Case Report
Copyright ©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Aug 28, 2015; 21(32): 9675-9682
Published online Aug 28, 2015. doi: 10.3748/wjg.v21.i32.9675
Granulomatous reaction in hepatic inflammatory angiomyolipoma after chemoembolization and spontaneous rupture
Keita Kai, Atsushi Miyosh, Shinichi Aishima, Kota Wakiyama, Shunya Nakashita, Shinji Iwane, Shinya Azama, Hiroyuki Irie, Hirokazu Noshiro
Keita Kai, Department of Pathology, Saga University Hospital, Saga 849-8501, Japan
Atsushi Miyosh, Shinichi Aishima, Kota Wakiyama, Hirokazu Noshiro, Department of Surgery, Saga University Faculty of Medicine, Saga 849-8501, Japan
Shinichi Aishima, Department of Pathology and Microbiology, Saga University Faculty of Medicine, Saga 849-8501, Japan
Shunya Nakashita, Shinji Iwane, Department of Internal Medicine, Saga University Faculty of Medicine, Saga 849-8501, Japan
Shinya Azama, Hiroyuki Irie, Department of Radiology, Saga University Faculty of Medicine, Saga 849-8501, Japan
Author contributions: Kai K is the main author of this article; Miyoshi A, Wakiyama K, Nakashita S, Iwane S and Azama S were attending doctors and performed clinical treatment and diagnosis including surgical operation; Aishima S, Irie H and Noshiro H reviewed the manuscript; all authors have read and approved the final manuscript.
Institutional review board statement: We consider that ethics approval is not needed because this is a single case report without personal information.
Informed consent statement: Written informed consent was obtained.
Conflict-of-interest statement: The authors declare that there are no conflicts of interest.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Keita Kai, MD, Department of Pathology, Saga University Hospital, Nabesima 5-1-1, Saga City, Saga 849-8501, Japan. kaikeit@cc.saga-u.ac.jp
Telephone: +81-952-343264 Fax: +81-952-342055
Received: February 20, 2015
Peer-review started: February 23, 2015
First decision: March 26, 2015
Revised: April 13, 2015
Accepted: June 9, 2015
Article in press: June 10, 2015
Published online: August 28, 2015
Processing time: 188 Days and 21.3 Hours
Abstract

A 77-year-old Japanese woman was transported to a nearby hospital due to sudden abdominal pain and transient loss of consciousness. Abdominal computed tomography (CT) suggested hemoperitoneum and hepatic nodule. She was conservatively treated. Contrast-enhanced CT two months later revealed an increased mass size, and the enhancement pattern suggested the possibility of hepatocellular carcinoma (HCC). Under a clinical diagnosis of HCC, transcatheter arterial chemoembolization (TACE) was performed. A subsequent imaging study revealed that most of the lipiodol used for the embolization was washed out. Therefore, surgical resection was performed. Histologically, the nodule contained numerous inflammatory cells including small lymphocytes, plasma cells and macrophages. Notably, epithelioid granulomatous features with multinucleated giant cells were observed in both the nodule and background liver. Some of the multinucleated giant cells contained oil lipid. Among the infiltrating inflammatory cells, spindle-shaped, histiocytoid or myoid tumor cells with eosinophilic cytoplasm were found. The tumor cells were positive for Melan A and HMB45. The nodule contained many IgG4-positive plasma cells; these were counted and found to number 72.6 cells/HPF (range: 61-80). The calculated IgG4:IgG ratio was 33.2%. The nodule was finally diagnosed as previously ruptured inflammatory angiomyolipoma modified by granulomatous reaction after TACE.

Keywords: Angiomyolipoma; Inflammatory; Rupture; Lipiodol; Liver

Core tip: Hepatic angiomyolipoma (AML) is a rare benign neoplasm. Particularly, the inflammatory variant of hepatic AML is extremely rare. Spontaneous rupture of hepatic AML is also extremely rare phenomenon. The authors herein present a case of spontaneously ruptured hepatic inflammatory AML associated with extensive granulomatous reaction induced by chemoembolization. This case is not only rare, but also suggests that chemoembolization rarely causes granulomatous reaction in hepatic tissue.