Case Report
Copyright ©2014 Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Jun 14, 2014; 20(22): 7049-7054
Published online Jun 14, 2014. doi: 10.3748/wjg.v20.i22.7049
Long-term durable response to lenalidomide in a patient with hepatic epithelioid hemangioendothelioma
Maria Caterina Pallotti, Margherita Nannini, Claudio Agostinelli, Simona Leoni, Valerio Di Scioscio, Anna Mandrioli, Cristian Lolli, Maristella Saponara, Stefano Pileri, Luigi Bolondi, Guido Biasco, Maria Abbondanza Pantaleo
Maria Caterina Pallotti, Margherita Nannini, Anna Mandrioli, Cristian Lolli, Maristella Saponara, Guido Biasco, Maria Abbondanza Pantaleo, Department of Specialized, Experimental, and Diagnostic Medicine, S. Orsola-Malpighi Hospital, University of Bologna, 40138 Bologna, Italy
Claudio Agostinelli, Stefano Pileri, Hematopathology Section, Department of Experimental, Diagnostic, and Specialty Medicine, S. Orsola-Malpighi Hospital, University of Bologna, 40138 Bologna, Italy
Simona Leoni, Luigi Bolondi, Division of Internal Medicine, Department of Digestive Disease and Internal Medicine, S. Orsola-Malpighi Hospital, University of Bologna, 40138 Bologna, Italy
Valerio Di Scioscio, Department of Radiology, S. Orsola-Malpighi Hospital, University of Bologna, 40138 Bologna, Italy
Guido Biasco, Maria Abbondanza Pantaleo, Interdepartmental Centre for Cancer Research “G. Prodi”, University of Bologna, 40138 Bologna, Italy
Author contributions: Pallotti MC and Pantaleo MA contributed equally to this work, designed and performed research, contributed analytic tools, analyzed data, and wrote the paper; Nannini M, Agostinelli C, Leoni S, Di Scioscio V, Mandrioli A, Lolli C, Saponara M, Pileri S, Bolondi L and Biasco G contributed equally to this work, performed research, and contributed analytic tools.
Correspondence to: Maria Caterina Pallotti, MD, Department of Specialized, Experimental, and Diagnostic Medicine, S. Orsola-Malpighi Hospital, University of Bologna, Via Massarenti 9, 40138 Bologna, Italy. mcaterinapallotti@gmail.com
Telephone: +39-51-6363812 Fax: +39-51-6364037
Received: October 24, 2013
Revised: November 29, 2013
Accepted: January 19, 2014
Published online: June 14, 2014
Processing time: 235 Days and 0.1 Hours
Abstract

Epithelioid hemangioendothelioma (EH) is a rare tumor arising from the vascular endothelial cells of soft tissue or visceral organs. The most common visceral site is the liver, where it is often involved in a multifocal manner known as hepatic EH (HEH). Surgical resection with curative intent represents the gold standard therapy. When surgery is not feasible, or in cases of metastatic disease, no standard medical treatment is currently indicated. In small series, drugs with anti-angiogenic activity (such as bevacizumab, sorafenib, thalidomide, and lenalidomide) have been proposed with promising results. We describe a 73-year-old man with multifocal non-resectable HEH treated with lenalidomide. Disease status was evaluated by abdominal ultrasound and magnetic resonance every four months. The patient was treated for a total of 39 mo with prolonged disease stabilization and, at the time of writing, is still under treatment with a good tolerance profile. During a short period of treatment discontinuation, the disease showed slight progression that immediately resolved after the reintroduction of lenalidomide. Lenalidomide may represent a valid treatment option for HEH due to its anti-angiogenic and antineoplastic activities. This preliminary result merits further study in a large series.

Keywords: Hepatic epithelioid hemangioendothelioma; Liver; Lenalidomide; Magnetic resonance; Abdominal ultrasound

Core tip: Hepatic epithelioid hemangioendothelioma (HEH) is a rare tumor. Currently, no standard medical treatment is indicated in unresectable disease. We describe the case of a patient with HEH who obtained stable disease after 40 mo of treatment with lenalidomide. Tolerance to treatment was very good. This report highlights the possibility of obtaining very long stable disease with an anti-angiogenesis drug. In recent years, the introduction of angiogenesis inhibitors has offered an interesting medical approach to this rare tumor.