Metastatic epitheloid hemangioendothelioma (EHE): Role of systemic therapy and survival.

Cioffi, A.; Italiano, Antoîne; Penel, Nicolas; Berge, Yann; Toulmonde, Maud; Salas, Sussan J.; Chevreau, Christine; Cesne, Axel Le; Duffaud, F.; D’Adamo, David R.; Keohan, Mary Lou; Génébès, C.; Antonescu, Cristian; Coindre, J.M.; Nguyen, Binh Bui; Maki, Robert G.

doi:10.1200/jco.2011.29.15_suppl.10079

Cited by 13 publications

(14 citation statements)

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“…Furthermore, because of the lack of a statistical hypothesis, the observed data remain difficult to interpret. Nevertheless, in a previously reported retrospective study, there was no objective response, whereas sorafenib provided 2 partial responses in the current study. However, 1 of those objective responses lasted 2 months, which may be considered an unconfirmed partial response.…”

Section: Discussioncontrasting

confidence: 60%

“…The median progression‐free survival was 4.8 months, and the median OS was 41.0 months. Nevertheless, in that retrospective study, the number of patients who experienced progressive disease before starting systemic treatment was not reported . Limited experience with treatments like thalidomide, interferon 2a‐α, and bevacizumab have been reported (Table ).…”

Section: Discussionmentioning

confidence: 96%

“…The literature on the treatment of EHE with antiangiogenic agents is very limited and consists of a large retrospective study that was presented as an abstract and is not yet published, some case reports, and 2 prospective series, as summarized in Table . Cioffi et al reported a retrospective study of 42 other patients with metastatic EHE.…”

Section: Discussionmentioning

confidence: 99%

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Sorafenib in patients with progressive epithelioid hemangioendothelioma

Chevreau

Cesne

Ray‐Coquard

et al. 2013

Cancer

104

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BACKGROUND There is no standard treatment for progressive epithelioid hemangioendothelioma (EHE). To investigate the significant vascularization of EHE, the activity/toxicity of sorafenib in patients with progressive EHE was explored. METHODS In this multicenter, 1‐stage, phase 2 trial of sorafenib (800 mg daily), the primary endpoint, which was chosen by default, was the 9‐month progression‐free rate. All patients had documented progressive disease at the time of study entry. RESULTS Fifteen patients were enrolled between June 2009 and February 2011. The median age was 57 years (range, 31‐76 years), and the ratio of men to women was 9:6. The performance status was zero in 10 patients and 1 in 5 patients. Twelve patients had metastases, mainly in the lung (12 patients), liver (5 patients), and bone (3 patients). Five patients had received prior chemotherapy (doxorubicin in 5 patients and taxane in 3 patients). The median sorafenib treatment duration was 124 days (range, from 27 to >271 days). Seven patients required dose reductions or transient treatment discontinuation. The 9‐month progression‐free rate was 30.7% (4 of 13 patients). The 2‐month, 4‐month, and 6‐month progression‐free rate was 84.6% (11 of 13 patients), 46.4% (6 of 13 patients), and 38.4% (5 of 13 patients), respectively. Two partial responses were observed that lasted 2 months and 9 months. CONCLUSIONS Further clinical trials exploring sorafenib as treatment of progressive EHE are needed. Cancer 2013;119:2639–2644. © 2013 American Cancer Society.

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Section: Discussioncontrasting

confidence: 60%

Section: Discussionmentioning

confidence: 96%

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Sorafenib in patients with progressive epithelioid hemangioendothelioma

Chevreau

Cesne

Ray‐Coquard

et al. 2013

Cancer

104

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“…However, partial responses were very rare (in 2 of the 15 patients), and the time to progression was 6 months [ 5 ]. Cioffi et al [ 6 ] reported a retrospective study of 42 additional patients with metastatic EHE; 34 of those patients received systemic treatment, including conventional doxorubicin-containing regimens, other chemotherapy regimens, and angiogenesis-targeting drugs or tyrosine ki nase inhibitors, but there was no objective response. The median progression-free survival was 4.8 months, and the median overall survival was 41.0 months.…”

Section: Discussionmentioning

confidence: 99%

Sorafenib Monotherapy in a Patient with Unresectable Hepatic Epithelioid Hemangioendothelioma

et al. 2016

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A 49-year-old Japanese man had multiple huge masses (max. size 60 mm diameter) in his liver. These tumors were pathologically diagnosed by tumor biopsy as epithelioid hemangioendotheliomas of the liver. In this case, multiple liver tumors existed in both lobes. Also this patient did not agree to receive surgical resection including liver transplantation. Chemotherapy with sorafenib at a dose of 400 mg/body twice a day was started. About 6 months later, CT findings revealed that these tumors were shrinking slightly; 33 months later, the tumors obviously showed a partial response in accordance with the Response Evaluation Criteria in Solid Tumors (RECIST). Also 60 months later, the partial response continued with sorafenib monotherapy.

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“…17 However, in retrospective cohorts, mixed efficacy has been reported with antiangiogenic treatments. Sorafenib, thalidomide, and cyclophosphamide have shown limited activity, 11 whereas responses have been reported using celecoxib 7 and pazopanib. 18 Partial responses (PRs) have also been reported with interferon.…”

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confidence: 99%