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Year : 2021  |  Volume : 5  |  Issue : 1  |  Page : 33-35

Primary osteosarcoma of breast

Department of Radiation Oncology, BJMC and GCRI, Ahmedabad, Gujarat, India

Date of Submission03-Sep-2019
Date of Decision12-Sep-2020
Date of Acceptance25-Nov-2020
Date of Web Publication14-Apr-2021

Correspondence Address:
Maitrik Mehta
Department of Radiation Oncology, BJMC and GCRI, Ahmedabad, Gujarat
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/oji.oji_38_19

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Primary osteosarcoma of breast is an extremely rare entity with a poor prognosis. Although resection is the main treatment, the optimal treatment remains uncertain. It has early tumor recurrence with a propensity for hematogenous spread, most commonly to the lungs rather than lymphatic spread. Herein, we report a case of primary osteosarcoma of the left breast in a 50-year-old female. Her metastatic workup did not show any evidence of regional or distant metastasis. The patient was treated by simple mastectomy with axillary lymph node dissection followed by adjuvant chemotherapy. The patient is disease free at a follow-up of 11 months.

Keywords: Adjuvant chemotherapy, breast, mastectomy, osteosarcoma

How to cite this article:
Mankada S, Mehta M, Poddar J, Kichloo A, Pandya A, Suryanarayana U. Primary osteosarcoma of breast. Oncol J India 2021;5:33-5

How to cite this URL:
Mankada S, Mehta M, Poddar J, Kichloo A, Pandya A, Suryanarayana U. Primary osteosarcoma of breast. Oncol J India [serial online] 2021 [cited 2023 Jun 2];5:33-5. Available from: https://www.ojionline.org/text.asp?2021/5/1/33/313669

  Introduction Top

Nonepithelial malignancies of the breast account for <1% of breast tumors and malignant phyllodes tumor being the most common among these consisting of 61% of patients. Whereas osteosarcoma of the breast is an extremely rare entity with only a few cases have been published in the literatures, and it constitutes 12.5% of all the breast sarcomas.[1],[2] The mechanism of tumorigenesis of an extraskeletal osteosarcoma of the breast is unclear and may be due to metaplastic differentiation of a preexisting benign or malignant tumor or de novo from normal breast tissue.[3]

Rarity of this disease with a paucity of data makes its treatment difficult. Therefore, we report a case of primary osteosarcoma of breast to draw attention toward its management.

  Case Report Top

A 50-year-old female presented to our institute with a lump in the left breast for 3 years. There was no history of previous breast irradiation or trauma and no family history of any malignancies. On breast examination, a 10 cm × 10 cm mobile nontender mass was present in upper inner quadrant of the left breast with hard in consistency, and the mass was not associated with any nipple discharge or skin involvement. Another 3 cm × 2 cm mobile lump was found in upper inner quadrant of the right breast with firm in consistency. No axillary or supraclavicular lymph nodes were palpable on either side.

Routine investigations including complete blood count, renal function test, chest X-ray, and ultrasonogram of abdomen and pelvis were within normal limits. Mammosonogram of left breast revealed a well-defined soft tissue opacity in upper inner and central quadrant which was suggestive of malignant breast mass. Mammosonogram of the right breast showed an opacity of size 1.7 cm × 1.5 cm in upper inner quadrant. Tru-cut biopsy from lesion of the left breast showed malignant tumor with osteoid matrix likely to be osteosarcoma. Contrast-enhanced computed tomography scan of thorax depicted a soft-tissue lesion involving upper quadrants of left breast of size 7 cm × 7 cm reaching up to the skin [Figure 1]. A whole-body bone scan showed no evidence of any skeletal metastasis.
Figure 1: Contrast-enhanced computed tomography scan of thorax showing large localized lesion on the left side breast

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After discussion with multidisciplinary team, the patient underwent simple mastectomy of left breast with axillary node dissection and lumpectomy of the right breast. Gross specimen of the left breast showed a tumor with size of 6.8 cm × 6.7 cm involving upper inner quadrant. On microscopic examination of the left breast, tumor with adequate sections showed areas of high-grade malignant spindle cells with high mitotic rate, abundant osteoclastic multinucleate giant cells [Figure 2]a, areas of cystic hemorrhage necrosis, calcification, and malignant osteoid formation [Figure 2]b. Immunohistochemistry (IHC) was negative for cytokeratin [Figure 2]c but positive for vimentin [Figure 2]d. There was no expression of estrogen, progesterone receptors, and HER2/neu oncoprotein. No evidence of benign entrapped and malignant ductal epithelial component which ruled out the possibility of malignant phyllodes tumor with osteosarcoma or metaplastic carcinoma. This suggested a primary stromal osteosarcoma of breast. All the axillary nodes dissected were free of malignancies. Overlying skin, nipple, base of resection, and margins were free from tumor. Microscopic examination of the right breast revealed features of sclerotic fibroadenoma.
Figure 2: Histopathological examination on H and E stain showing (a) malignant spindle cells resembling mesenchymal differentiation along with multinucleated giant cells (×10) and (b) osteoid formation on histopathology suggesting a bone tumor (×20), and immunohistochemical examination showing (c) immunonegative for cytokeratin (×20), (d) immunopositive for vimentin (×20)

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The patient was received three cycles of adjuvant chemotherapy with ifosfamide (3000 mg/m2) on day 1–3 and doxorubicin (75 mg/m2) on day 1 for each cycle repeated at 3 weekly intervals with manageable toxicity. The patient is currently disease free clinically and radiologically at a follow-up of 11 months.

  Discussion Top

Extraskeletal osteosarcomas are located in the soft tissue without skeletal attachment. These are rare entity and accounts for <1% of all the sarcomas. It is most commonly occurring in soft tissues of the lower extremities. Osteosarcoma with breast primary is an extremely rare entity, with only a few cases have been published in the literature.[4] Extraskeletal osteosarcomas are rarely seen in patients before 40 years of age.[5],[6] The mechanism of tumorigenesis is unclear. It may be caused by trauma to the breast, previously taking radiotherapy for a malignant lesion or preceding fibroadenoma or phyllodes tumor. A progressively enlarged mass is the most common presentation of osteosarcoma of breast.[5] Our case had no such medical history for the possible etiology. However, such a long history of breast lump for 3 years before the diagnosis of osteosarcoma creates a confusion whether the patient had undiagnosed fibroadenoma in the same side of breast which leads to osteosarcoma in the latter part. Moreover, the patient had diagnosed fibroadenoma of opposite breast at the time of diagnosis of osteosarcoma of breast.

There are two close differential diagnoses for osteosarcoma such as metaplastic carcinoma and phyllodes tumor.[4],[7] The absence of specific morphological features such as biphasic tumor, with leaf-like architecture and epithelial component will rule out the possibility of cystosarcoma phyllodes. Furthermore, calcification on microscopy is an unusual finding in phyllodes tumor. The absence of a carcinomatous component on H and E sections and cytokeratin negativity of the neoplastic spindle cells on IHC will rule out the possibility of metaplastic carcinoma. Hence, IHC plays an important role toward the diagnosis of osteosarcoma.[7] Our case had no such specific morphological features of phyllodes tumor, and cytokeratin was immunonegative on neoplastic spindle cells. Hence, we ruled out the possibility of phyllodes tumor and metaplastic carcinoma in our case.

Lung is the most common site of distant metastasis consisting of 80% of cases, followed by bone and liver, respectively. Breast osteosarcoma is aggressive in nature and tend to recur, with 5-year overall survival probability of 38% and the 10-year estimated survival of 32%.[8]

Wide excision with negative margins is the mainstay of treatment for primary osteosarcoma of breast. Most authors suggest that mastectomy should be the optimal management as wide local excision complicated by local recurrence.[9] The patients undergoing wide local excision had local recurrence rate of 67%, whereas it was 11% for those performed with mastectomy.[8] The role of nodal evolution is still unclear. A study by Silver and Tavassoli on primary osteosarcoma of breast found that all axillary nodes dissected in twenty patients were negative for malignancies.[8] Our case had similar findings of no nodal dissemination of dissected axillary nodes.

The overall survival advantage of adjuvant chemotherapy or radiotherapy is not established yet. These adjuvant therapies are prescribed as skeletal osteosarcoma, but the response is poor with extraskeletal osteosarcoma than skeletal osteosarcoma.[4] Nonetheless, some authors do prescribe chemotherapy may be to control the micrometastasis.[10]

  Conclusion Top

Primary osteosarcoma of breast is rare. Upfront surgical excision particularly with mastectomy is the mainstay of treatment and offers best outcome. Chemotherapy may be effective in adjuvant setting, but role of radiation is not proven.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Yoon CS, Kang SS. Primary osteosarcoma of the breast: A case report. Ann Surg Treat Res 2017;93:57-60.  Back to cited text no. 1
Young JL Jr., Ward KC, Wingo PA, Howe HL. The incidence of malignant non-carcinomas of the female breast. Cancer Causes Control 2004;15:313-9.  Back to cited text no. 2
Alghofaily KA, Almushayqih MH, Alanazi MF, Salamah AAB, Benediktsson H. Primary osteosarcoma of the breast arising in an intraductal papilloma. Case Rep Radiol 2017;2017:5787829.  Back to cited text no. 3
Kerkar PB, Daga G. Primary osteosarcoma of the breast. Indian J Surg Oncol 2018;9:578-80.  Back to cited text no. 4
Kurata K, Anan K, Ishikawa N, Koga K, Saimura M, Nishihara K, et al. A case of primary extraskeletal osteosarcoma of the breast. Surg Case Rep 2018;4:121.  Back to cited text no. 5
Siraj F, Jain D, Chopra P. Extraskeletal osteosarcoma of abdominal wall in a child. Ann Diagn Pathol 2011;15:131-4.  Back to cited text no. 6
Adem C, Reynolds C, Ingle JN, Nascimento AG. Primary breast sarcoma: Clinicopathologic series from the Mayo Clinic and review of the literature. Br J Cancer 2004;91:237-41.  Back to cited text no. 7
Silver SA, Tavassoli FA. Primary osteogenic sarcoma of the breast: A clinicopathologic analysis of 50 cases. Am J Surg Pathol 1998;22:925-33.  Back to cited text no. 8
Uccello M, Malaguarnera M, Giordano M, Leggio E, Catania VE, Consoli AS, et al. A large calcified retroperitoneal extraskeletal osteosarcoma with consequent bilateral hydronephrosis. Eur Rev Med Pharmacol Sci 2012;16:977-82.  Back to cited text no. 9
Gull S, Patil P, Spence RA. Primary osteosarcoma of breast. BMJ Case. DOI: 10.1136/bcr. 03.2011.4015.  Back to cited text no. 10


  [Figure 1], [Figure 2]


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