Objective:

To report a unique case of primary breast angiosarcoma with skull base and dural metastasis, highlighting its significance in the context of existing literature, and evaluate the efficacy of anlotinib and temozolomide as a salvage therapy.

Key Findings:

• First documented case of skull base metastasis and dural involvement from primary breast angiosarcoma, expanding the known spectrum of this malignancy.
• Anlotinib and temozolomide combination achieved rapid symptomatic and radiographic control, suggesting a novel therapeutic strategy.
• The treatment was well-tolerated with no adverse events reported.

Interpretation:

The combination therapy suggests a potential synergistic effect against both angiogenesis and central nervous system metastases in angiosarcoma, warranting further investigation into the underlying mechanisms.

Limitations:

• Single case study limits generalizability of findings; further studies are needed to validate the efficacy and safety of the treatment regimen.
• Long-term efficacy and safety of the treatment regimen remain to be established.

Conclusion:

The case highlights the importance of targeting angiogenesis and utilizing CNS-penetrating agents in treating aggressive malignancies like breast angiosarcoma with atypical metastasis.