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Hindawi Publishing Corporation Case Reports in Oncological Medicine Volume 2013, Article ID 858705, 3 pages http://dx.doi.org/10.1155/2013/858705 Case Report 1 2 3 3 4 Anna Rizzi, Alberto Soregaroli, Claudia Zambelli, Fausto Zorzi, Stefano Mutti, 4 1 1 Claudio Codignola, Paola Bertocchi, and Alberto Zaniboni Medical Oncology, Fondazione Poliambulanza, Via Bissolati 57, 25124 Brescia, Italy Radiology Department, Fondazione Poliambulanza, Via Bissolati 57, 25124 Brescia, Italy Pathology Department, Fondazione Poliambulanza, Via Bissolati 57, 25124 Brescia, Italy Surgical Department, Fondazione Poliambulanza, Via Bissolati 57, 25124 Brescia, Italy Correspondence should be addressed to Anna Rizzi; email@example.com Received 27 February 2013; Accepted 19 March 2013 Academic Editors: A. Goodman, J. I. Mayordomo, and J. M. Ribera Copyright © 2013 Anna Rizzi et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Introduction. Primary osteosarcoma of the breast is a rare soft-tissue form of osteosarcoma without involvement of the skeletal system. Due to the rarity of the disease, its clinical features and optimal treatment remain unclear. Case Presentation.This case report deals with a 62-year-old woman with pure osteosarcoma of the breast. Conclusions. eTh prognosis of primary osteosarcoma of the breast is poor. Recurrence is frequent, and it is oen ft associated with haematogenous spread of the disease to the lung. Treatment follows the model of sarcomas aec ff ting other locations and must be planned in a multidisciplinary fashion. Adjuvant chemotherapy should be considered for patients with tumors showing aggressive features. 1. Introduction Clinical examination showed an enlarged mass localized at the 1 o’clock position in the superior-outer quadrant of the Sarcomas of the breast are heterogeneous neoplasms derived left breast. from nonepithelial elements of the gland, and they represent Diagnostic mammography described a 3.7 cm× 3.6 cm less than 1% of breast cancers and less than 5% of all sarcomas mass with gross calcification in the centre and a cluster of . spotty calcification around ( Figure 1). No clinically enlarged Primary osteosarcoma of the breast is infrequently nodes were detected in axilla. reported. In fact, although osteosarcomas constitute a com- High resolution ultrasonography demonstrated a densely mon histology aer ft breast radiation therapy, they arise mostly shadowing mass, compatible with the extensive calcification from adjacent bone structures (sternum, ribs) and therefore identiefi donmammography. do not represent primary breast sarcomas . Doppler evaluation showed marked vascularity in the Because of the rarity of the disease, both clinical features surrounding tissue. and optimal treatment are still to be defined. Cytological (FNA) examination shows neoplastic cells In contrast to skeletal osteosarcoma affecting mainly (C5 according to the European classification scheme); the young patients, primary osteosarcoma of the breast occurs in cells were negative for epithelial markers (CAM5.2, CK older patients, with a mean age at presentation around 65 . AE1/AE3, CK 34𝛽 E12) on immunocytochemistry. Staging Although there are other case reports concerning primary CT scan and skeletal scintigraphy showed no evidence of osteosarcoma of the breast, we believe that each case can metastatic disease. contribute to improve the management of this rare disease. Laboratory findings were normal, including the preoper- 2. Case Presentation ative alkaline phosphatase. A 62-year-old Caucasian woman with a medical history sig- The patient underwent a left superior-outer quadrantec- nificant only for hypertension presented with a self-palpated tomy with a complete axillary dissection, as for patient’s pref- mass in her left breast. erence. Macroscopic examination of the surgical specimen 2 Case Reports in Oncological Medicine Figure 2: Tumor cells associated with band of osteoid with osteoblastic cells (hematoxylin-eosin 20x). Figure 1: Mammographic features of the lesion. revealed a well-circumscribed tumour (4,2× 3cm) brown in Figure 3: Malignant epithelioid cells and rare osteoblastic cells color and hard in consistency, with areas of calcification. (hematoxylin-eosin 20x). Microscopically, the tumour was composed of a diffuse proliferation of polygonal cells with round nuclei, evident nucleoli, and an elevated mitotic index (50 mitoses/10 HPF). Osteosarcomas of the breast may arise either from pre- Multinucleated giant cells resembling osteoclasts were inter- existing benign or malignant breast neoplasms or from pre- dispersed. Tumour cells produced osteoid and prominent viously normal breast tissue, and the histogenesis of primary woven bone and were supported by a diffuse network of osteosarcomas of the breast is unknown. Risk factors have reticulin b fi res (Figures 2 and 3). been identified for some extraskeletal osteosarcomas and Neither lymphatic nor venous invasion was observed. include prior local irradiation, trauma, and a foreign body. Eleven axillary lymph nodes were negative. On immuno- The present patient had no history of trauma or irradiation histochemistry neoplastic cells were negative for epithelial and did not have a biphasic tumor, in which case it has been markers (CAM 5.2, CK AE1/AE3, CK 34𝛽 E12, EMA) as well suggested that carcinogenesis could be due to transformation as for desmin,𝛼 -smooth-muscle actin, myogenin, and S100. of totipotent mesenchymal cells of the breast stroma. The tumour was negative for both estrogen and proges- A preoperative diagnosis is unusual and most patients teron receptors. have a correct diagnosis only aeft r histological examination A diagnosis of primitive osteoblastic osteosarcoma of the of the surgical specimen. Mammographic findings gener- breast was formulated. The patient then received four cycles ally consist of large masses with well-den fi ed margins and of adjuvant chemotherapy with Ifosfamide and Adriamycin lobulated borders, which often contain coarse or dense and currently shows no evidence of disease (NED) aer ft calcifications as in b fi roadenomas. A definitive diagnosis can thirteen months. only be made when an osteogenic sarcomas arising from the underlying bones is excluded, and immunohistochemical tests show positivity for vimentin with absence of epithelial, 3. Discussion neural, muscular, and other markers . Osteosarcoma of the breast is an extremely rare tumour which Due to the rarity of primary osteosarcoma of the breast, should be differentiated from other two similar entities, cys- there are no validated guidelines for treatment, and the tosarcoma phyllodes and metaplastic carcinoma. It is possible best therapeutic approach remains wide local excision or to recognize the former by specific morphological features mastectomy, depending on the size of the tumour and the andthe latter by thepresenceofcarcinomatous component remaining breast tissue. A complete resection with negative or cytokeratin immunopositivity on hematoxylin and eosin resection margins is needed, as margin involvement is a major sections . predictor for local disease recurrence. Case Reports in Oncological Medicine 3 Table1:Somecasereports reported in ourpaper. Radio Chemo ∗ † Author Age Size Surgery Histology LR DR Outcome Y/N Y/N Vorobiof et al. 69 3 cm Mastectomy N N Osteosarcoma Uk Uk Lost  Ogundiran et al. 40 18× 20 cm Mastectomy Refused N Osteosarcoma Uk Nodes 6 mo  Khan et al.  66 2 cm Mastectomy N N Osteosarcoma No No 8 y Singhal et al.  40 5 cm Mastectomy N N Osteosarcoma No No 5 y Local recurrence. Distant recurrence. Prognostic factors for primary osteosarcomas of the  G. Vorobiof, G. Hariparsad, W. Freinkel, H. Said, and D. A. Vorobiof, “Primary osteosarcoma of the breast: a case report,” breast include tumor size, number of mitoses, and presence Breast Journal,vol.9,no. 3, pp.231–233,2003. of stromal atypia . In general, osteosarcomas are aggressive  T.O.Ogundiran,S.A.Ademola,O.M.Oluwatosin,E.E.Akang, tumours with blood-borne spread more common than lym- and C. A. Adebamowo, “Primary osteogenic sarcoma of the phatic spread. For this reason lymphatic axillary dissection breast,” World Journal of Surgical Oncology,vol.4,pp. 90–93, is not considered as a mainstay of surgical treatment, and a diagnosis of metaplastic carcinoma should be considered in  S.Khan, E. A. Gritffi hs, N. Shah,and S. Ravi,“Primaryoste- the presence of lymph nodes metastases . ogenic sarcoma of the breast: a case report,” Cases Journal,vol. There is little evidence on the long-term prognosis of 1, pp. 148–151, 2008. the disease due to the small number of cases reported  V. Singhal, Chintamani, and J. M. Cosgrove, “Osteogenic sar- in the literature. In a study of 50 patients with primary coma of the breast arising in a cystosarcoma phyllodes: a case breast osteosarcoma, Silver and Tavassoli reported a 5-year report and review of the literature,” Journal of Medical Case survival of 38%, with 28% percent of patients developing local Reports, vol. 5, pp. 293–295, 2011. recurrence and 41% distant metastases . Hematogenous  C. Adem, C. Reynolds, J. N. Ingle, and A. G. Nascimento, metastases most commonly occurred in the lungs (80%), “Primary breast sarcoma: clinicopathologic series from the bone (20%), and liver (17%). Mayo Clinic and review of the literature,” British Journal of Indications for adjuvant chemotherapy and radiation Cancer,vol.91, no.2,pp. 237–241, 2004. therapy, in the absence of specicfi data on breast sarcoma,  S. A. Silver and F. A. Tavassoli, “Osteosarcomatous differenti- should follow those for soft-tissue sarcomas in general [ 9]. ation in phyllodes tumors,” American Journal of Surgical Pathol- The role of postoperative radiotherapy and chemotherapy in ogy,vol.23, no.7,pp. 815–821, 1999. curatively resected so-ft tissue sarcomas is still controversial.  B.J.Barrow, N. A. Janjan,H.Gutmanetal.,“Role of radiother- Whether adjuvant radiotherapy should be used remains apy in sarcoma of the breast—a retrospective review of the M.D. unclear, although several studies reporting on a small number Anderson experience,” Radiotherapy and Oncology,vol.52, no. of patients suggest that adjuvant chemotherapy may be of 2, pp. 173–178, 1999. value in patient management (Table 1). Conflict of Interests eTh authors declare that they have no competing interests. Consent Written informed consent was obtained from the patient for the publication of this case report and the accompanying images. References  I. A. Voutsadakis, K. Zaman, and S. Leyvraz, “Breast sarcomas: current and future perspectives,” Breast,vol.20, no.3,pp. 199– 204, 2011.  Y.M.Kirova, J. R. Vilcoq,B.Asselain, X. Sastre-Garau,and A. Fourquet, “Radiation-induced sarcomas aeft r radiotherapy for breast carcinoma: a large-scale single-institution review,” Can- cer,vol.104,no. 4, pp.856–863,2005. 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Published: Apr 7, 2013
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