Breast angiosarc0ma metastatic to the maxillary gingiva
Kyu Kyu Swe Win ~, Tadashi Yasuoka ~, Hiroaki Kamiya 2, Takuzo Jinno 2 1Department of Oral and Maxillofacial
Case report
Hospital, Nagoya, Japan
Surgery, Gifu University School of Medicine, Gifu, and 2Department of Oral and Maxiliofacial Surgery, Nagoya City University
K. K. S. Win, T. Yasuoka, H. Kamiya, T. Jinno." Breast angiosarcorna metastatic to the maxillary gingiva. Case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 282-283. Abstract. A case o f b r e a s t a n g i o s a r c o m a metastatic to the maxillary gingiva is reported. A review of the literature revealed only three previously r e p o r t e d cases. A n g i o s a r c o m a s often present as b e n i g n lesions. The surgeon should m a i n tain a high level of vigilance w h e n patients present with oral lesions a n d a history o f b r e a s t tumor.
Key words: breast angiosarcoma; metastasis; maxillary gingiva. Accepted for publication 14 July 1992
Case report
angiosarcoma of the right breast. The breast tumor had been initially noted as a mass about 30 mm in diameter 3 years before, and was excised with the overlying skin. The histopathology initially provided a diagnosis of capillary hemangioma. A local relapse was found 7 months after surgery, and the mass was again excised. The histopathology again confirmed the diagnosis of capillary hemangioma. Another local recurrence occurred 18 months after the first presentation, and an incisional biopsy then yielded a diagnosis of angiosarcoma. The patient was treated with 30 Gy of 6°Co irradiation for 3 months. Because of another local recurrence, a mastectomy of the breast was then performed. The maxillary right second and third molars were extracted to eliminate traumatic irritation (Fig. 1), and a second biopsy of the lesion confirmed the diagnosis of capillary hemangioma. The mass, however, was gradually growing in size during follow-up examinations, and a third biopsy was taken. This time the histopathologic features were consistent with the primary lesion of the breast,
T h e c o m m o n sites for metastasis o f b r e a s t a n g i o s a r c o m a are, in order o f frequency, lung, skin a n d s u b c u t a n e o u s tissue, bone, liver, brain, a n d o v a r y 2. T h e metastatic lesions are m a i n l y due to h e m a t o g e n o u s spread. A review of the literature yielded only three cases
A 59-year-old woman was referred to the Department of OMF Surgery, Nagoya City University Hospital, for assessment of a lump on the maxillary right molar gingiva. The patient had been aware of the tumor for 2 weeks. At the initial visit, an irregular, purple mass, approximately 20 mm in diameter, was found in the maxillary right first molar region. The mass was soft and slightly tender on palpation. A metal bridge was fixed between the maxillary right first premolar and second molar. A poorly demarcated radiolucency in the alveolar bone was noted on radiographic examination. The bridge was removed, and an incisional biopsy was performed at the buccal side of the lesion. The histopathology yielded a diagnosis of capillary hemangioma of the gingiva. The past medical history revealed an
Fig. 1. Clinical appearance of oral cavity after extraction of second and third molars. In mesial portion reddish vascular proliferation is visible. The lesion resembles a hemangioma.
Fig. 2. Photograph of specimen from third biopsy. Solid nests of endothelial cells are seen with scant cellular atypia. Close inspection discloses scant mitotic figures and collapsed capillaries (HE × 50).
A n g i o s a r c o m a is rare b o t h as p r i m a r y a n d as metastatic t u m o r in the oral cavity 4'5'6. A review of the literature revealed only three r e p o r t e d cases o f a n g i o s a r c o m a m e t a s t a t i c to the oral cavity 3'4'6. A n g i o s a r c o m a of the b r e a s t is also relatively rare, constituting app r o x i m a t e l y 3 9% o f all b r e a s t sarc o m a s 1'2. It often requires multiple biopsies before the diagnosis can be established because o f the t u m o r ' s resemblance in b o t h clinical a n d microscopic a p p e a r a n c e with other, b e n i g n lesions 2'8. There is also considerable v a r i a t i o n in b o t h b e h a v i o r a n d microscopic appearance in different a n a t o m i c locations. This p a p e r describes a case o f breast a n g i o s a r c o m a m e t a s t a t i c to the gingiva w i t h o u t dissemination.
and the diagnosis was subsequently established as a metastatic angiosarcoma (Fig. 2). Severe bleeding had been caused by traumatic occlusion, and for this reason, and also because of the seemingly well-controlled primary lesion and the fact that no obvious metastatic deposits in the other regions were present, a subtotal maxillectomy was undertaken by a Weber-Fergusson incision. The postoperative condition was uneventful for 1 month after the surgery. An acute cardiovascular failure then occurred, and the patient died because of it 2 d later. An autopsy was not performed.
Discussion
Breast angiosarcoma metastatic to gingiva of angiosarcoma metastatic to the oral cavity 3,4,6, and only one case o f metastatic angiosarcoma from the breast with multiple gingival involvement 4. Angiosarcoma metastatic to the maxillary gingiva is thus extremely rare. In the present case, the metastatic site was initially thought to be caused by an inflammation related to the poorly fitting bridge. Inflammatory lesions often cause misdiagnosis in the initial stage, as was also noted in previous cases o f gingival metastases f r o m angiosarcoma of distant origins 4'6. Angiosarcoma is often mistaken for hemangioma 2'7's or reactive inflammatory process 6. The false negative result in the histologic diagnosis may be attributable to a superficial biopsy which did not represent the tumor 7. Angiosarc o m a has a pleomorphic appearance with few mitotic figures. A relatively bland appearance of breast angiosarc o m a is usually maintained in the metastatic deposit 5. The appearance of recurrences and metastases is usually similar to that of the original neoplasm. There is, however, a tendency for recurrences and metastases to contain more solid tumor or to exhibit increased pleomorphism in a few instances 5. These
conditions may lead to misdiagnosis, as in the patient described. There is considerable variation in behavior when angiosarcomas occur at different anatomic sites. Therapeutic results for angiosarcoma in the oral cavity are more encouraging than those in other sites s, whereas breast angiosarcoma has the worst prognosis 2,5. M o s t patients with breast angiosarcoma die as a result of dissemination 2. Simple excision of the lesion as performed initially in many cases is invariably followed by local recurrence 2. The t u m o r appears radioresistant and responds poorly to chemotherapy j. Therefore, resection is the treatment of choice for the metastatic t u m o r when the primary lesion is under control. The case described emphasizes the need to maintain a high level of vigilance when inflammatory oral lesions are seen in patients with a history of breast angiosarcoma.
References 1. BUNDRED NJ, O'REILLY K, SMART JG. Long-term survival following bilateral breast angiosarcoma. Eur J Surg Oncol 1989: 15: 263-4. 2. CHEN KTK, KIRKEGAARDDD, BOCIAN
283
JJ. Angiosarcoma of the breast. Cancer 1980: 46:368 71. 3. CRYM~S T, TAYLOR RG. Angiosarcoma metastatic to the tongue. Report of a case. J Oral Surg 1966: 24: 63-6. 4. EPSTEIN JB, KNOWLING MA, LE RICHE JC. Multiple gingival metastases from angiosarcoma of the breast. Oral Surg 1987: 64: 554-7. 5. MADOOXJC, EvaNs HL. Angiosarcoma of skin and soft tissue: a study of fortyfour cases. Cancer 1981: 48: 1907-21. 6. TOTHBB, FLEMINGTJ, LOMBAJA, MARTIN JW. Angiosarcoma metastatic to the maxillary tuberosity gingiva. Oral Surg 1981: 52: 714. 7. WESLEY RK, MINTZ SM, WERTHEIMER FW. Primary malignant hemangioendothelioma of the gingiva: report of a case and review of the literature. Oral Surg 1975: 39: 103-12. 8. YASUOKAT, OKUMURAY, OKUDA T, OKA N. Hemangioma and malignant hemangioendothelioma of the maxillary sinus: case reports and clinical consideration. J Oral Maxillofac Surg 1990: 48:877 81. Address: Kyu Kyu Swe Win, BDS Department of Oral and Maxillofacial Surgery, Gifu University School of Medicine, 40, Tsukasamaehi, GiJu 500 Japan
Kyu Kyu Swe Win ~, Tadashi Yasuoka ~, Hiroaki Kamiya 2, Takuzo Jinno 2 1Department of Oral and Maxillofacial
Case report
Hospital, Nagoya, Japan
Surgery, Gifu University School of Medicine, Gifu, and 2Department of Oral and Maxiliofacial Surgery, Nagoya City University
K. K. S. Win, T. Yasuoka, H. Kamiya, T. Jinno." Breast angiosarcorna metastatic to the maxillary gingiva. Case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 282-283. Abstract. A case o f b r e a s t a n g i o s a r c o m a metastatic to the maxillary gingiva is reported. A review of the literature revealed only three previously r e p o r t e d cases. A n g i o s a r c o m a s often present as b e n i g n lesions. The surgeon should m a i n tain a high level of vigilance w h e n patients present with oral lesions a n d a history o f b r e a s t tumor.
Key words: breast angiosarcoma; metastasis; maxillary gingiva. Accepted for publication 14 July 1992
Case report
angiosarcoma of the right breast. The breast tumor had been initially noted as a mass about 30 mm in diameter 3 years before, and was excised with the overlying skin. The histopathology initially provided a diagnosis of capillary hemangioma. A local relapse was found 7 months after surgery, and the mass was again excised. The histopathology again confirmed the diagnosis of capillary hemangioma. Another local recurrence occurred 18 months after the first presentation, and an incisional biopsy then yielded a diagnosis of angiosarcoma. The patient was treated with 30 Gy of 6°Co irradiation for 3 months. Because of another local recurrence, a mastectomy of the breast was then performed. The maxillary right second and third molars were extracted to eliminate traumatic irritation (Fig. 1), and a second biopsy of the lesion confirmed the diagnosis of capillary hemangioma. The mass, however, was gradually growing in size during follow-up examinations, and a third biopsy was taken. This time the histopathologic features were consistent with the primary lesion of the breast,
T h e c o m m o n sites for metastasis o f b r e a s t a n g i o s a r c o m a are, in order o f frequency, lung, skin a n d s u b c u t a n e o u s tissue, bone, liver, brain, a n d o v a r y 2. T h e metastatic lesions are m a i n l y due to h e m a t o g e n o u s spread. A review of the literature yielded only three cases
A 59-year-old woman was referred to the Department of OMF Surgery, Nagoya City University Hospital, for assessment of a lump on the maxillary right molar gingiva. The patient had been aware of the tumor for 2 weeks. At the initial visit, an irregular, purple mass, approximately 20 mm in diameter, was found in the maxillary right first molar region. The mass was soft and slightly tender on palpation. A metal bridge was fixed between the maxillary right first premolar and second molar. A poorly demarcated radiolucency in the alveolar bone was noted on radiographic examination. The bridge was removed, and an incisional biopsy was performed at the buccal side of the lesion. The histopathology yielded a diagnosis of capillary hemangioma of the gingiva. The past medical history revealed an
Fig. 1. Clinical appearance of oral cavity after extraction of second and third molars. In mesial portion reddish vascular proliferation is visible. The lesion resembles a hemangioma.
Fig. 2. Photograph of specimen from third biopsy. Solid nests of endothelial cells are seen with scant cellular atypia. Close inspection discloses scant mitotic figures and collapsed capillaries (HE × 50).
A n g i o s a r c o m a is rare b o t h as p r i m a r y a n d as metastatic t u m o r in the oral cavity 4'5'6. A review of the literature revealed only three r e p o r t e d cases o f a n g i o s a r c o m a m e t a s t a t i c to the oral cavity 3'4'6. A n g i o s a r c o m a of the b r e a s t is also relatively rare, constituting app r o x i m a t e l y 3 9% o f all b r e a s t sarc o m a s 1'2. It often requires multiple biopsies before the diagnosis can be established because o f the t u m o r ' s resemblance in b o t h clinical a n d microscopic a p p e a r a n c e with other, b e n i g n lesions 2'8. There is also considerable v a r i a t i o n in b o t h b e h a v i o r a n d microscopic appearance in different a n a t o m i c locations. This p a p e r describes a case o f breast a n g i o s a r c o m a m e t a s t a t i c to the gingiva w i t h o u t dissemination.
and the diagnosis was subsequently established as a metastatic angiosarcoma (Fig. 2). Severe bleeding had been caused by traumatic occlusion, and for this reason, and also because of the seemingly well-controlled primary lesion and the fact that no obvious metastatic deposits in the other regions were present, a subtotal maxillectomy was undertaken by a Weber-Fergusson incision. The postoperative condition was uneventful for 1 month after the surgery. An acute cardiovascular failure then occurred, and the patient died because of it 2 d later. An autopsy was not performed.
Discussion
Breast angiosarcoma metastatic to gingiva of angiosarcoma metastatic to the oral cavity 3,4,6, and only one case o f metastatic angiosarcoma from the breast with multiple gingival involvement 4. Angiosarcoma metastatic to the maxillary gingiva is thus extremely rare. In the present case, the metastatic site was initially thought to be caused by an inflammation related to the poorly fitting bridge. Inflammatory lesions often cause misdiagnosis in the initial stage, as was also noted in previous cases o f gingival metastases f r o m angiosarcoma of distant origins 4'6. Angiosarcoma is often mistaken for hemangioma 2'7's or reactive inflammatory process 6. The false negative result in the histologic diagnosis may be attributable to a superficial biopsy which did not represent the tumor 7. Angiosarc o m a has a pleomorphic appearance with few mitotic figures. A relatively bland appearance of breast angiosarc o m a is usually maintained in the metastatic deposit 5. The appearance of recurrences and metastases is usually similar to that of the original neoplasm. There is, however, a tendency for recurrences and metastases to contain more solid tumor or to exhibit increased pleomorphism in a few instances 5. These
conditions may lead to misdiagnosis, as in the patient described. There is considerable variation in behavior when angiosarcomas occur at different anatomic sites. Therapeutic results for angiosarcoma in the oral cavity are more encouraging than those in other sites s, whereas breast angiosarcoma has the worst prognosis 2,5. M o s t patients with breast angiosarcoma die as a result of dissemination 2. Simple excision of the lesion as performed initially in many cases is invariably followed by local recurrence 2. The t u m o r appears radioresistant and responds poorly to chemotherapy j. Therefore, resection is the treatment of choice for the metastatic t u m o r when the primary lesion is under control. The case described emphasizes the need to maintain a high level of vigilance when inflammatory oral lesions are seen in patients with a history of breast angiosarcoma.
References 1. BUNDRED NJ, O'REILLY K, SMART JG. Long-term survival following bilateral breast angiosarcoma. Eur J Surg Oncol 1989: 15: 263-4. 2. CHEN KTK, KIRKEGAARDDD, BOCIAN
283
JJ. Angiosarcoma of the breast. Cancer 1980: 46:368 71. 3. CRYM~S T, TAYLOR RG. Angiosarcoma metastatic to the tongue. Report of a case. J Oral Surg 1966: 24: 63-6. 4. EPSTEIN JB, KNOWLING MA, LE RICHE JC. Multiple gingival metastases from angiosarcoma of the breast. Oral Surg 1987: 64: 554-7. 5. MADOOXJC, EvaNs HL. Angiosarcoma of skin and soft tissue: a study of fortyfour cases. Cancer 1981: 48: 1907-21. 6. TOTHBB, FLEMINGTJ, LOMBAJA, MARTIN JW. Angiosarcoma metastatic to the maxillary tuberosity gingiva. Oral Surg 1981: 52: 714. 7. WESLEY RK, MINTZ SM, WERTHEIMER FW. Primary malignant hemangioendothelioma of the gingiva: report of a case and review of the literature. Oral Surg 1975: 39: 103-12. 8. YASUOKAT, OKUMURAY, OKUDA T, OKA N. Hemangioma and malignant hemangioendothelioma of the maxillary sinus: case reports and clinical consideration. J Oral Maxillofac Surg 1990: 48:877 81. Address: Kyu Kyu Swe Win, BDS Department of Oral and Maxillofacial Surgery, Gifu University School of Medicine, 40, Tsukasamaehi, GiJu 500 Japan
