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A Murine Model of Fetal Exposure to Maternal Inflammation to Study the Effects of Acute Chorioamnionitis on Newborn Intestinal Development
Published on: June 24, 2020
Congenital diarrhea with intestinal inflammation and epithelial immaturity
Insights
This case study details congenital inflammatory bowel disease and intestinal epithelial immaturity presenting as secretory diarrhea in an infant. The condition, linked to polyamine deficiency, led to fatal intractable diarrhea.
Area of Science:
- Pediatric Gastroenterology
- Neonatal Pathology
Background:
- Congenital inflammatory bowel disease (IBD) is rare in neonates.
- Intestinal epithelial immaturity can cause severe diarrhea.
- Secretory diarrhea in infants requires thorough etiological investigation.
Observation:
- An infant presented with severe secretory diarrhea without identifiable infectious, metabolic, or anatomical causes.
- Histopathology revealed acute and chronic inflammation, submucosal fibrosis, and flat mucosa in both small and large intestines.
- The infant exhibited a polyamine deficiency, potentially impacting epithelial development.
Findings:
- The infant had a unique presentation of congenital IBD with intestinal epithelial immaturity.
- Combined intestinal involvement (small and large) and specific histopathological features were noted.
- Polyamine deficiency was observed, possibly contributing to delayed epithelial maturation.
Implications:
- This case highlights a severe form of neonatal enteropathy.
- It suggests a potential link between polyamine deficiency, IBD, and epithelial maturation defects.
- The findings underscore the complexity of diagnosing and managing intractable neonatal diarrhea.
Abstract:
We report an interesting case of congenital inflammatory bowel disease and intestinal epithelial immaturity that presented as secretory diarrhea. No infectious, metabolic, or anatomical basis for these findings was identified. As differentiated from previous reports of neonatal enteropathies, this infant demonstrated involvement of both the small and large intestine with histopathologic findings of acute and chronic inflammation, extensive submucosal fibrosis, and "flat" small intestine mucosa. In addition, this patient had a polyamine deficiency (a primary or secondary phenomenon), which may have contributed to delayed epithelial maturation. These findings suggest that the inflammatory bowel disease and altered epithelial maturation contributed to a fatal intractable diarrhea.
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