Hepatic epithelioid hemangioendothelioma is a rare liver mesenchymatous tumor with an epithelial aspect developed from endothelial cells with a lack of validated therapeutic options at metastatic stage. Metronomic chemotherapy has been described as an anti-angiogenic therapy leading to the depletion of circulating endothelial progenitors. We report the results of two patients treated for metastatic hemangioendothelioma with metronomic cyclophosphamide chemotherapy. Following initiation of metronomic cyclophosphamide chemotherapy (50mg once a day continuously), the two patients exhibited significant clinical improvement and decrease in metastasis size without any clinically relevant side-effect. Metronomic cyclophosphamide could be proposed as a new therapeutic option to treat metastatic hepatic epithelioid hemangioendothelioma.
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