Abstract

Abstract Background Emphysematous cystitis (EC) is characterized by the presence of air within the bladder walls, a complication of infection by gas producing organisms within the urinary system. However, rare cases of EC in the setting of infectious gastroenteritis have been reported suggesting an alternate etiology. We report a case of EC in the setting of severe Crohn’s colitis with no clinical evidence of urinary tract infection. Aims This report outlines the rare case of a patient with active Crohn’s disease who presented with bloody diarrhea, anemia, hypoalbuminemia and initial radiological evidence of EC. We expand on the subsequent clinical investigations, diagnosis, treatment plan and hospital course of the patient. Methods Case report, review of literature. Results A 43-year old female presented with a 2-month history of bloody diarrhea consisting of 8–12 bowel movements a day, 10 kg of weight loss and peripheral edema. She also had multiple ulcerated lesions on her abdominal wall and in the perianal region. Initial CT scan was significant for pancolitis, anasarca and EC. The follow-up CT cystogram, flexible cystoscopy and pelvic MRI confirmed the diagnosis of EC and ruled out any fistulous tracts in the pelvis including enterovesical/colovesical fistula. The patient did not report any urinary symptoms and the urinalysis was within normal limits. Infectious workup including urine and blood cultures, stool ova and parasite, stool C. difficile PCR, TB IGRA, Hep A, Hep B, Hep C and HIV serologies were all negative. Despite the paucity of infectious findings, the EC was empirically treated with an intravenous third-generation cephalosporin. Colonoscopy was significant for multiple ulcerated and hyperemic areas with pseudopolyps all throughout the right, transverse and left colon. Biopsies confirmed Crohn’s colitis with no evidence of granulomata or dysplasia, and were also negative for CMV immunohistochemistry. The perianal and abdominal wall lesions were suspected to be pyoderma gangrenosum although biopsies were equivocal. The colitis was initially treated with intravenous steroids followed by biologic therapy with Infliximab. While there was some interval improvement of symptoms, the in-hospital course was further complicated by a rectal bleed requiring surgical obliteration of the rectal artery, then a colonic perforation requiring total colectomy. Conclusions This is a rare case of EC in a patient with severe Crohn’s colitis. There was no clinical evidence of urinary tract infection or fistulising disease. According to our review, this is the first reported incident of EC in a patient with inflammatory bowel disease without any prior intra-abdominal surgeries. While active Crohn’s disease alone is a critical illness, we conclude that concomitant EC is a poor prognostic factor. Funding Agencies None

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