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Case Report - 6
Ulcerative Colitis-Like Chronic Colitis Confined to an Interposed Colon Segment In A Patient with Long-gap Esophageal Atresia: An Extremely Rare Complication after Colonic Interposition
Kübra Öztürk Yüzdemir 1, Hayriye Hızarcıoğlu Gülşen 2, Özlem Boybeyi 1, Tutku Soyer 1
1 Hacettepe University Faculty of Medicine Department of Pediatric Surgery
2 Hacettepe University Faculty of Medicine Department of Pediatrics, Pediatric Gastroenterelogy, Hepatology and Nutrition.
Background: Colonic interposition is a well-established surgical technique for esophageal replacement in patients with long-gap esophageal atresia (LGEA). Although long-term outcomes are generally favorable, the behavior of the transposed colon in the esophageal position over time remains incompletely understood. We present a pediatric case of chronic colitis suggestive of inflammatory bowel disease, localized predominantly to an interposed colonic segment.
Case Presentation: We report an extremely rare case of a male infant with trisomy 21, born at 31 weeks of gestation and diagnosed with isolated LGEA. He underwent cervical esophagostomy and followed by a retrosternal colonic interposition using the transverse and descending colon at 18 months. The postoperative period was uneventful, and oral feeding was successfully established. At age four, the patient presented with intermittent abdominal pain and hematochezia. Laboratory tests revealed mild anemia and elevated fecal calprotectin. Initial colonoscopy demonstrated mucosal inflammation in both the native colon and the interposed segment, for which he was started on oral mesalamine and a short course of corticosteroids. On follow-up endoscopy, inflammation was confined to the interposed colonic segment while the native colon and terminal ileum were endoscopically and histologically normal. Histopathologic evaluation revealed chronic inflammatory changes including crypt architectural distortion, focal mucosal hemorrhage, and lymphoplasmacytic infiltration. Given persistent segmental inflammation, azathioprine was added. The patient achieved clinical remission, and subsequent endoscopic evaluation demonstrated complete mucosal healing.
Conclusion: This case illustrates chronic inflammatory bowel disease-like colitis predominantly affecting an interposed colonic segment used for esophageal reconstruction. It highlights the need for long-term gastrointestinal surveillance in patients with colonic interposition and raises questions about the role of local immune and environmental factors in disease development in non-native gastrointestinal segments.