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Neuronal intestinal and fibromuscular arterial dysplasias associated with intraluminal mucosal web
E Kotiloğlu1, A O Ciftci, F C Tanyel
1Department of Pediatric Pathology, Hacettepe University Medical Faculty, Ankara, Turkey.
Abstract:
A two-year-old boy presenting with the signs and symptoms of partial intestinal obstruction caused by a mucosal web in the ileum is presented. Histopathological evaluation following resection and primary anastomosis revealed neuronal intestinal dysplasia associated with fibromuscular arterial dysplasia limited to the segment proximal to the web and normal findings in the distal part. These findings and a brief review of the literature support the hypothesis that neuronal and fibromuscular dysplasias may develop on the basis of congenital obstructive lesions of the bowel.
Insights
A rare case of partial intestinal obstruction in a two-year-old boy revealed neuronal intestinal dysplasia and fibromuscular arterial dysplasia. These conditions may stem from congenital bowel obstructions, suggesting a link between developmental abnormalities.
Area of Science:
- Pediatric Surgery
- Gastroenterology
- Vascular Pathology
Background:
- Intestinal obstruction in children can arise from various congenital anomalies.
- Mucosal webs are a recognized, though uncommon, cause of pediatric bowel obstruction.
- Associated vascular and neural anomalies are less frequently reported in such cases.
Observation:
- A two-year-old male presented with symptoms indicative of partial intestinal obstruction.
- Surgical intervention identified an ileal mucosal web as the obstructive lesion.
- Histopathological examination was performed on the resected segment and surrounding tissues.
Findings:
- Neuronal intestinal dysplasia (NID) was diagnosed in the affected bowel segment.
- Fibromuscular arterial dysplasia (FMD) was concurrently identified in arteries proximal to the web.
- Distal bowel segments showed normal histological findings, localizing the pathology.
Implications:
- The findings support a hypothesis linking congenital obstructive lesions to the development of NID and FMD.
- This case highlights the potential for co-occurrence of intestinal and arterial dysplasia in pediatric obstructive lesions.
- Understanding this association may inform future diagnostic and therapeutic strategies for similar pediatric conditions.