Case Report: Complete Remission With Anti−PD−1 and Anti−VEGF Combined Therapy of a Patient With Metastatic Primary Splenic Angiosarcoma

Xu, W.; Wang, Kai; Gu, Wenguang; Nie, Xinxin; Zhang, Hao; Tang, Chuanhao; Lin, Liang-In; Liang, Jun

doi:10.3389/fonc.2022.809068

Cited by 9 publications

(8 citation statements)

References 32 publications

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“…More trials are being conducted to investigate the efficacy of anlotinib monotherapy, anlotinib plus immunotherapy, and anlotinib plus chemotherapy in advanced sarcomas, particularly soft tissue sarcomas ( 31 ). So far, there have been few reports on the efficacy of anlotinib treatment in ASs, with only a few case reports referring to various lesions in head and neck AS ( 32 ) and cardiac AS ( 33 ) or combined immunotherapy in metastatic primary splenic AS ( 34 ). In the case we present, the patient experienced local recurrence in the ear as well as scalp metastases following surgery.…”

Section: Discussionmentioning

confidence: 99%

Case report: Unique FLT4 variants associated with differential response to anlotinib in angiosarcoma

Meng

et al. 2022

Front. Oncol.

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Angiosarcoma (AS) is a rare, clinically aggressive tumor with limited treatment options and a poor prognosis. Mutations involving the angiogenesis-related genesTP53, PTPRB, PLCG1, KDR as well as FLT4 amplification have been observed in AS. There is a potential therapeutic value of inhibition of the VEGF pathway against angiosarcoma. Our case first described a patient with two sites of cutaneous angiosarcomas (cASs) that responded differently to anlotinib. And genetic analysis revealed that those two sites had different FLT4 variants, suggesting that FLT4 amplification could be the cause of anlotinib non-response.

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Section: Discussionmentioning

confidence: 99%

Case report: Unique FLT4 variants associated with differential response to anlotinib in angiosarcoma

Meng

et al. 2022

Front. Oncol.

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“…[ 7 , 8 ] Immunotherapies targeting PD-L1 or the programmed cell death protein 1 PD-1 receptor have also demonstrated efficacy in treating primary splenic angiosarcomas, as recent case reports have shown significant clinical benefit with pembrolizumab monotherapy as well as with the anti-PD-1 toripalimab combined with the multikinase inhibitor anlotinib. [ 9 , 10 ] In addition to these novel therapies, other tyrosine kinase inhibitors that also inhibit the vascular endothelial growth factor (VEGF)/VEGF receptor (VEGFR) signaling pathway, such as sunitinib or sorafenib, have demonstrated clinical benefit in treating recurrent metastatic angiosarcomas. [ 11 , 12 ]…”

Section: Discussionmentioning

confidence: 99%

Images in Immunotherapy and Precision Oncology: Angiosarcoma of the Spleen and Liver

Deshpande

Muñoz

Kelemen

et al. 2022

Journal of Immunotherapy and Precision Oncology

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Primary splenic or hepatic angiosarcomas are ultra-rare and aggressive malignancies associated with poor prognosis. The mainstay treatments are surgical resection and chemotherapy. We report a case of angiosarcoma in a 50-year-old woman who presented with bruising, fatigue, ecchymosis, and hepatosplenomegaly. She was treated with the multi-kinase inhibitor sunitinib for 4 weeks before developing a splenic hemorrhage and succumbing. Recent studies have demonstrated the clinical benefit of immunotherapies in angiosarcomas. Additionally, sequencing techniques have showcased the diverse molecular aberrations involved in angiosarcomas, which offer opportunities for precision-matched targeted therapies such as inhibitors of the VEGF/VEGFR axis and PI3K/Akt/mTor pathway.

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“…Angiosarcoma (AS) is rare malignant endothelial-cell tumors of vascular or lymphatic origin, and is among the most aggressive subtypes of soft-tissue sarcomas ( 112 , 113 ). In recent years, immunotherapy targeting PD-1/PD-L1 has become a hotspot in the treatment of AS ( 114 , 115 ). A recent study analyzed PD-L1 expression levels in angiosarcomas at different sites in humans and showed that PD-L1 was abnormally expressed in about 66% of the samples ( 116 ).…”

Section: Pd-1/pd-l1 and Inhibitors In Human Solid Cancersmentioning

confidence: 99%

The role of PD-1/PD-L1 and application of immune-checkpoint inhibitors in human cancers

et al. 2022

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Programmed cell death protein-1 (PD-1) is a checkpoint receptor expressed on the surface of various immune cells. PD-L1, the natural receptor for PD-1, is mainly expressed in tumor cells. Studies have indicated that PD-1 and PD-L1 are closely associated with the progression of human cancers and are promising biomarkers for cancer therapy. Moreover, the interaction of PD-1 and PD-L1 is one of the important mechanism by which human tumors generate immune escape. This article provides a review on the role of PD-L1/PD-1, mechanisms of immune response and resistance, as well as immune-related adverse events in the treatment of anti-PD-1/PD-L1 immunotherapy in human cancers. Moreover, we summarized a large number of clinical trials to successfully reveal that PD-1/PD-L1 Immune-checkpoint inhibitors have manifested promising therapeutic effects, which have been evaluated from different perspectives, including overall survival, objective effective rate and medium progression-free survival. Finally, we pointed out the current problems faced by PD-1/PD-L1 Immune-checkpoint inhibitors and its future prospects. Although PD-1/PD-L1 immune checkpoint inhibitors have been widely used in the treatment of human cancers, tough challenges still remain. Combination therapy and predictive models based on integrated biomarker determination theory may be the future directions for the application of PD-1/PD-L1 Immune-checkpoint inhibitors in treating human cancers.

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Case Report: Complete Remission With Anti−PD−1 and Anti−VEGF Combined Therapy of a Patient With Metastatic Primary Splenic Angiosarcoma

Cited by 9 publications

References 32 publications

Case report: Unique FLT4 variants associated with differential response to anlotinib in angiosarcoma

Case report: Unique FLT4 variants associated with differential response to anlotinib in angiosarcoma

Images in Immunotherapy and Precision Oncology: Angiosarcoma of the Spleen and Liver

The role of PD-1/PD-L1 and application of immune-checkpoint inhibitors in human cancers

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