Case Report
Copyright ©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. May 28, 2015; 21(20): 6409-6416
Published online May 28, 2015. doi: 10.3748/wjg.v21.i20.6409
Liver transplantation for a giant mesenchymal hamartoma of the liver in an adult: Case report and review of the literature
Jiang Li, Jin-Zhen Cai, Qing-Jun Guo, Jun-Jie Li, Xiao-Ye Sun, Zhan-Dong Hu, David KC Cooper, Zhong-Yang Shen
Jiang Li, Jin-Zhen Cai, Qing-Jun Guo, Jun-Jie Li, Xiao-Ye Sun, Zhong-Yang Shen, Department of Transplant Surgery, Tianjin First Central Hospital, Tianjin 300192, China
Zhan-Dong Hu, Department of Pathology, Tianjin First Central Hospital, Tianjin 300192, China
David KC Cooper, Thomas E Starzl Transplantation Institute, University of Pittsburgh, Pittsburgh, PA 15260, United States
Author contributions: Shen ZY, Cai JZ and Guo QJ performed transplant surgery and provided the intellectual content; Li JJ summarized the clinical data; Sun XY performed the follow-up; Hu ZD contributed to the pathology; Li J performed a literature review and wrote the initial manuscript; Cooper DKC contributed critical comments and revised the manuscript; all authors read, contributed to, and approved the final manuscript.
Supported by National Natural Science Foundation of China, No. 81400680; and the National High Technology Research and Development Program of China, No. 2012 AA021001.
Ethics approval: Approval from the Ethics Committee of the Tianjin First Central Hospital was obtained for this study.
Informed consent: The patient gave her written informed consent before entering the study and gave consent to the study protocol.
Conflict-of-interest: We declare that we have no financial and personal relationships with other people or organizations that can inappropriately influence our work. There is no professional or other personal interest of any nature or kind in any product, service and/or company that could be construed as influencing the position presented in, or the review of, the manuscript.
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: Zhong-Yang Shen, MD, Department of Transplant Surgery, Tianjin First Central Hospital, Nankai District of Tianjin Rehabilitation Road No. 24, Tianjin 300192, China. shenzy_009@sina.com
Telephone: +86-22-23626860 Fax: +86-22-23626199
Received: November 2, 2014
Peer-review started: November 3, 2014
First decision: December 26, 2014
Revised: January 26, 2015
Accepted: February 11, 2015
Article in press: February 11, 2015
Published online: May 28, 2015
Processing time: 209 Days and 11.8 Hours
Abstract

Mesenchymal hamartomas of the liver (MHLs) in adults are rare and potentially premalignant lesions, which present as solid/cystic neoplasms. We report a rare case of orthotopic liver transplantation in a patient with a giant MHL. In 2013, a 34-year-old female sought medical advice after a 2-year history of progressive abdominal distention and respiratory distress. Physical examination revealed an extensive mass in the abdomen. Computed tomography (CT) of her abdomen revealed multiple liver cysts, with the diameter of largest cyst being 16 cm × 14 cm. The liver hilar structures were not clearly displayed. The adjacent organs were compressed and displaced. Initial laboratory tests, including biochemical investigations and coagulation profile, were unremarkable. Tumor markers, including levels of AFP, CEA and CA19-9, were within the normal ranges. The patient underwent orthotopic liver transplantation in November 2013, the liver being procured from a 40-year-old man after cardiac death following traumatic brain injury. Warm ischemic time was 7.5 min and cold ischemic time was 3 h. The recipient underwent classical orthotopic liver transplantation. The recipient operative procedure took 8.5 h, the anhepatic phase lasting for 1 h without the use of venovenous bypass. The immunosuppressive regimen included intraoperative induction with basiliximab and high-dose methylprednisolone, and postoperative maintenance with tacrolimus, mycophenolate mofetil, and prednisone. The recipient’s diseased liver weighed 21 kg (dry weight) and measured 41 cm × 32 cm × 31 cm. Histopathological examination confirmed the diagnosis of an MHL. The patient did not experience any acute rejection episode or other complication. All the laboratory tests returned to normal within one month after surgery. Three months after transplantation, the immunosuppressive therapy was reduced to tacrolimus monotherapy, and the T-tube was removed after cholangiography showed no abnormalities. Twelve months after transplantation, the patient remains well and is fulfilling all normal activities. Adult giant MHL is extremely rare. Symptoms, physical signs, laboratory results, and radiographic imaging are nonspecific and inconclusive. Surgical excision of the lesion is imperative to make a definite diagnosis and as a cure. Liver transplantation should be considered as an option in the treatment of a non-resectable MHL.

Keywords: Liver; Mesenchymal hamartoma; Adult; Organ donor; After cardiac death; Transplantation

Core tip: Mesenchymal hamartoma of the liver is a rare disease in adults. Only 45 patients with this condition have been reported worldwide. This report presents a rare case of adult giant mesenchymal hamartoma of the liver that could not be treated by partial hepatectomy. Orthotopic liver transplantation relieved compression of other organs and avoided the risk of malignant change. Liver transplantation should be considered as an option in the treatment of non-resectable benign hepatic tumors.