• Kazuhiko Hashimoto, Shunji Nishimura, Yu Shinyashiki, Tomohiko Ito, Hiroki Tanaka, Kazuhiro Ohtani, Ryosuke Kakinoki, and Masao Akagi.
• Department of Orthopedic Surgery, Kindai University Hospital, Osaka-Sayama City, Osaka, Japan.
• Medicine (Baltimore). 2022 Jul 15; 101 (28): e29621e29621.

RationaleThe genomic alteration of cutaneous angiosarcoma (cAS) is complex. Treatment efficacy of immunotherapy for cAS remains controversial and prognosis remains poor. Herein, we report a case of cAS with programmed cell death 1, programmed cell death ligand-1, New York esophageal squamous cell carcinoma-1, and melanoma-associated antigen 4.Patient ConcernsA 69-year-old man presented with a chief complaint of left thumb pain, with a soft tissue mass in the palmar side of the thumb. He had no past medical history. Three months prior, the man experienced the pain while scuba diving. He visited a nearby clinic, and magnetic resonance imaging revealed a soft tissue tumor on the palmar side of the thumb. He was referred to our hospital and a marginal excisional biopsy was performed.DiagnosisPathological findings revealed an angiosarcoma with high-flow serpentine vessels.InterventionsAn excision was performed from the base of the thumb to achieve a wide margin.OutcomesOne year after the treatment, the patient has not experienced recurrence, metastasis, or complications.LessonsHistopathology of the excised specimen was positive for programmed cell death 1, programmed cell death ligand-1, New York esophageal squamous cell carcinoma-1, and melanoma-associated antigen 4; their expression may be a therapeutic target for cAS. Combining immunotherapy with surgical treatment may be effective for cAS.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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