
==== Front
Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01034-4
10.1016/j.ijscr.2024.110253
110253
Case Report
Radiation-induced angiosarcoma of the breast: A case report
Gurleyik Emin egurleyik@yahoo.com
⁎
Yekenkurul Erman
Gursoy Fatih
Gonullu Emin
Department of Surgery, Duzce University, Faculty of Medicine, Duzce, Turkey
⁎ Corresponding author. egurleyik@yahoo.com
10 9 2024
10 2024
10 9 2024
123 11025311 7 2024
30 8 2024
5 9 2024
© 2024 The Author(s)
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction and importance

Today, breast-conserving surgery (BCS) and adjuvant radiotherapy are preferred treatments for patients with early invasive breast cancer. Radiation-induced angiosarcoma (RIAS) of the breast is a rare but serious complication of radiotherapy.

Case presentation

Seventy-one-year-old woman is presented to our department with a locally advanced dark red polypoid lesion on her left breast. She had left BCS, axillary dissection, and adjuvant radiotherapy for invasive breast cancer 8 years before presentation. A small tissue sample from the breast lesions was sent for histopathologic examination that the diagnosis was angiosarcoma of the breast. She had neoadjuvant chemotherapy. Following the completion of chemotherapy, a total mastectomy was performed as surgical treatment. The final histopathologic diagnosis was well-differentiated angiosarcoma.

Clinical discussion

RIAS of the breast is rare disease that develops after a several-year latency period. Locally advanced disease was initially treated with neoadjuvant chemotherapy which appears to be effective for significant disease regression. Patients who respond well to chemotherapy in vivo may have higher disease-specific survival rates. After chemotherapy-induced regression of locally advanced sarcoma, total mastectomy was performed for radical treatment.

Conclusion

RIAS of the breast is defined as the histological diagnosis of angiosarcoma in an irradiated region after a long latency period in a patient who has previously received radiotherapy for breast carcinoma. Based on clinical and nuclear imaging data, we may conclude that neoadjuvant chemotherapy can result in significant disease regression, and following neoadjuvant chemotherapy the treatment of angiosarcoma is completed by radical breast surgery.

Highlights

• Radiation-induced breast angiosarcoma (BA) is a rare complication of irradiation.

• BA is a serious disease occurring in irradiated field after a latency period of many years.

• Neoadjuvant chemotherapy could be beneficial for significant regression of BA

• Breast surgery providing locoregional control, completes radical treatment of BA.

• Breast cancer cases who received radiotherapy should be carefully followed for angiosarcoma.

Keywords

Radiation
Breast cancer
Sarcoma
Vascular tumour
Case report
==== Body
pmc1 Introduction

Breast angiosarcoma (BA) is classified as primary or secondary, with primary incidence being infrequent when compared with secondary disease. Radiation-induced angiosarcoma (RIAS) is a rare complication of breast irradiation for locoregional treatment of invasive breast carcinoma after several years of adjuvant radiotherapy. BA is a rare malignancy that accounts for <1 % of all soft tissue cancers. Both primary and secondary BA have multifocal presentation and are uncommon, highly aggressive malignancies with poor prognosis [[1], [2], [3], [4]]. Breast-conserving surgery (BCS) and adjuvant radiotherapy are currently standard treatments for early-stage breast cancer. BCS for the treatment of invasive breast cancer has progressively increased during the previous three decades. Following breast conservation, breast irradiation for locoregional control, which is a crucial component of treatment, is becoming increasingly common. Therefore, RIAS of the breast presents as a rare but serious complication of breast irradiation. The cumulative incidence of angiosarcoma following breast radiotherapy increases continuously, reaching 1.4 ‰ after 20 years [5].

In this paper, we present a case of RIAS of the breast caused by adjuvant radiotherapy following BCS for the treatment of invasive breast carcinoma.

2 Case presentation

This case has been reported in line with the SCARE criteria [6]. Full informed consent of the patient, and Institutional Review Board approval was obtained for this case report. Seventy-one-year-old woman with a large dark red polypoid lesion on her left breast skin was referred to our department. She noticed the first lesion ten to twelve months before presentation. The lesion has gradually expended until she was referred to our department. Her medical history revealed that she had left BCS, axillary dissection, and adjuvant radiotherapy for invasive breast cancer 8 years before presentation. She underwent whole breast external beam radiotherapy 50Gy in 25 fractions (5 days a week over 5 weeks) and an additional 5 days of tumour bed boost was given. Physical examination revealed a large, painless polypoid lesion on the left breast (Fig. 1).Fig. 1 Locally advanced lesions on the left breast skin.

Fig. 1

2.1 Pathologic analysis

A small tissue sample from the breast lesions was sent for histopathologic examination. The diagnosis was a malignant vascular tumour (angiosarcoma) of the left breast.

F-18 fluorodeoxyglucose positron emission tomography/computed tomography (F-18 FDG PET/CT) was used for disease staging. PET/CT demonstrated increased FDG absorption (SUVmax 7.8) in three skin regions of the left breast, the largest lesion measuring 45 mm, and non-FDG accumulation on other distant sides of the body.

The patient was examined by a multidisciplinary tumour board. The decision was to proceed with neoadjuvant chemotherapy (NAC) and mastectomy. She underwent 6 of neoadjuvant gemcitabine/docetaxel. Macroscopic improvement was observed, and skin lesions regressed significantly after chemotherapy (Fig. 2). After NAC, a control PET/CT (F-18 FDG PET/CT) scan indicated FDG uptake (SUVmax 4.5) in the inner skin region of the left breast. According to previous nuclear imaging, local control of angiosarcoma was improved, and thickness, local invasion extent, and metabolic activity of malignant skin lesions decreased.Fig. 2 Left breast after neoadjuvant chemotherapy.

Fig. 2

2.2 Surgery

The patient had a left mastectomy. The breast was excised with macroscopically free margins. The wound was primarily closed at the end of surgery (Fig. 3).Fig. 3 Primary wound closure after mastectomy.

Fig. 3

2.3 Pathologic analysis

The final histopathologic diagnosis was well-differentiated angiosarcoma with two foci. Lesions measure 15 × 15 × 5 and 10 × 10 × 5 mm. The mitosis rate is <1/mm2, with Ki-67 levels ranging from 5 % to 10 %. The surgical margins are free of malignant cells. The pathologic response to chemotherapy is >95 %.

At the sixth month postoperatively, a control PET/CT (F-18 FDG PET/CT) was performed, and no metastatic focus was found in the entire body scan. PET/CT revealed no evidence of hypermetabolic activity or pathologic FDG accumulation in the body.

3 Discussion

Breast angiosarcoma is a rare disease, <1 % of soft tissue sarcoma and 0.04 %–1 % of breast malignancies [1,[7], [8], [9]]. BCS followed by radiotherapy has been and continues to be the standard treatment for the majority of early breast cancers. Chau et al. [10] reported that the incidence of RIAS of the breast increased threefold during the previous 30 years, from roughly 10/100,000 to 30/100,000 person-years. Our present patient appears to be a typical case of breast radiotherapy complication after BCS for breast cancer. Diagnostic criteria for RIAS of the breast include a history of breast irradiation, a several-year latency period, the presence of sarcoma in the irradiated field, and histological evidence of a vascular tumour [7].

Our patient was a woman who was diagnosed with BA 8 years after breast irradiation. Patient characteristics and the latency period between radiotherapy and diagnosis of angiosarcoma are important markers of secondary disease. RIAS of the breast is a disease that affects postmenopausal, relatively elderly women, often over the age of 60 years. According to previous researches, the mean age of patients with RIAS of the breast ranges from 60 to 77 years [2,3,5,[7], [8], [9],[11], [12], [13], [14]]. Recent researches indicated a mean latency period of 6–8 years from radiotherapy to angiosarcoma, ranging from 2,5 to 39 years [5,7,8,[11], [12], [13], [14]].

Due to a locally advanced vascular tumour, the multidisciplinary board determined that NAC followed by surgery was the proper management for our patient. Recent researches typically provide two alternative treatment methods: NAC followed by surgery or upfront surgery with adjuvant chemotherapy [[1], [2], [3],5,8,[10], [11], [12],14]. Our patient received NAC before surgery, which allows for in vivo evaluation of tumour response to chemotherapy. According to postoperative pathological analysis, the tumour was significantly (>90 %) reduced with NAC. The pathological response to NAC may alter the prognosis of BA. Chang et al. [2] reported that their NAC-induced histologic response to BA was unsatisfactory in half of their patients, whereas the remaining half responded well (>90 %). Good responders showed significantly higher disease-specific survival [2,7,14]. Based on the significant tumour regression in our patient following NAC, we may conclude that NAC may be effective for disease control in some patients. Oleander et al. [5] using the Swedish cancer registry, reported a 5-year overall survival (OS) of 35 % for RIAS of the breast. In the Japanese dataset, the median disease-free survival and OS were 14 (1–75) and 23 (4–84) months, respectively [14]. In the Surveillance, Epidemiology, and End Results (SEER) database (2004–2015), the 1-, 3-, and 5-year OS rates were 80 %, 42 %, and 34 %, respectively [15]. In the SEER database (1975–2016), the 5-year OS was 44.9 % [16].

4 Conclusion

Based on the present patient characteristics, RIAS of the breast could be characterized as a histological diagnosis of angiosarcoma in an irradiated area after a long latency period in a patient who has previously received radiotherapy for breast carcinoma. Based on clinical, pathological, and nuclear imaging data, we may conclude that NAC followed by surgery may be effective for significant disease regression and for radical treatment in a patient with locally advanced BA. Breast cancer patients treated with BCS and adjuvant radiotherapy should be thoroughly monitored for secondary sarcoma of vascular origin.

Ethical approval

Special ethics clearance was not necessary for routine chemotherapy and surgery for invasive breast sarcoma case. This is a case report of a rare breast disease secondary to breast radiotherapy for invasive breast cancer. Publication of this case report was approved by Institutional Review Board of Medical Faculty of Duzce University (IRB no. 2024-05-05).

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Author contribution

Gurleyik E. The conception and design of the study. Analysis and interpretation of data. Drafting the article. Final approval of the version.

Yekenkurul E. Analysis and interpretation of data. Revising the article critically for important intellectual content. Final approval of the version.

Gursoy F. Acquisition of data. Revising the article critically for important intellectual content. Final approval of the version.

Gonullu E Acquisition of data. Revising the article critically for important intellectual content. Final approval of the version.

Research registration number

N/A.

Conflict of interest statement

The Authors declare no conflicts of interest.
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