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Osteosarcoma of the breast—an unusual presentation
Clinical Radiology (2006) 61, 291–293
CASE REPORT
Osteosarcoma of the breast—an unusual presentation S. Choudharya,*, F. Hearna, J. Halla, I. Laidlawb Departments of aRadiology, and bSurgery, Frimley Park Hospital NHS Foundation Trust, Frimley, Surrey, UK
Introduction Cystosarcoma phyllodes is an uncommon primary breast malignancy which undergoes osteosarcomatous differentiation in a small subgroup of patients. Uptake of 99m technetium methylene diphosphonate (99mTcMDP) is known to occur within pulmonary, lymph node and soft tissue metastases arising from osteosarcomas of bone and is related to the presence of osteoid within these tumours. Uptake of 99mTcMDP has also been reported from pulmonary metastasis arising from osteosarcomas of the breast.1 Similar uptake would be expected from osteoid elements of the breast sarcoma itself, but this has not been previously documented. We present a case of an osteosarcoma of breast showing up as intense uptake on bone scintigraphy performed for unrelated joint pains. The hot spot seen projected over the right upper chest could not be explained satisfactorily at the time of the study and was thought to represent an artefact. A few weeks later, the patient presented with a breast lump, which was subsequently diagnosed as an osteosarcoma in phyllodes tumour. Retrospectively, the unexplained hot spot on bone scintigraphy corresponded to the site of tumour in the right breast.
Case report A 76-year-old female presented with gradually worsening bilateral thigh and knee pain of unknown aetiology. Clinical examination of the hips and knees revealed no significant abnormal findings. Bone scintigraphy was arranged to investigate further. The resulting image (Fig. 1(a) and (b)) * Correspondent: S. Choudhary, 80 Hilltop Road, Oldbury, Birmingham B68 9DY, UK. Tel.: C44 7810861699. E-mail address: [email protected] (S. Choudhary).
showed a solitary area of intense uptake anteriorly between the second and third ribs. The area was not due to her clothing, which was removed, and there was no evidence of contamination on her fingers. The chest radiograph was normal. A satisfactory explanation as to the origin of this uptake could not be found and it was thought to be an artefact. In addition, there was increased uptake around the right hip and medial compartment of the knees, suggesting degenerative changes that were not particularly severe. A few weeks later she presented with a lump in her right breast, which she had recently discovered. She had no previous breast problems and there was no family history of breast cancer. Examination of her right breast revealed a 4 cm diameter hard mobile lump in the superior aspect of her right breast, which was slightly tender to touch. The overlying skin was normal. Her axillae and left breast were also normal. She underwent further evaluation with bilateral mammograms and right breast ultrasound and fineneedle aspiration (FNA). The mammogram showed a well-defined lobulated mass with dense calcification (in retrospect ossification), in the upper medial part of the breast (Fig. 2). Ultrasound was difficult to interpret because of the calcification producing significant acoustic shadowing. It appeared like a well-defined, lobulated mass measuring 42!24 mm. Aspiration produced a small amount of turbid fluid, but not surprisingly with the calcification, complete aspiration was not possible. The imaging appearances were thought to be unusual and possibilities suggested included an intracystic papillary lesion or perhaps just calcification in an old cyst. Cytology of the fluid showed apocrine atypia in a cyst with old haemorrhage (C3). A trucut biopsy was performed, which suggested the presence of phyllodes tumour with focal cartilaginous and osseous metaplasia. Lipomatous, cartilaginous and osseous metaplasia have been
0009-9260/$ - see front matter Q 2005 The Royal College of Radiologists. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.crad.2005.11.007
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S. Choudhary et al.
Wide local excision of the lump was carried out. On macroscopic examination a well-defined nodular lesion measuring 3!2.8!2.5 cm with one third of the lesion showing calcification (?bone) and some cystic areas. Microscopic examination showed mixed epithelial and stromal components with stromal overgrowth and leaf like structures consistent with a phyllodes tumour. There were large areas of osteoid deposition associated with cellular neoplastic cartilaginous and bony tissue. The appearances were of osteosarcoma arising within phyllodes tumour. No vascular invasion was seen. As these tumours have a potential for distant metastasis and local recurrence, total mastectomy was performed following a body computed tomography (CT) that showed no distant metastasis. In retrospect, the ossification was not visible on the chest radiograph.
Discussion
Figure 1 (a) and (b) Tc99m MDP bone scan—anterior and posterior views. Increased uptake is demonstrated around the right hip and medial compartment of both knees suggesting moderate degenerative changes. A focal spot of intense uptake is present overlying the right anterior chest wall.
described within phyllodes tumours—though they are rare in those that are histologically benign. Keeping this in mind, complete excision with assessment of margins was suggested for accurate prediction of behaviour.
Figure 2 Mammogram showing a well defined lobulated mass with dense calcification (in retrospect ossification), in the upper medial part of the breast.
The vast majority of malignant breast neoplasms are of ductal or lobular epithelial origin. Mammary tumours containing bone, cartilage or osteoid tissue are rare in humans. On the basis of macroscopic and microscopic findings, these bone and cartilage containing tumours can be divided into four groups 2: (1) intra-ductal papillomas with stromal metaplasia (mixed tumours); (2) phyllode cystosarcoma with bone and cartilage as stromal components; (3) stromal sarcomas including osteogenic sarcoma of the breast; and (4) adenocarcinomas containing bone and cartilage owing to metaplasia of the epithelial cells. A histopathological review of 22 cases estimated that osteosarcomas arise within 1.3% of phyllodes tumours and histologically resemble those of skeletal origin.2 In 40% of cases, osteogenic sarcoma of breasts develop in a phyllode tumour. Roggen et al. reported a case of local recurrence of a phyllodes tumour of the breast presenting with widespread differentiation to a telangiectatic osteosarcoma 3. Osteogenic sarcomas may also arise from sarcomatous transformation of connective tissue elements of pre-existing benign breast neoplasms, notably fibroadenomas and intra-ductal papillomas.4–7 In exceptional cases, osteogenic sarcoma might represent a non-phyllodes sarcoma of the breast arising from the soft tissues of a previously normal breast.8 Breast tumours with malignant bone formation are usually metaplastic carcinomas. These tumours are characterized by rapid growth after an initial period of latency. Haematogenous
Osteosarcoma of the breast
spread occurs usually to the lungs. Infrequently, metaplastic bone formation may be found in other sarcomas of the breast-like angiosarcoma, malignant fibrous histiocytoma, pleomorphic liposarcoma and malignant melanoma. Nuclear medicine techniques play a definitive role in the diagnosis and follow-up of breast cancers.9 Scintimammography with technetium99m sestamibi is a useful non-invasive test to determine the presence of axillary lymph node metastases in breast carcinoma. Initial experience with 2-[F-18]fluoro-2-deoxy-D-glucose (FDG) positron emission tomography (PET) mammography has demonstrated its usefulness in diagnosis of primary breast carcinoma in patients with dense breast tissue, significant fibrocystic change, fibrosis after radiotherapy and patients with inconclusive results from other imaging methods.10 PET also has a high positive predictive value for regional lymph node involvement, detection of recurrent or metastatic carcinoma and identifying response to therapy earlier than any other imaging method. Bone scintigraphy is used for detection of skeletal metastases. Causes of soft tissue hot spots on bone imaging, especially in the region of the chest, include lung secondaries from osteosarcomas, soft tissue osteosarcomas arising from the chest wall or breast, and rarely, uptake in a primary breast carcinoma and breast prosthesis has also been documented. Ossification of soft tissue haematomas, hyperparathyroidism, post-ischaemic dystrophic soft tissue calcification as in previously infarcted myocardium and chronic pericardial effusion due to uraemic renal disease and
293
malignancy are other unusual causes of soft tissue tracer uptake on bone scintigraphy. The present case highlights the importance of following up unusual hot spots on bone scintigraphy, particularly those arising in soft tissues.
References 1. Bhartia SK, Kashyap P. Osteogenic pulmonary metastasis originating from a phyllodes tumour of the breast with osteosarcomatous differentiation. Aust Radiol 2005;49: 63—5. 2. Silver S, Tavassoli F. Osteosarcomatous differentiation in phyllodes tumors. Am J Surg Pathol 1999;23:815—21. 3. Graadt van Roggen JF, Zonderland HM, Welvaart K, Peterse JL, Hogendoorn PCW. Local recurrence of a phyllodes tumour of the breast presenting with widespread differentiation to a telangiectatic osteosarcoma. J Clin Pathol 1998;51:706—8. 4. Going JJ, Lumsden AB, Anderson TJ. A classical sarcoma of the breast: histology, immunohistochemistry and ultrastructure. Histopathology 1986;10:631—41. 5. Nishida Y, Kohno N, Furuya Y, et al. Mammary fibroadenoma showing osseous metaplasia: a case report. Jap J Cancer Clin 1989;35:1461—5. 6. Killick SB, McCann BG. Osteogenic sarcoma of the breast associated with fibroadenoma. Clin Oncol 1995;7:132—3. 7. Benediktsdottir K, Lagerberg F, Lundell L, Thulin A. Osteogenic sarcoma of the breast. Report of a case. Acta Pathol Microbiol Scand 1980;88A:161—5. 8. Arraztoa J, Oddo D, SanMartin S, Arraztoa JA, Becker P, Valuenzela J. Unusual sarcomas of the breast. Report of three cases. Rev Med Chil 1989;117:435—9. 9. Raileanu I, Grahek D, Rusu V, et al. Role of radioisotopic investigations in breast cancer diagnosis and follow-up. Rev Med Chir Soc Med Nat Iasi 2004;108:251—62. 10. Kumar A, Alavi A. FDG PET in the management of breast cancer. Radiol Clin North Am 2004;42:1113—22 [ix].
CASE REPORT
Osteosarcoma of the breast—an unusual presentation S. Choudharya,*, F. Hearna, J. Halla, I. Laidlawb Departments of aRadiology, and bSurgery, Frimley Park Hospital NHS Foundation Trust, Frimley, Surrey, UK
Introduction Cystosarcoma phyllodes is an uncommon primary breast malignancy which undergoes osteosarcomatous differentiation in a small subgroup of patients. Uptake of 99m technetium methylene diphosphonate (99mTcMDP) is known to occur within pulmonary, lymph node and soft tissue metastases arising from osteosarcomas of bone and is related to the presence of osteoid within these tumours. Uptake of 99mTcMDP has also been reported from pulmonary metastasis arising from osteosarcomas of the breast.1 Similar uptake would be expected from osteoid elements of the breast sarcoma itself, but this has not been previously documented. We present a case of an osteosarcoma of breast showing up as intense uptake on bone scintigraphy performed for unrelated joint pains. The hot spot seen projected over the right upper chest could not be explained satisfactorily at the time of the study and was thought to represent an artefact. A few weeks later, the patient presented with a breast lump, which was subsequently diagnosed as an osteosarcoma in phyllodes tumour. Retrospectively, the unexplained hot spot on bone scintigraphy corresponded to the site of tumour in the right breast.
Case report A 76-year-old female presented with gradually worsening bilateral thigh and knee pain of unknown aetiology. Clinical examination of the hips and knees revealed no significant abnormal findings. Bone scintigraphy was arranged to investigate further. The resulting image (Fig. 1(a) and (b)) * Correspondent: S. Choudhary, 80 Hilltop Road, Oldbury, Birmingham B68 9DY, UK. Tel.: C44 7810861699. E-mail address: [email protected] (S. Choudhary).
showed a solitary area of intense uptake anteriorly between the second and third ribs. The area was not due to her clothing, which was removed, and there was no evidence of contamination on her fingers. The chest radiograph was normal. A satisfactory explanation as to the origin of this uptake could not be found and it was thought to be an artefact. In addition, there was increased uptake around the right hip and medial compartment of the knees, suggesting degenerative changes that were not particularly severe. A few weeks later she presented with a lump in her right breast, which she had recently discovered. She had no previous breast problems and there was no family history of breast cancer. Examination of her right breast revealed a 4 cm diameter hard mobile lump in the superior aspect of her right breast, which was slightly tender to touch. The overlying skin was normal. Her axillae and left breast were also normal. She underwent further evaluation with bilateral mammograms and right breast ultrasound and fineneedle aspiration (FNA). The mammogram showed a well-defined lobulated mass with dense calcification (in retrospect ossification), in the upper medial part of the breast (Fig. 2). Ultrasound was difficult to interpret because of the calcification producing significant acoustic shadowing. It appeared like a well-defined, lobulated mass measuring 42!24 mm. Aspiration produced a small amount of turbid fluid, but not surprisingly with the calcification, complete aspiration was not possible. The imaging appearances were thought to be unusual and possibilities suggested included an intracystic papillary lesion or perhaps just calcification in an old cyst. Cytology of the fluid showed apocrine atypia in a cyst with old haemorrhage (C3). A trucut biopsy was performed, which suggested the presence of phyllodes tumour with focal cartilaginous and osseous metaplasia. Lipomatous, cartilaginous and osseous metaplasia have been
0009-9260/$ - see front matter Q 2005 The Royal College of Radiologists. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.crad.2005.11.007
292
S. Choudhary et al.
Wide local excision of the lump was carried out. On macroscopic examination a well-defined nodular lesion measuring 3!2.8!2.5 cm with one third of the lesion showing calcification (?bone) and some cystic areas. Microscopic examination showed mixed epithelial and stromal components with stromal overgrowth and leaf like structures consistent with a phyllodes tumour. There were large areas of osteoid deposition associated with cellular neoplastic cartilaginous and bony tissue. The appearances were of osteosarcoma arising within phyllodes tumour. No vascular invasion was seen. As these tumours have a potential for distant metastasis and local recurrence, total mastectomy was performed following a body computed tomography (CT) that showed no distant metastasis. In retrospect, the ossification was not visible on the chest radiograph.
Discussion
Figure 1 (a) and (b) Tc99m MDP bone scan—anterior and posterior views. Increased uptake is demonstrated around the right hip and medial compartment of both knees suggesting moderate degenerative changes. A focal spot of intense uptake is present overlying the right anterior chest wall.
described within phyllodes tumours—though they are rare in those that are histologically benign. Keeping this in mind, complete excision with assessment of margins was suggested for accurate prediction of behaviour.
Figure 2 Mammogram showing a well defined lobulated mass with dense calcification (in retrospect ossification), in the upper medial part of the breast.
The vast majority of malignant breast neoplasms are of ductal or lobular epithelial origin. Mammary tumours containing bone, cartilage or osteoid tissue are rare in humans. On the basis of macroscopic and microscopic findings, these bone and cartilage containing tumours can be divided into four groups 2: (1) intra-ductal papillomas with stromal metaplasia (mixed tumours); (2) phyllode cystosarcoma with bone and cartilage as stromal components; (3) stromal sarcomas including osteogenic sarcoma of the breast; and (4) adenocarcinomas containing bone and cartilage owing to metaplasia of the epithelial cells. A histopathological review of 22 cases estimated that osteosarcomas arise within 1.3% of phyllodes tumours and histologically resemble those of skeletal origin.2 In 40% of cases, osteogenic sarcoma of breasts develop in a phyllode tumour. Roggen et al. reported a case of local recurrence of a phyllodes tumour of the breast presenting with widespread differentiation to a telangiectatic osteosarcoma 3. Osteogenic sarcomas may also arise from sarcomatous transformation of connective tissue elements of pre-existing benign breast neoplasms, notably fibroadenomas and intra-ductal papillomas.4–7 In exceptional cases, osteogenic sarcoma might represent a non-phyllodes sarcoma of the breast arising from the soft tissues of a previously normal breast.8 Breast tumours with malignant bone formation are usually metaplastic carcinomas. These tumours are characterized by rapid growth after an initial period of latency. Haematogenous
Osteosarcoma of the breast
spread occurs usually to the lungs. Infrequently, metaplastic bone formation may be found in other sarcomas of the breast-like angiosarcoma, malignant fibrous histiocytoma, pleomorphic liposarcoma and malignant melanoma. Nuclear medicine techniques play a definitive role in the diagnosis and follow-up of breast cancers.9 Scintimammography with technetium99m sestamibi is a useful non-invasive test to determine the presence of axillary lymph node metastases in breast carcinoma. Initial experience with 2-[F-18]fluoro-2-deoxy-D-glucose (FDG) positron emission tomography (PET) mammography has demonstrated its usefulness in diagnosis of primary breast carcinoma in patients with dense breast tissue, significant fibrocystic change, fibrosis after radiotherapy and patients with inconclusive results from other imaging methods.10 PET also has a high positive predictive value for regional lymph node involvement, detection of recurrent or metastatic carcinoma and identifying response to therapy earlier than any other imaging method. Bone scintigraphy is used for detection of skeletal metastases. Causes of soft tissue hot spots on bone imaging, especially in the region of the chest, include lung secondaries from osteosarcomas, soft tissue osteosarcomas arising from the chest wall or breast, and rarely, uptake in a primary breast carcinoma and breast prosthesis has also been documented. Ossification of soft tissue haematomas, hyperparathyroidism, post-ischaemic dystrophic soft tissue calcification as in previously infarcted myocardium and chronic pericardial effusion due to uraemic renal disease and
293
malignancy are other unusual causes of soft tissue tracer uptake on bone scintigraphy. The present case highlights the importance of following up unusual hot spots on bone scintigraphy, particularly those arising in soft tissues.
References 1. Bhartia SK, Kashyap P. Osteogenic pulmonary metastasis originating from a phyllodes tumour of the breast with osteosarcomatous differentiation. Aust Radiol 2005;49: 63—5. 2. Silver S, Tavassoli F. Osteosarcomatous differentiation in phyllodes tumors. Am J Surg Pathol 1999;23:815—21. 3. Graadt van Roggen JF, Zonderland HM, Welvaart K, Peterse JL, Hogendoorn PCW. Local recurrence of a phyllodes tumour of the breast presenting with widespread differentiation to a telangiectatic osteosarcoma. J Clin Pathol 1998;51:706—8. 4. Going JJ, Lumsden AB, Anderson TJ. A classical sarcoma of the breast: histology, immunohistochemistry and ultrastructure. Histopathology 1986;10:631—41. 5. Nishida Y, Kohno N, Furuya Y, et al. Mammary fibroadenoma showing osseous metaplasia: a case report. Jap J Cancer Clin 1989;35:1461—5. 6. Killick SB, McCann BG. Osteogenic sarcoma of the breast associated with fibroadenoma. Clin Oncol 1995;7:132—3. 7. Benediktsdottir K, Lagerberg F, Lundell L, Thulin A. Osteogenic sarcoma of the breast. Report of a case. Acta Pathol Microbiol Scand 1980;88A:161—5. 8. Arraztoa J, Oddo D, SanMartin S, Arraztoa JA, Becker P, Valuenzela J. Unusual sarcomas of the breast. Report of three cases. Rev Med Chil 1989;117:435—9. 9. Raileanu I, Grahek D, Rusu V, et al. Role of radioisotopic investigations in breast cancer diagnosis and follow-up. Rev Med Chir Soc Med Nat Iasi 2004;108:251—62. 10. Kumar A, Alavi A. FDG PET in the management of breast cancer. Radiol Clin North Am 2004;42:1113—22 [ix].