• Jin A Yoon, Myung Jun Shin, Yong Beom Shin, Byeong Ju Lee, Kyung Un Choi, and Joo Hyoung Kim.
• Department of Rehabilitation Medicine, Pusan National University School of Medicine and Biomedical Research Institute, Pusan National University Hospital, Busan, Republic of Korea.
• Medicine (Baltimore). 2021 Dec 3; 100 (48): e27985.

IntroductionAngiosarcoma secondary to post-irradiation and lymphedema is rare, but it is aggressive with a poor prognosis. It is essential to understand these patients' clinical features and distinguish them from benign diseases or other malignant tumors.Patient ConcernsThree patients who had radiotherapy for cancer treatment and chronic lymphedema admitted to the hospital with specific skin lesions at upper or lower extremities.DiagnosisExcisional biopsies revealed prominent, highly atypical cells with a vasoformative area, composed of atypical, large epithelioid cells with vesicular nuclei, prominent nucleoli, and mitoses. Immunohistochemistry revealed diffuse expression of endothelial cell markers suggestive of angiosarcoma.InterventionsOne patient had shoulder disarticulation with wide excision with adjuvant radiotherapy and chemotherapy and other 2 discontinued the treatment.OutcomesAfter the treatment, one patient was transferred to rehabilitation department for shoulder disarticulation prosthesis fitting without recurrence sign for 1 year. Two patient refused further treatment and was lost to follow-up.ConclusionIn cases of patients with irratiation and chronic lymphedema, clinical findings suggestive of angiosarcoma, biopsy and imaging studies should be performed as soon as possible.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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