URINARY BLADDER NECROSIS - AN EXTRAINTESTINAL MANIFESTATION OF INFLAMMATORY BOWEL DISEASE IN A PEDIATRIC PATIENT

Abstract

Abstract Extraintestinal manifestations are common in inflammatory bowel disease (IBD), and genitourinary complications are known, including nephrolithiasis, fistulas, and ureteral obstruction. Bladder involvement is less common, and limited to cystitis including xanthogranulomatous cystitis. Bladder necrosis has not been previously described in association with IBD. Here, we describe an unusual case of IBD associated with total bladder necrosis at the time of initial diagnosis. An 8-year-old female with a history of chronic microcytic anemia presented with 1 week of hematochezia, hematuria, and fever with associated abdominal pain. Her exam was significant for mild lower quadrant abdominal tenderness including the suprapubic area, but was otherwise unremarkable. Blood work showed microcytic anemia and mild thrombocytopenia, elevated inflammatory markers, and hypoalbuminemia. Infectious workup was unremarkable. Upon admission, she developed oliguria and hypertension and rapidly progressed to renal failure requiring dialysis catheter placement and renal replacement therapy. A kidney biopsy revealed acute tubular injury but showed no glomerular involvement and had no evidence of hemolytic uremic syndrome or vasculitis. Imaging obtained including CT of her abdomen showed a thickened transverse colon and a thickened bladder wall with dilation of the renal collecting system, worse on the left. Bladder biopsy showed full-thickness necrosis of unclear etiology and had negative staining for microorganisms. She had bilateral nephrostomy tubes placed for drainage of her kidneys and her renal function subsequently returned to normal. MR imaging also revealed necrosis and confirmed complete bladder involvement. Her renal failure was presumed to be a secondary to ureterovesical junction obstruction from the bladder necrosis. She underwent esophagogastroduodenoscopy (EGD) and colonoscopy due to persistent hematochezia. The EGD was unremarkable. Her colonoscopy was grossly abnormal with evidence of colitis from the cecum to the rectum. Biopsies revealed both active and chronic inflammation with sparing of the terminal ileum, and immunohistochemical staining for infection was negative. Capsule endoscopy was largely unremarkable. She was diagnosed with ulcerative colitis and started on steroids and sulfasalazine. Her abdominal pain and stool consistency improved and her bloody stools resolved. Steroids were tapered and she continued sulfasalazine as maintenance therapy. This case presents a novel association of IBD and bladder necrosis. Vascular obstruction secondary to a hypercoagulable state might explain such an event, but this seems less likely given the diffuse nature of bladder involvement. A clinical index of suspicion for bladder involvement and/or necrosis is warranted in patients with IBD who present with obstructive urinary symptoms or hematuria.