Case Report
Copyright ©2008 The WJG Press and Baishideng. All rights reserved.
World J Gastroenterol. Nov 21, 2008; 14(43): 6748-6749
Published online Nov 21, 2008. doi: 10.3748/wjg.14.6748
An unusual presentation of primary sclerosing cholangitis
Franklin Whitney Goldwire, William E Norris, Jonathan M Koff, Zachary D Goodman, Milton T Smith
Franklin Whitney Goldwire, Internal Medicine Walter Reed Army Medical Center, Washington DC 20307, United States
William E Norris, Jonathan M Koff, Milton T Smith, Gastroenterology Walter Reed Army Medical Center, Washington DC 20307, United States
Zachary D Goodman, Armed Forces Institute of Pathology, Washington DC 20307, United States
Author contributions: Goldwire FW and Norris WE contributed equally to research and development of this work; and Koff JM, Goodman ZD, and Smith MT contributed to the final analysis of the data.
Correspondence to: Dr. Franklin Whitney Goldwire, Internal Medicine Walter Reed Army Medical Center, Washington DC 20307, United States. franklin.goldwire@mac.com
Telephone: +1-10-31461059 Fax: +1-202-7825183
Received: July 18, 2008
Revised: September 23, 2008
Accepted: September 30, 2008
Published online: November 21, 2008
Abstract

This case report describes the unusual presentation of a patient who had findings which were initially suggestive of a type IV  choledochal cyst. Her liver biopsy demonstrated biliary cirrhosis. She was treated with endoscopic retrograde cholangiopancreatography and biliary stent exchanges over one year. Her cholangiogram one year later demonstrated resolution of the biliary cystic dilation which led to her initial diagnosis, with beading and stricturing of the hepatic ducts consistent with primary sclerosing cholangitis. Liver-associated enzymes and physical findings also improved. A liver biopsy one year later demonstrated a marked improvement in hepatic fibrosis with no evidence of cirrhosis.

Keywords: Biliary cysts, Cholangiogram, Endoscopic retrograde cholangiopancreatography, Liver fibrosis, Primary sclerosing cholangitis