Abstract

Case: Ulcerative colitis (UC) is a chronic inflammatory disorder of the colon with a growing prevalence. Traditionally, UC is thought to affect the rectum and colon exclusively, and spare the remainder of the gastrointestinal (GI) tract. The course of illness can range from mild colitis to full blown toxic megacolon requiring complete colonic resection. While various treatment options are available, total colectomy is viewed as potentially curative since it has been shown to result in complete cessation of symptoms. We report an unusual case of UC in a patient who presented with oral and esophageal ulcers that resolved completely following total colectomy. A 26-year-old male with no significant past medical history presented with a 1-week history of severe oral ulceration, odynophagia and fevers along with a 4-month history of intermittent bloody diarrhea. Infectious workup was negative. CT scan of the abdomen and pelvis showed evidence of pancolitis concerning for inflammatory bowel disease (IBD). An upper endoscopy (UE) showed multiple clear base, non-bleeding esophageal ulcers and a colonoscopy revealed severe ulceration spanning the entire colon. Patient’s hospital course was complicated by development of toxic megacolon requiring urgent total colectomy with end ileostomy. Following surgery patient reported significant improvement in overall symptoms. Biopsies from the colon revealed evidence of mucosal inflammation with crypt abscesses and absence of granulomas. Biopsies from the esophagus revealed acute esophagitis with no evidence of viral or fungal infection. Patient was discharged with 4 weeks of pantoprazole therapy and inhaled budesonide however no treatment for IBD was started. A follow up magnetic resonance enterography was obtained which showed no evidence of small bowel inflammation. Postoperatively patient continued to have improvement in symptoms with adequate weight gain. His budesonide and pantoprazole therapy was discontinued. An UE was repeated 5 months later which showed normal esophageal mucosa and no evidence of ulceration. Biopsies from the esophagus showed normal mucosa with no signs of inflammation. An ileoscopy was performed which was unremarkable and biopsies showed no evidence of small intestinal inflammation. Patient continues to do well off therapy with plans of undergoing a proctocolectomy with ileal pouch-anal anastomosis. There have been approximately 20 case reports that describe upper gastrointestinal (GI) involvement in patients with UC. These patients tend to be young and have evidence of severe UC. Clean base esophageal ulcers were observed in majority of these cases. Significant advances have been made in the treatment of UC however total proctocolectomy with ileo-anal anastomosis remains as a last resort therapy which can alleviate all symptoms including extraintestinal manifestations in up to 58% of UC patients. We therefore describe a rare case of UC that presented with primarily upper GI manifestations and emphasize the notion that UC may not be restricted to the colon only and that a total colectomy can serve as a treatment option for resolution of these symptoms.

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