Malignant transformation of infantile hemangioma to angiosarcoma: Response to chemotherapy with bevacizumab

Jeng, Michael; Fuh, Beng; Blatt, Julie; Gupta, Anita; Merrow, Arnold C.; Hammill, Adrienne M.; Adams, Denise M.

doi:10.1002/pbc.25067

Cited by 58 publications

(29 citation statements)

References 20 publications

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“…Therefore, our data favor the possibility that early lymphatic vascular malformations can progress to cutaneous lymphangiosarcomas. Similar progressions have been seen in some clinical observations (Jeng et al, 2014; Quarmyne et al, 2012; Rossi and Fletcher, 2002). …”

Section: Discussionsupporting

confidence: 88%

Constitutive Activation of mTORC1 in Endothelial Cells Leads to the Development and Progression of Lymphangiosarcoma through VEGF Autocrine Signaling

et al. 2015

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Summary Angiosarcoma/lymphangiosarcoma is a rare malignancy with poor prognosis. We generated a mouse model with inducible endothelial cell-specific deletion of Tsc1 to examine mTORC1 signaling in lymphangiosarcoma. Tsc1 loss increased retinal angiogenesis in neonates, and led to endothelial proliferative lesions from vascular malformations to vascular tumors in adult mice. Sustained mTORC1 signaling was required for lymphangiosarcoma development and maintenance. Increased VEGF expression in tumor cells was seen, and blocking autocrine VEGF signaling abolished vascular tumor development and growth. We also found significant correlations between mTORC1 activation and VEGF, HIF1α, and c-Myc expression in human angiosarcoma samples. These studies demonstrated critical mechanisms of aberrant mTORC1 activation in lymphangiosarcoma, and validate the mice as a valuable model for further study.

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Section: Discussionsupporting

confidence: 88%

Constitutive Activation of mTORC1 in Endothelial Cells Leads to the Development and Progression of Lymphangiosarcoma through VEGF Autocrine Signaling

et al. 2015

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“…A phase II trial concluded that bevacizumab is an effective and well-tolerated single-agent treatment for metastatic or locally advanced angiosarcoma (20). Several case reports demonstrated that combination therapy with bevacizumab and chemotherapy or radiotherapy may improve quality of life and survival in patients with metastatic angiosarcoma (21)(22)(23)(24)(25). In addition, another VEGF inhibitor, pazopanib, may prolong the PFS of metastatic non-adipocytic soft-tissue sarcoma after previous chemotherapy (26), particularly when used as maintenance therapy for PCA (16).…”

Section: Discussionmentioning

confidence: 99%

Comprehensive treatment of unresectable cardiac angiosarcoma: A case report and review of literature

Wang

Shi

Yang

et al. 2017

Molecular and Clinical Oncology

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Abstract. Cardiac angiosarcoma is a rare but lethal tumor that is difficult to diagnose and treat, due to its rapid local relapse and high incidence of systemic metastasis. The prognosis of cardiac angiosarcoma is dismal, with a mean life expectancy of only a few months. We herein report a case of unresectable angiosarcoma arising from the right atrium. The patient received first-line chemotherapy with weekly paclitaxel, and second-line therapy with vinorelbine and bevacizumab upon disease progression. The progression-free survival was 6 months and the overall survival was 7 months. The patient eventually succumbed to respiratory failure. A study of the present case and a review of the relevant literature suggest that treatment decisions for unresectable locally advanced or metastatic cardiac angiosarcoma are difficult, as the published literature on this disease mainly consists of case reports without sufficient data. Therefore, further clinical trials specific to the treatment of unresectable cardiac angiosarcoma are warranted.

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“…Similar transformation from vascular malformations to malignant vascular tumors have been seen in some clinical observations. 4,5 Our data indicate that sustained hyper-activation of mTORC1 signaling is necessary for the vascular tumor growth and maintenance in the mouse model. Rapamycin, a potent mTORC1 inhibitor, effectively leads to the regression of established tumors in the mutant mice.…”

mentioning

confidence: 95%

Modeling lymphangiosarcoma in mice

Sun

Guan

2016

Cell Cycle

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Malignant transformation of infantile hemangioma to angiosarcoma: Response to chemotherapy with bevacizumab

Cited by 58 publications

References 20 publications

Constitutive Activation of mTORC1 in Endothelial Cells Leads to the Development and Progression of Lymphangiosarcoma through VEGF Autocrine Signaling

Constitutive Activation of mTORC1 in Endothelial Cells Leads to the Development and Progression of Lymphangiosarcoma through VEGF Autocrine Signaling

Comprehensive treatment of unresectable cardiac angiosarcoma: A case report and review of literature

Modeling lymphangiosarcoma in mice

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