Case Report
Copyright ©2006 Baishideng Publishing Group Co., Limited. All rights reserved.
World J Gastroenterol. Mar 14, 2006; 12(10): 1645-1648
Published online Mar 14, 2006. doi: 10.3748/wjg.v12.i10.1645
Spontaneous regression of hepatic inflammatory pseudotumor with primary biliary cirrhosis: Case report and literature review
Hiroshi Koide, Ken Sato, Toshio Fukusato, Kenji Kashiwabara, Noriaki Sunaga, Takafumi Tsuchiya, Saeko Morino, Naondo Sohara, Satoru Kakizaki, Hitoshi Takagi, Masatomo Mori
Hiroshi Koide, Ken Sato, Noriaki Sunaga, Takafumi Tsuchiya, Saeko Morino, Naondo Sohara, Satoru Kakizaki, Hitoshi Takagi, Masatomo Mori, Department of Medicine and Molecular Science, Gunma University Graduate School of Medicine, Maebashi, Gunma 371-8511, Japan
Toshio Fukusato, Department of Pathology, Teikyo University School of Medicine, Itabashi-ku, Tokyo 173-8605, Japan
Kenji Kashiwabara, Clinical Pathology, Gunma University Graduate School of Medicine, Maebashi, Gunma 371-8511, Japan
Author contributions: All authors contributed equally to the work.
Correspondence to: Ken Sato, MD, PhD, Department of Medicine and Molecular Science, Gunma University Graduate School of Medicine, 3-39-15 Showa-machi, Maebashi, Gunma 371-8511, Japan. satoken@showa.gunma-u.ac.jp
Telephone: +81-27-2208127 Fax: +81-27-2208136
Received: June 7, 2005
Revised: June 28, 2005
Accepted: July 28, 2005
Published online: March 14, 2006
Abstract

Hepatic inflammatory pseudotumor (IPT) is a rare benign non-neoplastic lesion characterized by proliferating fibrous tissue infiltrated by inflammatory cells. The exact etiology of IPT remains unclear. Although the association of IPT with systemic inflammatory disorders has been well established, a specific relationship with cholangitis is distinctly rare. We report a case of spontaneous regression of hepatic IPT with primary biliary cirrhosis (PBC). To date, only two cases of IPT with PBC have been reported. In our case, however, IPT developed during the course of improvement of cholangitis of PBC induced by effective treatment, differing from two previously reported cases. Our case indicates that the development of IPT does not also relate to the activity of cholangitis and/or hyper gamma-globulinemia, since our case was confirmed radiologically to be free of IPT when biliary enzymes and immunoglobulins were much higher than the corresponding values on admission. Comparison of our case with the two previously reported cases suggests that IPT occurring with PBC does not represent the same disease entity or be a bystander for PBC.

Keywords: Hepatic inflammatory pseudotumor; Primary biliary cirrhosis; Spontaneous regression; Ursodeoxycholic acid; Bezafibrate