Background: Extraosseous osteosarcoma is a rare disease with approximately 150 cases reported in the literature. Primary osteogenic sarcoma of the breast is considered a particularly rare localisation. Approximately 20 cases of osteogenic sarcoma of the breast have been reported in the literature. The radiographic findings of the primary lesion are often misinterpreted as benign fibroadenomas the diagnosis of this rare disease is based on histological criteria. Osteogenic sarcomas of the breast have a high tendency towards local recurrences early haematogenous pread particularly pulmonary metastases. Case Report: Osteogenic sarcoma of the breast was diagnosed in a 48-year-old woman. Following mastectomy axillary lymph node dissection the patient developed liver thoracic wall metastases. She obtained a complete remission to COSS-86 chemotherapy but subsequently relapsed died from disseminated tumour despite repeated surgical attempts postoperative radiotherapy palliative chemotherapy 24 months after the primary diagnosis. Conclusions: Based on the currently available data of the cases reported radical resection of the primary tumour including axillary lymph node dissection is probably the appropriate surgical approach for primary osteogenic sarcoma of the breast. Since chemosensitivity of this malignancy has been observed adjuvant treatment according to the regimens employed for the treatment of osteogenic sarcoma of the bone (COSS treatment protocol) is recommended in order to improve the otherwise poor prognosis of patients with osteogenic sarcoma of the breast.

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