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Hospital de Sant Pau Universitat Autònoma de Barcelona 08025, Barcelona, Spain
A 42-year-old woman presented with a 2-month history of a rapidly enlarging mass in the right breast. Clinical examination revealed a large, hard fixed mass with skin ulceration. Helical CT revealed a large, heterogeneous, relatively well-circumscribed mass, with central hypodense nonenhancing areas that were suggestive of necrosis, interspersed with peripheral and central zones of intense contrast enhancement and punctate calcifications (Fig. 2A). Laboratory tests revealed a highly elevated serum alkaline phosphatase at 951 U/L (normal range, 82-198 U/L).
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Mammography performed 2 weeks after the CT examination showed a large 9-cm mass with lobulated borders and well-defined margins that contained several calcifications closely resembling bone. In addition, fine eggshell calcifications at the periphery of the tumor were also noticed (Fig. 2B). Compared with the findings on the initial CT scan, the number and size of calcifications had clearly increased significantly. High-resolution sonography showed a complex mass with both cystic and solid regions interspersed with focal areas of intense acoustic shadowing that were interpreted as coarse calcifications. Because a bone-forming neoplasm was suggested, 99mTc-diphosphonate bone imaging was performed, revealing an intense focal area of high activity in the region of the right breast. On T2-weighted MR images, the mass appeared well circumscribed and partially delineated by a fine low-signal peripheral band. This structure resembled a capsule and had a markedly heterogeneous internal signal with high-signal-intensity areas interspersed with low-signal zones, which were interpreted as necrohemorrhagic and bone-forming areas, respectively (Fig. 2C). On the two-dimensional dynamic images, the tumor showed nonhomogeneous rapid enhancement and washout phenomenon, suggesting malignancy.
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Pathologically, the tumor was composed of pleomorphic spindle cells, osteoblastlike cells, and osteoclastlike giant cells, among areas of necrosis and malignant osteoid. Epithelial elements were excluded after extensive sampling, including negative results for cytokeratin and positive results for vimentin (Fig. 2D).
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A modified radical mastectomy and axillary lymph node dissection were performed, and no lymph node metastases were found. At follow-up 16 months later, the patient showed no evidence of metastatic or recurrent disease.
Primary pure osteosarcoma of the breast is an uncommon malignancy that has been only briefly reported in the radiology literature [1, 2]. The neoplasm we described fulfills the requirements for the diagnosis of extraosseous osteosarcoma [3]. The presence of bone in breast lesions is not diagnostic of osteosarcoma because osteoid tissue has been reported in epithelial and mesenchymal neoplasms, both benign and malignant, such as fibroadenoma, phyllodes tumor, and metaplastic carcinomas. Thus, a diagnosis of mammary osteosarcoma can be established only after metaplastic carcinoma or biphasic tumor has been adequately excluded [3].
The previously reported
[1,2,3,4]
mammographic appearances of breast osteosarcomas vary, although most of the
tumors presented as large masses with relatively well-defined margins and
lobulated borders, often containing calcifications. Calcifications were
usually coarse, dense, and sometimes similar to fibroadenomas. Masses with
irregular margins have also been described
[1,
2]. The imaging features seen
with our patient, particularly the pattern of calcifications, are quite
distinctive. Large areas of necrosis have been found in only 10% of breast
osteosarcomas. This feature, which has been correlated with a bleak prognosis,
was easily observed on sonography
[1,2,3].
99mTc-diphosphonate is a specific radionuclide marker for osteoid
tumoral tissue [4]. Intense
focal uptake of this radionuclide in a soft-tissue tumor is strongly
suggestive of bone-forming neoplasms. In addition, alkaline phosphatase has
been found to be elevated in patients with osteoid-forming
neoplasms.
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Treatment is surgery. Axillary node involvement is rare, and axillary dissection, although commonly performed, is not recommended. Overall, the prognosis remains poor because of early local recurrence and hematogenous metastases [3].
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  J. A. Harvey Unusual Breast Cancers: Useful Clues to Expanding the Differential Diagnosis Radiology, March 1, 2007; 242(3): 683 - 694. [Abstract] [Full Text] [PDF]
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