Case Report
Copyright ©2009 The WJG Press and Baishideng. All rights reserved.
World J Gastroenterol. Sep 21, 2009; 15(35): 4446-4448
Published online Sep 21, 2009. doi: 10.3748/wjg.15.4446
Free perforation of the small intestine in collagenous sprue
Hugh J Freeman, Douglas L Webber
Hugh J Freeman, Departments of Medicine (Gastroenterology), University of British Columbia, Vancouver, BC V6T 1W5, Canada
Douglas L Webber, Departments of Pathology, University of British Columbia, Vancouver, BC V6T 1W5, Canada
Author contributions: Freeman HJ and Webber DL contributed equally to this work and wrote the paper.
Correspondence to: Dr. Hugh James Freeman, MD, FRCPC, FACP, Department of Medicine (Gastroenterology), University of British Columbia Hospital, 2211 Wesbrook Mall, Vancouver, BC V6T 1W5, Canada. hugfree@shaw.ca
Telephone: +1-604-8227216 Fax: +1-604-8227236
Received: June 18, 2009
Revised: July 16, 2009
Accepted: July 23, 2009
Published online: September 21, 2009
Abstract

A 67-year-old man with celiac disease developed recurrent diarrhea, profound weakness and weight loss, with evidence of marked protein depletion. His clinical course was refractory to a strict gluten-free diet and steroid therapy. Postmortem studies led to definition of unrecognized collagenous sprue that caused ulceration and small intestinal perforation. Although PCR showed identical monoclonal T-cell populations in antemortem duodenal biopsies and postmortem jejunum, careful pathological evaluation demonstrated no frank lymphoma. Rarely, overt or even cryptic T-cell lymphoma may complicate collagenous sprue, however, small intestinal ulcers and perforation may also develop independently. The dramatic findings here may reflect an underlying or early molecular event in the eventual clinical appearance of overt T-cell lymphoma.

Keywords: Collagenous sprue; Intestinal perforation; Celiac disease; Refractory sprue; Intestinal neoplasms; Lymphoma