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Canadian Journal of Gastroenterology
Volume 11, Issue 1, Pages 45-48
Brief Communication

Unusual Presentation of Primary Sclerosing Cholangitis

BC Pineau,1 LP Pattee,1 S McGuire,1 A Sekar,1,2 and LJ Scully1

1Department of Medicine, University of Saskatchewan, Saskatoon, Saskatchewan, Canada
2Department of Family Medicine and Department of Medicine, University of Ottawa, Ottawa, Ontario, Canada

Received 20 June 1995; Accepted 21 May 1996

Copyright © 1997 Hindawi Publishing Corporation. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


A 23-year-old man presenting with acute pancreatitis and autoimmune hemolytic anemia was diagnosed with primary sclerosing cholangitis (PSC) without evidence of ulcerative colitis. This constellation of rare associations constitutes a unique mode of presentation of PSC. Within two years he also developed ankylosing spondylitis with sacroiliitis. Disordered immune regulation as a major factor in the mechanism of injury in PSC is supported by its increased association with other immunologically mediated disorders, most notably ulcerative colitis. Autoimmune hemolytic anemia, however, has been reported to be associated with PSC on only two occasions, and ankylosing spondylitis in the absence of ulcerative colitis is also unusual. In addition, the presentation of PSC with acute pancreatitis has rarely been described. This patient presented with several unusual features of PSC.