Abstract
Angiosarcoma is an uncommon malignancy with a poor prognosis. Systemic therapy options for patients with metastatic disease generally have limited effectiveness. In this case study, a 73-year-old male with metastatic angiosarcoma who previously declined chemotherapy and developed progressive disease after checkpoint inhibitor immunotherapy elected to try thalidomide based on 6 case reports describing its effectiveness. Thalidomide resulted in stable disease for 9 months, but due to severe neuropathy as a side effect, lenalidomide was then substituted for thalidomide. The patient continued to have stable disease on lenalidomide for an additional 16 months and ongoing. This is the first case study to report on effective treatment of angiosarcoma with lenalidomide. Further investigation of lenalidomide in the management of angiosarcoma is warranted.