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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 4  |  Issue : 1  |  Page : 112-115

Primary osteosarcoma of the breast presenting as a large breast abscess


1 Registrar in Surgery, Teaching Hospital Kandy, Sri Lanka
2 Senior Registrar in Surgery, Teaching Hospital Kandy, Sri Lanka
3 Consultant Pathologist, Teaching Hospital Kandy, Sri Lanka
4 Consultant General Surgeon, Teaching Hospital Kandy, Sri Lanka

Date of Web Publication2-Jun-2016

Correspondence Address:
Nadeesha J Nawarathna
Registrar in Surgery, Teaching Hospital Kandy
Sri Lanka
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-4848.183358

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  Abstract 

Primary extra osseous osteogenic sarcoma is one of the rarest forms of malignant tumor of the breast. It can arise as a result of osseous metaplasia of a preexisting neoplasm or from a none-phylloides sarcoma of a previously normal breast. Due to its rarity, the natural history and optimal treatment methods remain unclear. Sixty-year-old patient presented to the surgical casualty with a large breast abscess. Abscess wall histology revealed an osteosarcoma of the breast. Left total mastectomy with axillary clearance was performed. Histology and subsequent imunohistochemical studies confirmed the diagnosis of osteogenic sarcoma without lymph nodal metastasis. Patient was referred to the oncologist for further management. Rare types of breast tumors can be presented as breast abscess. Incision and drainage together with wall biopsy aid to exclude associated sinister pathologies. Diagnosis of primary osteosarcoma of the breast was made using histological and immunohistochemical findings once the possible primary from the bones of sternum and ribs was excluded. Treatment is as for sarcomas affecting other locations and should comprise a multidisciplinary approach.

Keywords: Breast, breast abscess, primary osteosarcoma


How to cite this article:
Nawarathna NJ, Kmarasinghe NR, Ratnayake P, Senevirathne RJ. Primary osteosarcoma of the breast presenting as a large breast abscess. Arch Med Health Sci 2016;4:112-5

How to cite this URL:
Nawarathna NJ, Kmarasinghe NR, Ratnayake P, Senevirathne RJ. Primary osteosarcoma of the breast presenting as a large breast abscess. Arch Med Health Sci [serial online] 2016 [cited 2019 Aug 25];4:112-5. Available from: http://www.amhsjournal.org/text.asp?2016/4/1/112/183358


  Introduction Top


Breast cancer is the most common cancer in females. Primary osteosarcomas of the breast are extremely rare and represent about 12.5% of the mammary sarcomas and <1% of all primary breast malignancies.[1],[2] Histological differentiation, from conventional osteosarcoma of bone or other extra skeletal forms of osteosarcoma, is difficult.[2] Extra skeletal forms of osteosarcoma have been reported in other organs like thyroid, kidney, bladder, colon, heart, testes, penis, gall bladder and the cerebellum.[2] Although primary osteosarcoma of bone is common in the young, primary osteosarcoma of the breast is seen in an older age group with a mean age of 65 years.[1],[3]


  Case Report Top


A 60-year-old unmarried lady presented to the surgical casualty ward, with a large abscess in the left breast. She had noticed a lump on her left breast about 3 months ago, which gradually increased in size. Over the last month, the lump increased in size rapidly showing signs of inflammation. Clinical examination of the breast revealed a grossly distended breast with features suggestive of a breast abscess. The consistency of the lump was irregular with many hard areas with cystic areas interspersed, suggestive of pus collection. Some enlarged lymph nodes were noted in the axilla. Imaging revealed a breast abscess with variable echogenic areas. Incision and drainage of the abscess done and pus sent for culture and ABST. Histological assessment of abscess wall and breast tissue revealed a primary osteogenic sarcoma of the breast. Total mastectomy and level II axillary lymph node clearance was performed.

Macroscopic examination of the mastectomy specimen showed 100 mm × 80 mm × 90 mm size growth spreading across both upper quadrants as well as the lower lateral quadrant.

Microscopy revealed a tumor composed of markedly pleomorphic spindle cells admixed with multinucleate osteoclast-like giant cells with areas showing tumor osteoid formation. Scattered, round neoplastic cells were also observed. A high mitotic activity was noted (6/10 HPF) with numerous cells with apoptotic nuclei. There was minimal tumor necrosis. The overall appearances revealed osteosarcoma of the breast [Figure 1]a and [Figure 1]b. As the tumor showed prominent osteoblastic differentiation with malignant osteoid formation, it was classified as osteoblastic osteosarcoma. Immunohistochemical assessment confirmed the diagnosis as high-grade osteosarcoma of the breast. All level I and level II lymph nodes revealed reactive changes only. Multiple sections of the breast lump failed to reveal any other epithelial or sarcomatous component, suggestive of carcinosarcoma. No primary tumor elsewhere was present.
Figure 1:

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Figure 2: Macroscopic appearance following surgery: During recovery

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Mammography of the nonaffected breast was normal. Other investigations performed such as ultrasound scan of the abdomen, staging contrast-enhanced computed tomography abdomen and chest, and skeletal scintigraphy showed no evidence of metastatic disease. Following surgery patient was referred to oncologist for adjuvant chemotherapy [Figure 2].{Figure 4}


  Discussion Top


Primary osteosarcoma of the breast is a rare malignant tumor and accounts for <1% of all primary breast malignancies.[1],[4],[5] It commonly affects elderly women but has also been reported in men.[6] It is a highly aggressive tumor, accounting for about 12.5% of mammary sarcoma.[1]

Histogenesis of this rare tumor is not clear. Origin from totipotent mesenchymal cells of the breast stroma or as a transformation from preexisting fibroadenoma or phyllodes tumor has been suggested.[3],[4],[5] Absence of metaplastic transformation of a preexisting fibroadenoma or phylloiod tumor suggests that this tumor arose from previously normal breast tissue.

Immunohistochemistry plays an important role to differentiate mesenchymal neoplasia from undifferentiated carcinoma. After the exclusion of epithelial neoplasia, it is necessary to define the histogenesis of the lesion.[7] Tumor cells were strongly positive for vimentin, a mesenchymal marker. Pan cytokeratin (CK) (epithelial cell marker), CD34 and bcl-2 were negative in tumor cells [Figure 1]c.{Figure 3}

Primary osteosarcoma of the breast should be differentiated from two other similar entities, namely, malignant phylloides tumor and metaplastic carcinoma. This can be done by specific morphological features with the presence of carcinomatous component and its CK immunopositivity respectively.[8]

Malignant phylloides tumor consists of a predominant mesenchymal component and a benign epithelial component.[7] Macroscopically malignant lesions show necrotic and hemorrhagic areas with infiltrative growth pattern. Stromal features simulate a sarcoma. Although markedly pleomorphic spindle cells admixed with multinucleated giant cells are seen in this patient, the lack of epithelial component in multiple random sections examined ruled out the diagnosis of malignant phyllodes tumor.

Metaplastic carcinoma is a term used to define breast tumor that shows features different to that of epithelial or typical ductal carcinoma. This rare form of heterogenous neoplasm is characterized by mixture of adenocarcinoma with areas of spindle cells, squamous cells and other cells of mesenchymal differentiation.[9] But none of these features were present in this specimen. This form of the tumor shows more aggressive behavior than typical ductal carcinoma.

Differential diagnoses include secondary lesion from a primary osteosarcoma of bone or direct extension of an osteogenic sarcoma arising from nearby ribs or sternum. This patient revealed no radiological evidence of primary osteosarcoma of bone. No underlying bone pathology was noted during surgery of the region. The tumor was not infiltrating through the chest wall musculature, and there was no evidence of microscopic infiltration of chest wall musculature by the tumor histologically.

It is very difficult to diagnose osteosarcoma of the breast from the clinical features, mammogram and ultrasound scan alone. For example, this patient presented with large breast abscess. Mammographically osteosarcoma of the breast appears as, well circumscribed dense lesions which have regular or irregular borders, with focal or extensive coarse calcification in one-third of cases. Its appearance may imitate a benign fibroadenoma.[10]

The basic requirement for the diagnoses of a primary osteosarcoma of the breast, according to Allan and Soule include:

  1. The presence of neoplastic osteoid or bone.
  2. The exclusion of origin in the bone and,
  3. The absence of epithelial component.[11]


In this patient, negative results of an isotope skeletal bone scan aided in excluding the possibility of a primary tumor while histological and immunohistochemical analysis showed no evidence of epithelial differentiation.

Osteosarcoma is classified into many subtypes; the commonest are fibroblastic, osteoblastic, and osteoclastic osteogenic sarcomas. Fibroblastic osteogenic osteosarcoma being associated with better survival outcome makes histological differentiation important.[12],[13]

Early recurrence and propensity for hematogenous rather than lymphatic spread have been noted in this highly aggressive rare tumor. Common sites for hematogenous metastasis are the lung (80%), bone (20%) and liver (7%).[4]

As the optimal management of localized disease includes total excision of the neoplasm with adequate resection margins, mastectomy was performed for this patient in order to control local recurrence. Although axillary lymph nodes clearance is not indicated for negative nodes, as we did not have facilities to assess fresh frozen section, level II axillary clearance was performed. In the presence of a partial epithelial differentiation, the basic requirement for the diagnosis of a primary sarcoma of the breast is the exclusion of epithelial origin, which requires axillary lymph node dissection. In that purpose, axillary clearance was done, although biopsy report revealed it as an osteosarcoma of the breast.

In the presence of lymph node, metastasis diagnosis of metaplastic carcinoma should be considered. In this patient, all lymph nodes were free of metastasis. However, axillary dissection has been generally considered unnecessary for diagnosed osteosarcoma of the breast since these tumors rarely spread through the lymphatic system.[14],[15]

Limited data on osteosarcoma indicate an aggressive clinical course and high incidence of recurrence and metastasis. Its management is significantly different from management of breast adenocarcinoma while it was the most important predictor of long-term survival in patients with different histological types of breast sarcoma.[14] Surgical management is the mainstay of treatment for breast osteosarcoma.

As with all rare tumors, osteosarcoma should be manage in reference centers to determine whether treatment of choice is surgical excision or total mastectomy and whether some form of adjuvant therapy can have beneficial effects. Indications for adjuvant therapy should follow those for another soft tissue sarcoma.[16] The role of adjuvant chemotherapy and/or radiotherapy also has been unclear although this patient is receiving chemotherapy at the moment.[14],[15]

Following curative resection of the primary tumor the role of postoperative radiotherapy and chemotherapy is still controversial. Although several studies report adjuvant chemotherapy may be of value in patient management, use of adjuvant radiotherapy remains unclear. But chemotherapy is the main treatment modality for metastatic disease.[17],[18]

Prognostic factors for osteosarcoma include; tumor size, number of mitosis, presence of stromal atypia, histological subtype and resection margin involvement.[3] The limited number of cases reported so far in the literature leaves a lot of controversy regarding the long-term prognosis of this disease.

 
  References Top

1.
Bahrami A, Resetkova E, Ro JY, Ibañez JD, Ayala AG. Primary osteosarcoma of the breast: Report of 2 cases. Arch Pathol Lab Med 2007;131:792-5.  Back to cited text no. 1
    
2.
Patidar AK, Kumar HS, Walke RV, Beniwal S. Primary osteogenic sarcoma of the breast: A case report. Indian J Surg Oncol 2012;3:255-6.  Back to cited text no. 2
    
3.
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. 3
    
4.
Remadi S, Doussis-Anagnostopoulu I, Mac GW. Primary osteosarcoma of the breast. Pathol Res Pract 1995;191:471-4.  Back to cited text no. 4
    
5.
Going JJ, Lumsden AB, Anderson TJ. A classical osteogenic sarcoma of the breast: Histology, immunohistochemistry and ultrastructure. Histopathology 1986;10:631-41.  Back to cited text no. 5
    
6.
Fletcher CD, Unni KK, Mertens F. World Health Organization Classification of Tumors: Pathology and Genetics of Tumors of Soft Tissue and Bone. Lyon, France: IARC Press; 2002.  Back to cited text no. 6
    
7.
Al-Nafussi A. Spindle cell tumours of the breast: Practical approach to diagnosis. Histopathology 1999;35:1-13.  Back to cited text no. 7
    
8.
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. 8
    
9.
Guarino M, Tricomi P, Giordano F, Cristofori E. Sarcomatoid carcinomas: Pathological and histopathogenetic considerations. Pathology 1996;28:298-305.  Back to cited text no. 9
    
10.
Harvey JA. Unusual breast cancers: Useful clues to expanding the differential diagnosis. Radiology 2007;242:683-94.  Back to cited text no. 10
    
11.
Allan CJ, Soule EH. Osteogenic sarcoma of the somatic soft tissues. Clinicopathologic study of 26 cases and review of literature. Cancer 1971;27:1121-33.  Back to cited text no. 11
    
12.
Sando N, Oka K, Moriya T, Saito H, Nagakura S, Mori N, et al. Osteosarcoma arising in the breast. APMIS 2006;114:581-7.  Back to cited text no. 12
    
13.
Ogundiran TO, Ademola SA, Oluwatosin OM, Akang EE, Adebamowo CA. Primary osteogenic sarcoma of the breast. World J Surg Oncol 2006;4:90.  Back to cited text no. 13
    
14.
Shabahang M, Franceschi D, Sundaram M, Castillo MH, Moffat FL, Frank DS, et al. Surgical management of primary breast sarcoma. Am Surg 2002;68:673-7.  Back to cited text no. 14
    
15.
McGowan TS, Cummings BJ, O'Sullivan B, Catton CN, Miller N, Panzarella T. An analysis of 78 breast sarcoma patients without distant metastases at presentation. Int J Radiat Oncol Biol Phys 2000;46:383-90.  Back to cited text no. 15
    
16.
Barrow BJ, Janjan NA, Gutman H, Benjamin RS, Allen P, Romsdahl MM, et al. Role of radiotherapy in sarcoma of the breast — a retrospective review of the M.D. Anderson experience. Radiother Oncol 1999;52:173-8.  Back to cited text no. 16
    
17.
Ellmann A, Jawa ZM, Maharaj M. Primary osteogenic sarcoma of the breast detected on skeletal scintigraphy. Clin Nucl Med 2006;31:474-5.  Back to cited text no. 17
    
18.
Irshad K, Mann BS, Campbell H. Primary osteosarcoma of the breast. Breast 2003;12:72-4.  Back to cited text no. 18
    


    Figures

  [Figure 2], [Figure 1]



 

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