment.
Observation:
- Two infants presented with FIPO, intestinal malrotation, and volvulus.
- Both cases involved aperistaltic bowel segments requiring surgical intervention (ileostomy/colostomy).
- Antenatal and postnatal diagnosis of megacystis was noted in both patients.
Findings:
- Surgical findings included short small bowel, intestinal malrotation, and complete aperistalsis.
- Post-operative complications such as adhesions necessitated further surgeries.
- Despite initial challenges, both children showed appropriate growth by age three.
Implications:
- Highlights the association between FIPO, intestinal malrotation, and urologic anomalies like megacystis.
- Emphasizes the need for comprehensive evaluation in infants with FIPO.
- Suggests that multidisciplinary management can lead to favorable outcomes in select cases.
Abstract:
The authors report 2 cases of functional intestinal pseudo-obstruction in infancy associated with intestinal and urologic anomalies. In the first case many intestinal obstructions occurred from the age of 3 weeks and the boy was operated on at 14 months of age. A short small bowel and an intestinal malrotation were found at surgery; the alimentary canal was completely aperistaltic, and an ileostomy was performed. Further operations were carried out, for obstruction due to adhesions, and lastly to perform another ileostomy. An antenatal diagnosis of megacystis had been made with ultrasonography. In the second case, the pseudo-obstruction syndrome occurred at the age of 1 month, due to a volvulus of the small bowel with malrotation. A second operation, one month later because of lack of intestinal transit showed an aperistaltic bowel and a colostomy was performed. The intestinal continuity was set up again at the age of 9 months and a fractional feeding was started. A megacystis was found during urologic investigations. Growth is correct for both children at 3 years of age. A review of the literature allowed to list the most frequent digestive or extradigestive anomalies associated with this syndrome.