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Bo Shen, Ana E. Bennett, Udayakumar Navaneethan, IgG4-associated Pouchitis, Inflammatory Bowel Diseases, Volume 17, Issue 5, 1 May 2011, Pages 1247–1248, https://doi.org/10.1002/ibd.21441
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To the Editor:
Elevation of serum IgG4 is one of the hallmarks for autoimmune pancreatitis (AIP). Immunostaining of the affected organs reveals abundant infiltration with IgG4-positive plasma cells.1,–3 It was postulated that AIP may represent a spectrum of autoimmune disorders, involving extrapancreatic organs including the bile ducts, gallbladder, salivary glands, retroperitoneum, lymph nodes, pleura, lungs, kidneys, duodenum, and colon.
We report a 27-year-old Caucasian male patient with ulcerative colitis (UC) diagnosed at age 15. He developed adenocarcinoma, for which he underwent staged restorative proctocolectomy with ileal pouch-anal anastomosis (IPAA) and mucosectomy in 2005. He had not received biological therapy before colectomy. Computerized tomography scans of the abdomen in 2006 and 2007 showed diffuse “fatty liver changes” and normal extra- and intrahepatic ducts and pancreas.
He was referred to our Pouchitis Clinic in 2008 for persistent diarrhea, abdominal cramps, and urgency. Pouchoscopy showed inflammatory changes of in the mucosa of the pouch body and a long segment of the afferent limb, with granularity, diffuse small ulcers, friability, and exudates. Light microscopic examination of the pouch biopsy showed acute and chronic inflammatory changes with neutrophil and mononuclear cell-mediated injury and epithelia alterations classified as “indefinite for dysplasia.” Serum IgG4 was 194 mg/dL (normal range 11-112) without history of pancreatitis. He was treated with oral budesonide (3 mg twice a day) for suspected “autoimmune pouchitis.” He had intermittent mild elevation of liver enzymes since the pouch surgery and work-up at a local institution in 2009, including liver biopsy, suggested that he may have “primary sclerosing cholangitis (PSC).”
