- Email: [email protected]
Metastatic cutaneous carcinosarcoma to the tongue
Metastatic cutaneous carcinosarcoma to the tongue
1033
Case Report Head and Neck Oncology
Metastatic cutaneous carcinosarcoma to the tongue N. Tanaka, S. Hidaka, M. Yamamura, H. Ohyama, M. Urade: Metastatic cutaneous carcinosarcoma to the tongue. Int. J. Oral Maxillofac. Surg. 2010; 39: 1033–1036. # 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
N. Tanaka1,3,, S. Hidaka1, M. Yamamura1, H. Ohyama1,2, M. Urade1 1 Department of Oral and Maxillofacial Surgery, Japan; 2Department of Pathology, Hyogo College of Medicine, Hyogo, Japan; 3 The First Department of Oral and Maxillofacial Surgery, Osaka University Graduate School of Dentistry, Osaka, Japan
Abstract. Carcinosarcoma is a rare malignant tumour composed of a mixture of carcinomatous and sarcomatous elements. Carcinosarcoma metastatic to the tongue is extremely rare. An 84-year-old woman presented with a rapidly growing mass on the tongue. She had a history of surgery for carcinosarcoma of the occipital skin 9 months before. An excisional biopsy of the tongue mass was performed, and the lesion was histopathologically diagnosed as carcinosarcoma. PET after diagnosis showed multiple hot uptakes in the whole body. The patient died of the disease 2 months after diagnosis. Therapies for patients with metastatic malignant tumours to the oral cavity are difficult, especially in aggressive case such as this. To the authors’ knowledge, this is the first case of metastatic carcinosarcoma to the tongue.
Accepted for publication 21 April 2010 Available online 1 June 2010
Metastatic malignant tumours to the oral mucosa are uncommon and represent approximately 0.3% of all oral malignant tumours2,3,4. The tongue is the second most frequent site of metastasis in the oral mucosa and represents approximately 28% of all malignant metastatic tumours of the oral mucosa3. Carcinosarcoma is a rare malignant tumour composed of both carcinomatous and sarcomatous elements, which has been described in the breast, colon, lung and urogenital tract7. Primary cutaneous carcinosarcomas are rare, and approximately 50 cases have been reported in the English literature7. In cutaneous carcinosarcoma, metastasis has occurred in 7–29% of all patients6,7,9. A search revealed no case of carcinosarcoma metastatic to the oral mucosa. The authors present a rare case of metastatic cutaneous carcinosarcoma to the tongue.
(Fig. 2B). Based on these findings, the tumour was diagnosed as metastatic cutaneous carcinosarcoma to the tongue. CT scans of the neck and lung taken 1 month before the first visit to the authors’ clinic showed no obvious recurrence and metastasis. The patient was examined for the presence of other metastatic lesions. Positron emission tomography (PET) revealed multiple metastases to the lung, pericardium, liver, kidney, adrenal, bones, and muscles (Fig. 3). Tumour recurrence in the tongue was found 2 weeks after the excisional biopsy, although tumour cells were not seen in the surgical margin. The authors recommended systemic chemotherapy including cisplatin and docetaxel, but the patient refused it. She received palliative treatment including pain control and died of the disease 2 months after diagnosis.
Case report
An 84-year-old woman was referred with a rapidly growing mass on the right side of the tongue. She had a medical history of cutaneous carcinosarcoma of the occipital
region, which had been treated surgically 9 months before. She had been followed up after surgery with no evidence of recurrence and metastasis. On physical examination, a demarcated hard mass measuring 2.0 cm 1.0 cm was seen in the right inferior surface of the tongue (Fig. 1). The authors clinically diagnosed it as a benign tongue tumour, but were aware of the possibility of metastatic carcinosarcoma. An excisional biopsy with a 5 mm safety margin was removed for histological diagnosis. Histopathologically, the tumour showed carcinomatous and sarcomatous components (Fig. 2A). The carcinomatous tumour cells formed nests without keratinization and the sarcomatous tumour cells showed a fibrous spindle shape. Immunohistochemically, the carcinomatous tumour cells were positive for pan-keratin (AE1/AE3; Fig. 2B), vimentin (Fig. 2C), and neuron specific enolase (NSE), but negative for a-smooth muscle actin (a-SMA; Fig. 2D) and cytokertatin 20 (CK20). The sarcomatous tumour cells were positive for vimentin (Fig. 2C), NSE, a-SMA (Fig. 2D), and CK20, but negative for AE1/AE3
Discussion
The gingiva is the most common metastatic site for malignant tumours to the oral cavity and the tongue is the second3. The most common primary sites are the lung in men
1034
Tanaka et al.
Fig. 1. A bulging mass with clear boundary in the right inferior surface of the tongue.
and the breast in women. The skin is the third primary site. 90% of metastatic cutaneous tumours are malignant melanoma and 10% are basal cell carcinoma3. A search revealed no reported case of metastatic cutaneous carcinosarcoma to the oral mucosa. Prognosis for patients with metastatic tumour to the oral mucosa is generally
poor because most die within a year. Palliative therapies such as chemotherapy and/or radiotherapy are usually chosen for management of metastatic tumour to the oral mucosa. In the case of a single metastatic focus, surgical excision may be chosen as a therapy for control of the metastatic tumour. The authors decided to perform excisional biopsy in this case
because it was a demarcated tumour, although the possibility of metastatic carcinosarcoma could not be ruled out. In the event of the tumour being diagnosed as metastatic carcinosarcoma, the authors intended to perform systemic screening to aid treatment design. From the result of this screening, they suggested systemic chemotherapy to the patient. Carcinosarcoma is a rare malignant tumour composed of a malignant epithelial component and a malignant mesenchymal component. Cutaneous carcinosarcoma is extremely rare, and there are fewer than 50 cases to date in the literature7. These tumours are commonly surgically excised. There is no evidence for the usefulness of radiotherapy or chemotherapy for cutaneous carcinosarcoma because only a small number of patients received such therapy8. No report described the treatment regimen for cutaneous carcinosarcoma in detail. Chemotherapy with platinum compounds and taxanes has been reported for female genital carcinosarcoma1, although the usefulness of this regimen was unclear. The authors suggested systemic chemotherapy with cisplatin and docetaxel in this case.
Fig. 2. Histological and immunohistochemical staining of the biopsy specimen. (A) A mixture of carcinomatous and sarcomatous tumour cells was seen (H&E stain, original magnification 100). The carcinomatous tumour cells were positive for AE1/AE3 (B) and vimentin (C), but negative for a-SMA (D). The sarcomatous tumour cells were positive for vimentin (C) and a-SMA (D), but negative for AE1/AE3 (B) except a few cells in the border of the sarcomatous and the epithelial component that were positive for AE1/AE3 (arrow) (B–D original magnification 200).
Metastatic cutaneous carcinosarcoma to the tongue
1035
with vimentin. Immunohistochemistry revealed a few sarcomatous cells positive for AE1/AE3 in the border of the sarcomatous and the epithelial component. This suggested the presence of a single common precursor cell. The pathogenesis of this case was considered to be divergence theory. In conclusion, the authors presented a case of metastatic cutaneous carcinosarcoma to the tongue, which is the first case of metastatic carcinosarcoma to the oral cavity. The pathogenesis of carcinosarcoma is unclear, but this tumour was very aggressive, resulting in poor prognosis. These metastatic tumours are thought to be more aggressive and the prognosis is very poor. More research to elucidate the pathogenesis of carcinosarcoma and to develop an effective therapy are needed.
Funding
None.
Competing interests
None declared. Fig. 3. Coronal PET showing multiple metastases to the lung, pericardium, liver, kidney, adrenal, bones, and muscles.
Ethical approval
Not required. This tumour often has a period of recent rapid growth and metastasis occurs in 7– 29%6,7,9. The most common metastatic site was the lung (19%), followed by the bone and brain (5%)9. A search revealed that few cases develop multiple metastases6. In the present case, the tongue lesion was one of the systemic metastases showing rapid growth. This tumour was thought to be aggressive and the authors could not find such a progressive case. Three major theories have been proposed for the pathogenesis of carcinosarcomas. The first, known as the collision theory, proposes that two individual stem cells may simultaneously and independently undergo malignant transformation and are actually separate tumours that have collided7. The second, composition theory is that the spindle cell component is a reaction to the carcinoma7. The third, divergence theory is that individual elements are derived from a single common precursor cell7. In most cases of oesophageal carcinosarcoma, it has been suggested that the
sarcomatous component generally results from differentiation of squamous cell carcinoma cells into mesenchymal tumour cells. The reasons for this suggestion are that most carcinosarcomas contain a transitional zone between carcinomatous and sarcomatous components10 and that identical genetic alterations are observed in both components5. The pathogenesis of most oesophageal carcinosarcomas has been linked to divergence theory and this theory has also been applied to cutaneous carcinosarcoma6. When considering the pathogenesis of carcinosarcoma, the presence of a transitional zone or genuine sarcomatous components such as osteoblastic or rhabdomyoid features, or both, is a key feature. Differential diagnosis with spindle cell carcinoma or reactive spindle cell is important in the absence of genuine sarcomatous components. In the present case, the sarcomatous component is considered a true tumour, since it showed cellular atypism, abnormal increase of mitosis, a negative reaction with AE1/AE3 and a positive reaction
References 1. Ang JE, Shah RN, Everard M, Keyzor C, Coombes I, Jenkins A, Thomas K, A’Herm R, Jones RL, Blake P, Gabra H, Hall G, Gore ME, Kaye SB. A feasibility study of sequential doublet chemotherapy comprising carboplatin– doxorubicin and carboplatin–paclitaxel for advanced endometrial adenocarcinoma and carcinosarcoma. Ann Oncol 2009: 20: 1787–1793. 2. Aniceto GS, Penin AG. Tumor metastatic to the mandible: analysis of nine cases and review of literature. J Oral Maxillofac Surg 1990: 48: 246–251. 3. Hirshberg A, Leibovich P, Buchner A. Metastases to the oral mucosa: analysis of 157 cases. J Oral Pathol Med 1993: 22: 385–390. 4. Hirshberg A, Leibovich P, Buchner A. Metastatic tumors to the jawbones: analysis of 390 cases. J Oral Pathol Med 1994: 23: 337–341. 5. Matsumoto T, Fujii H, Arakawa A, Yamasaki S, Sonoue H, Hattori K, Kajiyama Y, Hirose S, Tsurumaru M. Loss of heterozygosity analysis shows monoclonal evolution with frequent genetic progression and divergence in
1036
Tanaka et al.
esophageal carcinosarcoma. Hum Pathol 2004: 35: 322–327. 6. Patel NK, McKee PH, Smith NP, Fletcher CD. Primary metaplastic carcinoma (carcinosarcoma) of the skin. A clinicopathologic study of four cases and review of the literature. Am J Dermatopathol 1997: 19: 363–372. 7. Rose RF, Merchant W, Stables GI, Lyon CL, Platt A. Basal cell carcinoma with a sarcomatous component (carcinosarcoma): a series of 5 cases and a review of the literature. J Am Acad Dermatol 2008: 59: 627–632. 8. Syme-Grant J, Syme-Grant NJ, Motta L, Stevenson JH, Evans AT.
Are primary cutaneous carcinosarcomas underdiagnosed? Five cases and a review of the literature. J Plast Reconstr Surg 2006: 59: 1402–1408. 9. Tran TA, Muller S, Chaudahri P, Carlson JA. Cutaneous carcinosarcoma: adnexal vs. epidermal type define high- and low-risk tumors. Result of a meta-analysis. J Cutan Pathol 2005: 32: 2–11. 10. Wang ZY, Itabashi M, Hirota T, Watanabe H, Kato H. Immunohistochemical study of the histogenesis of esophageal carcinosarcoma. Jpn J Clin Oncol 1992: 226: 377–386.
Address: Noriaki Tanaka The First Department of Oral and Maxillofacial Surgery Osaka University Graduate School of Dentistry 1-8, Yamadaoka Suita Osaka 565-0871 Japan Tel: +81 6 6879 2936 Fax: +81 6 6876 5298 E–mail: [email protected] doi:10.1016/j.ijom.2010.04.041
Case Report Oral Medicine
Plasmablastic lymphoma of the oral cavity in immunocompetent patients: report of two cases
D. D. Rao1, N. Aggarwal1, V. Anehosur1, H. Doddihal2, M. Shiraganvi1, K. Gopalkrishnan1 1 Department of Oral and Maxillofacial Surgery, S.D.M. College of Dental Sciences and Hospital, Sattur, Dharwad, Karnataka, India; 2Karnataka Cancer Therapy & Research Institute, Navnagar, Hubli, Karnataka, India
D. D. Rao, N. Aggarwal, V. Anehosur, H. Doddihal, M. Shiraganvi, K. Gopalkrishnan: Plasmablastic lymphoma of the oral cavity in immunocompetent patients: report of two cases. Int. J. Oral Maxillofac. Surg. 2010; 39: 1036–1039. # 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. Plasmablastic lymphoma (PBL) is a diffuse large B-cell lymphoma, a new entity, the incidence of which is increasing, predominantly in the oral cavity of immunocompromised patients, most of whom are infected with human immunodeficiency virus (HIV); only four cases have been reported in non-HIVinfected patients. This report details two cases of PBL of the oral cavity in immunocompetent patients. In both cases, the tumour manifested as a solitary, firm mandibular swelling with dense infiltration of plasma and blastoid cells in the stroma with strong positivity for CD 138 and negativity for CD 20. The patients received radiotherapy and chemotherapy, which resulted in complete remission of the lesion. This is the only case report of two cases of PBL in the oral cavity in immunocompetent patients in which both patients received treatment and showed complete remission of the lesion.
Accepted for publication 21 April 2010
Non-Hodgkin’s lymphoma (NHL) comprises a group of malignant lympho-proliferative diseases and often disseminates into extra-nodal sites (33%). The commonest site of extra-nodal presentation of NHL is the gastrointestinal tract. Primary extra-
extra-nodal lymphomas4 with the palate being the predominant site3. Plasmablastic lymphoma (PBL) is an aggressive subtype of non-Hodgkin’s lymphoma, which frequently arises in the oral cavity of human immunodeficiency virus
nodal lymphomas of the head and neck region are relatively rare and comprise about 4% of all head and neck tumours, with Waldeyer’s ring being the most common site of presentation. The involvement of the oral cavity accounts for 0.1–5% of
1033
Case Report Head and Neck Oncology
Metastatic cutaneous carcinosarcoma to the tongue N. Tanaka, S. Hidaka, M. Yamamura, H. Ohyama, M. Urade: Metastatic cutaneous carcinosarcoma to the tongue. Int. J. Oral Maxillofac. Surg. 2010; 39: 1033–1036. # 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
N. Tanaka1,3,, S. Hidaka1, M. Yamamura1, H. Ohyama1,2, M. Urade1 1 Department of Oral and Maxillofacial Surgery, Japan; 2Department of Pathology, Hyogo College of Medicine, Hyogo, Japan; 3 The First Department of Oral and Maxillofacial Surgery, Osaka University Graduate School of Dentistry, Osaka, Japan
Abstract. Carcinosarcoma is a rare malignant tumour composed of a mixture of carcinomatous and sarcomatous elements. Carcinosarcoma metastatic to the tongue is extremely rare. An 84-year-old woman presented with a rapidly growing mass on the tongue. She had a history of surgery for carcinosarcoma of the occipital skin 9 months before. An excisional biopsy of the tongue mass was performed, and the lesion was histopathologically diagnosed as carcinosarcoma. PET after diagnosis showed multiple hot uptakes in the whole body. The patient died of the disease 2 months after diagnosis. Therapies for patients with metastatic malignant tumours to the oral cavity are difficult, especially in aggressive case such as this. To the authors’ knowledge, this is the first case of metastatic carcinosarcoma to the tongue.
Accepted for publication 21 April 2010 Available online 1 June 2010
Metastatic malignant tumours to the oral mucosa are uncommon and represent approximately 0.3% of all oral malignant tumours2,3,4. The tongue is the second most frequent site of metastasis in the oral mucosa and represents approximately 28% of all malignant metastatic tumours of the oral mucosa3. Carcinosarcoma is a rare malignant tumour composed of both carcinomatous and sarcomatous elements, which has been described in the breast, colon, lung and urogenital tract7. Primary cutaneous carcinosarcomas are rare, and approximately 50 cases have been reported in the English literature7. In cutaneous carcinosarcoma, metastasis has occurred in 7–29% of all patients6,7,9. A search revealed no case of carcinosarcoma metastatic to the oral mucosa. The authors present a rare case of metastatic cutaneous carcinosarcoma to the tongue.
(Fig. 2B). Based on these findings, the tumour was diagnosed as metastatic cutaneous carcinosarcoma to the tongue. CT scans of the neck and lung taken 1 month before the first visit to the authors’ clinic showed no obvious recurrence and metastasis. The patient was examined for the presence of other metastatic lesions. Positron emission tomography (PET) revealed multiple metastases to the lung, pericardium, liver, kidney, adrenal, bones, and muscles (Fig. 3). Tumour recurrence in the tongue was found 2 weeks after the excisional biopsy, although tumour cells were not seen in the surgical margin. The authors recommended systemic chemotherapy including cisplatin and docetaxel, but the patient refused it. She received palliative treatment including pain control and died of the disease 2 months after diagnosis.
Case report
An 84-year-old woman was referred with a rapidly growing mass on the right side of the tongue. She had a medical history of cutaneous carcinosarcoma of the occipital
region, which had been treated surgically 9 months before. She had been followed up after surgery with no evidence of recurrence and metastasis. On physical examination, a demarcated hard mass measuring 2.0 cm 1.0 cm was seen in the right inferior surface of the tongue (Fig. 1). The authors clinically diagnosed it as a benign tongue tumour, but were aware of the possibility of metastatic carcinosarcoma. An excisional biopsy with a 5 mm safety margin was removed for histological diagnosis. Histopathologically, the tumour showed carcinomatous and sarcomatous components (Fig. 2A). The carcinomatous tumour cells formed nests without keratinization and the sarcomatous tumour cells showed a fibrous spindle shape. Immunohistochemically, the carcinomatous tumour cells were positive for pan-keratin (AE1/AE3; Fig. 2B), vimentin (Fig. 2C), and neuron specific enolase (NSE), but negative for a-smooth muscle actin (a-SMA; Fig. 2D) and cytokertatin 20 (CK20). The sarcomatous tumour cells were positive for vimentin (Fig. 2C), NSE, a-SMA (Fig. 2D), and CK20, but negative for AE1/AE3
Discussion
The gingiva is the most common metastatic site for malignant tumours to the oral cavity and the tongue is the second3. The most common primary sites are the lung in men
1034
Tanaka et al.
Fig. 1. A bulging mass with clear boundary in the right inferior surface of the tongue.
and the breast in women. The skin is the third primary site. 90% of metastatic cutaneous tumours are malignant melanoma and 10% are basal cell carcinoma3. A search revealed no reported case of metastatic cutaneous carcinosarcoma to the oral mucosa. Prognosis for patients with metastatic tumour to the oral mucosa is generally
poor because most die within a year. Palliative therapies such as chemotherapy and/or radiotherapy are usually chosen for management of metastatic tumour to the oral mucosa. In the case of a single metastatic focus, surgical excision may be chosen as a therapy for control of the metastatic tumour. The authors decided to perform excisional biopsy in this case
because it was a demarcated tumour, although the possibility of metastatic carcinosarcoma could not be ruled out. In the event of the tumour being diagnosed as metastatic carcinosarcoma, the authors intended to perform systemic screening to aid treatment design. From the result of this screening, they suggested systemic chemotherapy to the patient. Carcinosarcoma is a rare malignant tumour composed of a malignant epithelial component and a malignant mesenchymal component. Cutaneous carcinosarcoma is extremely rare, and there are fewer than 50 cases to date in the literature7. These tumours are commonly surgically excised. There is no evidence for the usefulness of radiotherapy or chemotherapy for cutaneous carcinosarcoma because only a small number of patients received such therapy8. No report described the treatment regimen for cutaneous carcinosarcoma in detail. Chemotherapy with platinum compounds and taxanes has been reported for female genital carcinosarcoma1, although the usefulness of this regimen was unclear. The authors suggested systemic chemotherapy with cisplatin and docetaxel in this case.
Fig. 2. Histological and immunohistochemical staining of the biopsy specimen. (A) A mixture of carcinomatous and sarcomatous tumour cells was seen (H&E stain, original magnification 100). The carcinomatous tumour cells were positive for AE1/AE3 (B) and vimentin (C), but negative for a-SMA (D). The sarcomatous tumour cells were positive for vimentin (C) and a-SMA (D), but negative for AE1/AE3 (B) except a few cells in the border of the sarcomatous and the epithelial component that were positive for AE1/AE3 (arrow) (B–D original magnification 200).
Metastatic cutaneous carcinosarcoma to the tongue
1035
with vimentin. Immunohistochemistry revealed a few sarcomatous cells positive for AE1/AE3 in the border of the sarcomatous and the epithelial component. This suggested the presence of a single common precursor cell. The pathogenesis of this case was considered to be divergence theory. In conclusion, the authors presented a case of metastatic cutaneous carcinosarcoma to the tongue, which is the first case of metastatic carcinosarcoma to the oral cavity. The pathogenesis of carcinosarcoma is unclear, but this tumour was very aggressive, resulting in poor prognosis. These metastatic tumours are thought to be more aggressive and the prognosis is very poor. More research to elucidate the pathogenesis of carcinosarcoma and to develop an effective therapy are needed.
Funding
None.
Competing interests
None declared. Fig. 3. Coronal PET showing multiple metastases to the lung, pericardium, liver, kidney, adrenal, bones, and muscles.
Ethical approval
Not required. This tumour often has a period of recent rapid growth and metastasis occurs in 7– 29%6,7,9. The most common metastatic site was the lung (19%), followed by the bone and brain (5%)9. A search revealed that few cases develop multiple metastases6. In the present case, the tongue lesion was one of the systemic metastases showing rapid growth. This tumour was thought to be aggressive and the authors could not find such a progressive case. Three major theories have been proposed for the pathogenesis of carcinosarcomas. The first, known as the collision theory, proposes that two individual stem cells may simultaneously and independently undergo malignant transformation and are actually separate tumours that have collided7. The second, composition theory is that the spindle cell component is a reaction to the carcinoma7. The third, divergence theory is that individual elements are derived from a single common precursor cell7. In most cases of oesophageal carcinosarcoma, it has been suggested that the
sarcomatous component generally results from differentiation of squamous cell carcinoma cells into mesenchymal tumour cells. The reasons for this suggestion are that most carcinosarcomas contain a transitional zone between carcinomatous and sarcomatous components10 and that identical genetic alterations are observed in both components5. The pathogenesis of most oesophageal carcinosarcomas has been linked to divergence theory and this theory has also been applied to cutaneous carcinosarcoma6. When considering the pathogenesis of carcinosarcoma, the presence of a transitional zone or genuine sarcomatous components such as osteoblastic or rhabdomyoid features, or both, is a key feature. Differential diagnosis with spindle cell carcinoma or reactive spindle cell is important in the absence of genuine sarcomatous components. In the present case, the sarcomatous component is considered a true tumour, since it showed cellular atypism, abnormal increase of mitosis, a negative reaction with AE1/AE3 and a positive reaction
References 1. Ang JE, Shah RN, Everard M, Keyzor C, Coombes I, Jenkins A, Thomas K, A’Herm R, Jones RL, Blake P, Gabra H, Hall G, Gore ME, Kaye SB. A feasibility study of sequential doublet chemotherapy comprising carboplatin– doxorubicin and carboplatin–paclitaxel for advanced endometrial adenocarcinoma and carcinosarcoma. Ann Oncol 2009: 20: 1787–1793. 2. Aniceto GS, Penin AG. Tumor metastatic to the mandible: analysis of nine cases and review of literature. J Oral Maxillofac Surg 1990: 48: 246–251. 3. Hirshberg A, Leibovich P, Buchner A. Metastases to the oral mucosa: analysis of 157 cases. J Oral Pathol Med 1993: 22: 385–390. 4. Hirshberg A, Leibovich P, Buchner A. Metastatic tumors to the jawbones: analysis of 390 cases. J Oral Pathol Med 1994: 23: 337–341. 5. Matsumoto T, Fujii H, Arakawa A, Yamasaki S, Sonoue H, Hattori K, Kajiyama Y, Hirose S, Tsurumaru M. Loss of heterozygosity analysis shows monoclonal evolution with frequent genetic progression and divergence in
1036
Tanaka et al.
esophageal carcinosarcoma. Hum Pathol 2004: 35: 322–327. 6. Patel NK, McKee PH, Smith NP, Fletcher CD. Primary metaplastic carcinoma (carcinosarcoma) of the skin. A clinicopathologic study of four cases and review of the literature. Am J Dermatopathol 1997: 19: 363–372. 7. Rose RF, Merchant W, Stables GI, Lyon CL, Platt A. Basal cell carcinoma with a sarcomatous component (carcinosarcoma): a series of 5 cases and a review of the literature. J Am Acad Dermatol 2008: 59: 627–632. 8. Syme-Grant J, Syme-Grant NJ, Motta L, Stevenson JH, Evans AT.
Are primary cutaneous carcinosarcomas underdiagnosed? Five cases and a review of the literature. J Plast Reconstr Surg 2006: 59: 1402–1408. 9. Tran TA, Muller S, Chaudahri P, Carlson JA. Cutaneous carcinosarcoma: adnexal vs. epidermal type define high- and low-risk tumors. Result of a meta-analysis. J Cutan Pathol 2005: 32: 2–11. 10. Wang ZY, Itabashi M, Hirota T, Watanabe H, Kato H. Immunohistochemical study of the histogenesis of esophageal carcinosarcoma. Jpn J Clin Oncol 1992: 226: 377–386.
Address: Noriaki Tanaka The First Department of Oral and Maxillofacial Surgery Osaka University Graduate School of Dentistry 1-8, Yamadaoka Suita Osaka 565-0871 Japan Tel: +81 6 6879 2936 Fax: +81 6 6876 5298 E–mail: [email protected] doi:10.1016/j.ijom.2010.04.041
Case Report Oral Medicine
Plasmablastic lymphoma of the oral cavity in immunocompetent patients: report of two cases
D. D. Rao1, N. Aggarwal1, V. Anehosur1, H. Doddihal2, M. Shiraganvi1, K. Gopalkrishnan1 1 Department of Oral and Maxillofacial Surgery, S.D.M. College of Dental Sciences and Hospital, Sattur, Dharwad, Karnataka, India; 2Karnataka Cancer Therapy & Research Institute, Navnagar, Hubli, Karnataka, India
D. D. Rao, N. Aggarwal, V. Anehosur, H. Doddihal, M. Shiraganvi, K. Gopalkrishnan: Plasmablastic lymphoma of the oral cavity in immunocompetent patients: report of two cases. Int. J. Oral Maxillofac. Surg. 2010; 39: 1036–1039. # 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. Plasmablastic lymphoma (PBL) is a diffuse large B-cell lymphoma, a new entity, the incidence of which is increasing, predominantly in the oral cavity of immunocompromised patients, most of whom are infected with human immunodeficiency virus (HIV); only four cases have been reported in non-HIVinfected patients. This report details two cases of PBL of the oral cavity in immunocompetent patients. In both cases, the tumour manifested as a solitary, firm mandibular swelling with dense infiltration of plasma and blastoid cells in the stroma with strong positivity for CD 138 and negativity for CD 20. The patients received radiotherapy and chemotherapy, which resulted in complete remission of the lesion. This is the only case report of two cases of PBL in the oral cavity in immunocompetent patients in which both patients received treatment and showed complete remission of the lesion.
Accepted for publication 21 April 2010
Non-Hodgkin’s lymphoma (NHL) comprises a group of malignant lympho-proliferative diseases and often disseminates into extra-nodal sites (33%). The commonest site of extra-nodal presentation of NHL is the gastrointestinal tract. Primary extra-
extra-nodal lymphomas4 with the palate being the predominant site3. Plasmablastic lymphoma (PBL) is an aggressive subtype of non-Hodgkin’s lymphoma, which frequently arises in the oral cavity of human immunodeficiency virus
nodal lymphomas of the head and neck region are relatively rare and comprise about 4% of all head and neck tumours, with Waldeyer’s ring being the most common site of presentation. The involvement of the oral cavity accounts for 0.1–5% of