前舌腺由来と考えられた脱分化型上皮筋上皮癌の1例

Epithelial myoepithelial carcinoma (EMC) is a rare malignant tumor accounting for approximately 4.1% of all salivary gland malignant tumors arising mainly in the parotid gland. We report an extremely rare case of EMC with high grade transformation originating from the anterior tongue gland. The pati...

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Published in日本口腔外科学会雑誌 Vol. 66; no. 9; pp. 444 - 449
Main Authors 粕谷, 賢志, 下山, 泰明, 関口, 眞人, 飯野, 光喜, 櫻井, 博理
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.09.2020
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ISSN0021-5163
2186-1579
DOI10.5794/jjoms.66.444

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Abstract Epithelial myoepithelial carcinoma (EMC) is a rare malignant tumor accounting for approximately 4.1% of all salivary gland malignant tumors arising mainly in the parotid gland. We report an extremely rare case of EMC with high grade transformation originating from the anterior tongue gland. The patient was a 66-year-old man. On clinical examination, a 50 × 35 mm elastic, hard mass was found in the tongue tip on the left side. CT and MRI showed a clear boundary tumor mass with a necrotic or bleeding region. After incisional biopsy, the mass was diagnosed as EMC. Partial glossectomy was performed under general anesthesia. Histologically, the tumor had two distinct carcinomatous components. One component showed the features of EMC, and the other showed high-grade transformation features. The Ki-67 labelling index of the EMC component was 3%, and that of the high-grade transformation component was 50%. The final histological diagnosis was EMC with high grade transformation. Six months postoperatively, he was hospitalized because of ileus caused by metastatic peritoneal dissemination of EMC and died of multiple organ failure.
AbstractList Epithelial myoepithelial carcinoma (EMC) is a rare malignant tumor accounting for approximately 4.1% of all salivary gland malignant tumors arising mainly in the parotid gland. We report an extremely rare case of EMC with high grade transformation originating from the anterior tongue gland. The patient was a 66-year-old man. On clinical examination, a 50 × 35 mm elastic, hard mass was found in the tongue tip on the left side. CT and MRI showed a clear boundary tumor mass with a necrotic or bleeding region. After incisional biopsy, the mass was diagnosed as EMC. Partial glossectomy was performed under general anesthesia. Histologically, the tumor had two distinct carcinomatous components. One component showed the features of EMC, and the other showed high-grade transformation features. The Ki-67 labelling index of the EMC component was 3%, and that of the high-grade transformation component was 50%. The final histological diagnosis was EMC with high grade transformation. Six months postoperatively, he was hospitalized because of ileus caused by metastatic peritoneal dissemination of EMC and died of multiple organ failure.
Author 下山, 泰明
櫻井, 博理
飯野, 光喜
粕谷, 賢志
関口, 眞人
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References 27) Lima FJ, Port DE, et al : Epithelial-myoepithelial carcinoma of high grade transformation: the case report in the buccal mucosa. Open Dent J 6: 111-117, 2012.
21) Fonseca I, Felix A, et al : Dedifferentiation in salivary glamd carcinomas. Am J Surg Pathol 24: 469-471, 2000.
4) Nagao T : “Dedifferentiation” and high-grade transformation in salivary gland carcinomas. Head and Neck Pathol 7: 37-47, 2013.
6) 柴田章夫, 木村将士, 他:前舌腺由来と考えられた腺様囊胞癌の1例.口腔腫瘍 28: 15-19, 2016.
7) Seethala RR, Barnes EL, et al : Epithelial-myoepithelial carcinoma : a review of the clinicopathologic spectrum and immunophenotypic characteristics in 61 tumors of the salivary glands and upper aero digestive tract. Am J Surg Pathol 31: 44-57, 2007.
19) Simpson RH, Clarke TJ, et al : Epithelial-myoepithelial carcinoma of salivary glands. J Clin Pathol 44: 419-423, 1991.
26) 関 伸彦, 山崎徳和, 他:耳下腺に発生した脱分化型上皮筋上皮癌の1例.耳喉頭頸 83: 630-633, 2011.
11) Kusafuka K, Takizawa Y, et al : Dedifferentiated epithelial-myoepithelial carcinoma of the parotid gland : a case report of immunohistochemical analysis and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 106: 85-91, 2008.
23) Niederhagen M, Zengel P, et al : Secondary high-malignant transformation of a low-malignant epithelial-myoepithelial carcinoma. Pathologe 30: 461-465, 2009.
9) 原田博史:唾液腺癌におけるいわゆる“脱分化”について.病理と臨床 27: 486-487, 2009.
2) Fonseca I and Soares J : Epithelial-myoepithelial carcinoma. In Barnes L, Eveson JW, eds ; WHO Classification of Tumours. Pathology and Genetics of Head and Neck Tumours. IARC Press, Lyon, 2005, p225-226.
12) Baker AR, Ohanessian SE, et al : Dedifferentiated epithelial-myoepithelial carcinoma : analysis of a rare entity based on a case report and literature review. Int J Surg Pathol 21: 514-519, 2012.
18) Nagao T, Sugano I, et al : Hybrid carcinomas of the salivary glands: report of nine cases with a clinicopathologic, immunohistochemical, and p53 gene alteration analysis. Mod Pthol 15: 724-733, 2002.
22) Manuel S, Mathews A, et al : Carcinosarcoma of the parotid gland with epithelial-myoepithelial carcinoma and pleomorphic sarcoma components. Br J Oral Maxillofac Surg 40: 480-483, 2002.
15) 小栗千里, 出田ゆか, 他:下顎歯肉扁平上皮癌治療後早期に肝門部リンパ節転移と腹膜播種性転移を認めた1例.口腔腫瘍 31: 97-103, 2019.
16) Jaehne M, Roeser K, et al : Clinical and immunohistologic typing of salivary duct carcinoma: a report of 50 cases. Cancer 103: 2526-2533, 2005.
25) Sarode VR, Truelson J, et al : Dedifferentiated epithelial-myoepithelial carcinoma of the parotid gland with aberrant expression of prostate specific antigen: a case report. Int J Surg Pathol 18: 401-405, 2010.
20) Alos L, Carrillo R, et al : High grade carcinoma component in epithelial-myoepithelial carcinoma of salivary glands clinicopathological, immunohistochemical and flow cytometric study of three cases. Virchows Arch 434: 291-299, 1999.
13) 岡田康男, 加藤譲治, 他:顎・口腔悪性腫瘍剖検症例の臨床病理学的研究.歯学 84: 255-274, 1996.
17) Roy P, Bullock MJ, et al : Epithelial-myoepithelial carcinoma with high grade transformation. Am J Surg Pathol 34: 1258-1265, 2010.
1) Donath K, Seifert G, et al : Diagnosis and ultrastructure of the tubular carcinoma of salivary gland ducts. Epithelial-myoepithelial carcinoma of the intercalated ducts. Virchows Arch A Pathol Pathol Anat 356: 16-31, 1972.
28) Aydin A, Taskin U, et al : Case study of a parotid gland adenocarcinoma dedifferentiated from epithelial-myoepithelial carcinoma. Case Rep Otolaryngol Sep 9, 2014.
8) Cheuk W and Chan JK : Advances in salivary gland pathology. Histopathology 51: 1-20, 2007.
24) Suzuki T, Murata S, et al : Epithelial myoepithelial carcinoma with myoepithelial anaplasia: report of a case with cytologic findings of a rare variant. Acta Cytol 54: 605-610, 2010.
5) Vazquez A, Patel T, et al : Epithelial-myoepithelial carcinoma of the salivary glamds: an analysis of 246 cases. Otolaryngology-Head and Neck Surgery 153: 569-574, 2015.
29) Li B, Yang H, et al : Epithelial-myoepithelial carcinoma with high-grade transformation of protid gland: A case report and literature review. Medicine December 96, 2017.
3) 原田博史, 大内知之:稀な唾液腺腫瘍の病理 近年新たに報告された腫瘍型を含めて.病理と臨床 29: 602-608, 2011.
10) Yang S and Chen X : Epithelial-myoepithelial carcinoma with high grade transformation. Int J Oral Maxillofac Surg 41: 810-813, 2012.
14) Kim YW, Lee BJ, et al : Solitary peritoneal lymph node metastasis of head and neck cancer diagnosed with FDG-PET/CT imaging. Ear Nose Throat J 93: E28-E30, 2014.
References_xml – reference: 24) Suzuki T, Murata S, et al : Epithelial myoepithelial carcinoma with myoepithelial anaplasia: report of a case with cytologic findings of a rare variant. Acta Cytol 54: 605-610, 2010.
– reference: 25) Sarode VR, Truelson J, et al : Dedifferentiated epithelial-myoepithelial carcinoma of the parotid gland with aberrant expression of prostate specific antigen: a case report. Int J Surg Pathol 18: 401-405, 2010.
– reference: 10) Yang S and Chen X : Epithelial-myoepithelial carcinoma with high grade transformation. Int J Oral Maxillofac Surg 41: 810-813, 2012.
– reference: 13) 岡田康男, 加藤譲治, 他:顎・口腔悪性腫瘍剖検症例の臨床病理学的研究.歯学 84: 255-274, 1996.
– reference: 9) 原田博史:唾液腺癌におけるいわゆる“脱分化”について.病理と臨床 27: 486-487, 2009.
– reference: 26) 関 伸彦, 山崎徳和, 他:耳下腺に発生した脱分化型上皮筋上皮癌の1例.耳喉頭頸 83: 630-633, 2011.
– reference: 8) Cheuk W and Chan JK : Advances in salivary gland pathology. Histopathology 51: 1-20, 2007.
– reference: 12) Baker AR, Ohanessian SE, et al : Dedifferentiated epithelial-myoepithelial carcinoma : analysis of a rare entity based on a case report and literature review. Int J Surg Pathol 21: 514-519, 2012.
– reference: 1) Donath K, Seifert G, et al : Diagnosis and ultrastructure of the tubular carcinoma of salivary gland ducts. Epithelial-myoepithelial carcinoma of the intercalated ducts. Virchows Arch A Pathol Pathol Anat 356: 16-31, 1972.
– reference: 22) Manuel S, Mathews A, et al : Carcinosarcoma of the parotid gland with epithelial-myoepithelial carcinoma and pleomorphic sarcoma components. Br J Oral Maxillofac Surg 40: 480-483, 2002.
– reference: 19) Simpson RH, Clarke TJ, et al : Epithelial-myoepithelial carcinoma of salivary glands. J Clin Pathol 44: 419-423, 1991.
– reference: 21) Fonseca I, Felix A, et al : Dedifferentiation in salivary glamd carcinomas. Am J Surg Pathol 24: 469-471, 2000.
– reference: 27) Lima FJ, Port DE, et al : Epithelial-myoepithelial carcinoma of high grade transformation: the case report in the buccal mucosa. Open Dent J 6: 111-117, 2012.
– reference: 28) Aydin A, Taskin U, et al : Case study of a parotid gland adenocarcinoma dedifferentiated from epithelial-myoepithelial carcinoma. Case Rep Otolaryngol Sep 9, 2014.
– reference: 20) Alos L, Carrillo R, et al : High grade carcinoma component in epithelial-myoepithelial carcinoma of salivary glands clinicopathological, immunohistochemical and flow cytometric study of three cases. Virchows Arch 434: 291-299, 1999.
– reference: 14) Kim YW, Lee BJ, et al : Solitary peritoneal lymph node metastasis of head and neck cancer diagnosed with FDG-PET/CT imaging. Ear Nose Throat J 93: E28-E30, 2014.
– reference: 7) Seethala RR, Barnes EL, et al : Epithelial-myoepithelial carcinoma : a review of the clinicopathologic spectrum and immunophenotypic characteristics in 61 tumors of the salivary glands and upper aero digestive tract. Am J Surg Pathol 31: 44-57, 2007.
– reference: 4) Nagao T : “Dedifferentiation” and high-grade transformation in salivary gland carcinomas. Head and Neck Pathol 7: 37-47, 2013.
– reference: 5) Vazquez A, Patel T, et al : Epithelial-myoepithelial carcinoma of the salivary glamds: an analysis of 246 cases. Otolaryngology-Head and Neck Surgery 153: 569-574, 2015.
– reference: 29) Li B, Yang H, et al : Epithelial-myoepithelial carcinoma with high-grade transformation of protid gland: A case report and literature review. Medicine December 96, 2017.
– reference: 16) Jaehne M, Roeser K, et al : Clinical and immunohistologic typing of salivary duct carcinoma: a report of 50 cases. Cancer 103: 2526-2533, 2005.
– reference: 17) Roy P, Bullock MJ, et al : Epithelial-myoepithelial carcinoma with high grade transformation. Am J Surg Pathol 34: 1258-1265, 2010.
– reference: 23) Niederhagen M, Zengel P, et al : Secondary high-malignant transformation of a low-malignant epithelial-myoepithelial carcinoma. Pathologe 30: 461-465, 2009.
– reference: 6) 柴田章夫, 木村将士, 他:前舌腺由来と考えられた腺様囊胞癌の1例.口腔腫瘍 28: 15-19, 2016.
– reference: 11) Kusafuka K, Takizawa Y, et al : Dedifferentiated epithelial-myoepithelial carcinoma of the parotid gland : a case report of immunohistochemical analysis and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 106: 85-91, 2008.
– reference: 15) 小栗千里, 出田ゆか, 他:下顎歯肉扁平上皮癌治療後早期に肝門部リンパ節転移と腹膜播種性転移を認めた1例.口腔腫瘍 31: 97-103, 2019.
– reference: 2) Fonseca I and Soares J : Epithelial-myoepithelial carcinoma. In Barnes L, Eveson JW, eds ; WHO Classification of Tumours. Pathology and Genetics of Head and Neck Tumours. IARC Press, Lyon, 2005, p225-226.
– reference: 18) Nagao T, Sugano I, et al : Hybrid carcinomas of the salivary glands: report of nine cases with a clinicopathologic, immunohistochemical, and p53 gene alteration analysis. Mod Pthol 15: 724-733, 2002.
– reference: 3) 原田博史, 大内知之:稀な唾液腺腫瘍の病理 近年新たに報告された腫瘍型を含めて.病理と臨床 29: 602-608, 2011.
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SubjectTerms 上皮筋上皮癌
前舌腺
脱分化
Title 前舌腺由来と考えられた脱分化型上皮筋上皮癌の1例
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