Case Report
Copyright ©2008 The WJG Press and Baishideng. All rights reserved.
World J Gastroenterol. Jul 14, 2008; 14(26): 4241-4244
Published online Jul 14, 2008. doi: 10.3748/wjg.14.4241
Hemobilia as the initial manifestation of cholangiocarcinoma in a hemophilia B patient
Anastassios C Manolakis, Andreas N Kapsoritakis, Antonis D Tsikouras, Fotis D Tsiopoulos, Athanassios K Psychos, Spyros P Potamianos
Anastassios C Manolakis, Andreas N Kapsoritakis, Antonis D Tsikouras, Fotis D Tsiopoulos, Athanassios K Psychos, Spyros P Potamianos, Department of Gastroenterology, University of Thessaly, School of Medicine, Larissa 41447, Greece
Author contributions: Manolakis AC drafted the manuscript; Kapsoritakis AN offered the conception and designed the study; Tsikouras AD was involved in the management of the patient; Tsiopoulos FD was involved in the acquisition of data; Psychos AK carried out the endoscopic procedures; Potamianos SP was responsible for the management of the patient and the supervisor of the study.
Correspondence to: Dr. Spyros P Potamianos, Department of Gastroenterology, University of Thessaly, 16 Sklirou st, Larissa 41447, Greece. spotam@med.uth.gr
Telephone: +30-2410-536823
Fax: +30-2410-536823
Received: January 17, 2008
Revised: April 9, 2008
Accepted: April 16, 2008
Published online: July 14, 2008
Abstract

Hemobilia is a rare manifestation of hemophilia and is usually iatrogenic following liver biopsy. There are only few reports of spontaneous hemobilia in hemophilia patients. Cholangiocarcinoma is a well-established cause of hemobilia. We describe a case of a 70-year-old male, with known haemophilia B and a past history of papillotomy, who presented with classical symptoms of hemobilia. The initial diagnostic work-up failed to demonstrate a potential cause of bleeding other than the coagulopathy. Three months later, he was readmitted to our hospital with a second episode of hemobilia. During the second work-up, a cholangiocarcinoma was diagnosed both by imaging studies and by a significant elevation of cancer antigen 19-9. Although hemobilia could be attributed to hemophilia, especially in a patient with previous papillotomy, an underlying malignancy of the biliary tree should be suspected.

Keywords: Hemobilia; Hemophilia; Cholangiocarcinoma; Cancer antigen 19-9; Cholangiopancreatography