Introduction: Radiotherapy-associated angiosarcoma (RAAS) is a rare but highly aggressive secondary malignancy that may arise several years after breast irradiation. The estimated prevalence of RAAS is 0.05%. One of the challenges in treating angiosarcoma is the high rate of recurrence, which was reported at 73%. We present the case of a 52-year-old woman presenting with breast RAAS five years after the primary breast cancer. She then underwent mastectomy and adjuvant chemotherapy, but the RAAS recurred three years later and was then treated with systemic therapy. Case Presentation: A 52-year-old woman had a history of triple-negative right breast cancer stage I and underwent neoadjuvant chemotherapy, breast-conserving surgery, and radiotherapy. Five years later, she developed a lump in her right breast, which was confirmed as high-grade angiosarcoma, sized 15 × 3 mm. We performed a simple mastectomy with R0 ≥ 5 mm followed by chemotherapy. Two years after the mastectomy, upon the request of the patient, she underwent delayed reconstruction with a transverse rectus abdominis myocutaneous (TRAM) flap. However, three years after the initial RAAS treatment, the patient presented with a mass and a bluish-purple discoloration in the upper and lower surgical scar on her right reconstructed breast, respectively, which was confirmed to be recurrent high-grade angiosarcoma. The patient is currently undergoing chemotherapy. Conclusions: RAAS is an aggressive tumor that may recur even after complete surgical resection. Chemotherapy may provide temporary disease control; however, treatment response should be closely monitored to determine the effectiveness of therapy.
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