上唇に発生した多形腺腫由来癌の1例

Carcinoma ex pleomorphic adenoma is a rare malignant salivary gland tumor. It occurs more commonly in the parotid gland and rarely develops in the upper lip. We report a case of carcinoma ex pleomorphic adenoma arising in the left upper lip in a 63-year-old woman. The patient visited our hospital wi...

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Published in日本口腔外科学会雑誌 Vol. 68; no. 5; pp. 226 - 232
Main Authors 濱田, 良樹, 新井, 剛, 馬杉, 亮彦, 長谷部, 充彦, 重松, 宏昭, 宮田, 幸長
Format Journal Article
LanguageJapanese
Published 公益社団法人 日本口腔外科学会 20.05.2022
Subjects
上唇
多形腺腫
多形腺腫由来癌
小唾液腺
Online AccessGet full text
ISSN0021-5163
2186-1579
DOI10.5794/jjoms.68.226

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Abstract Carcinoma ex pleomorphic adenoma is a rare malignant salivary gland tumor. It occurs more commonly in the parotid gland and rarely develops in the upper lip. We report a case of carcinoma ex pleomorphic adenoma arising in the left upper lip in a 63-year-old woman. The patient visited our hospital with the chief complaint of left upper lip swelling, which had been present for 10 years. In the clinical examination, an elastic-hard, painless, movable 25 × 15 mm mass was palpated in the left upper lip. MRI revealed a mass lesion with well-defined borders in the left upper lip area. T1-weighted images indicated moderate intensity, whereas fat-suppressed T2-weighted images showed high intensity. An incisional biopsy was performed under local anesthesia, and the initial histopathological diagnosis was pleomorphic adenoma. The tumor was subsequently removed under general anesthesia. Histological examination of the resected specimen led to a diagnosis of noninvasive carcinoma ex pleomorphic adenoma with a malignant component of salivary duct carcinoma. No sign of recurrence or metastasis has been observed in the two years and two months after surgery.
AbstractList Carcinoma ex pleomorphic adenoma is a rare malignant salivary gland tumor. It occurs more commonly in the parotid gland and rarely develops in the upper lip. We report a case of carcinoma ex pleomorphic adenoma arising in the left upper lip in a 63-year-old woman. The patient visited our hospital with the chief complaint of left upper lip swelling, which had been present for 10 years. In the clinical examination, an elastic-hard, painless, movable 25 × 15 mm mass was palpated in the left upper lip. MRI revealed a mass lesion with well-defined borders in the left upper lip area. T1-weighted images indicated moderate intensity, whereas fat-suppressed T2-weighted images showed high intensity. An incisional biopsy was performed under local anesthesia, and the initial histopathological diagnosis was pleomorphic adenoma. The tumor was subsequently removed under general anesthesia. Histological examination of the resected specimen led to a diagnosis of noninvasive carcinoma ex pleomorphic adenoma with a malignant component of salivary duct carcinoma. No sign of recurrence or metastasis has been observed in the two years and two months after surgery.
Author 馬杉, 亮彦
宮田, 幸長
重松, 宏昭
長谷部, 充彦
新井, 剛
濱田, 良樹
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References 32) Chen AM, Garcia J, et al : The role of postoperative radiation therapy in carcinoma ex pleomorphic adenoma of the parotid gland. Int J Radiat Oncol Biol Phys 67: 138-143, 2007.
27) 橋本和樹， 山元英崇， 他：唾液腺多形腺腫由来癌における病理組織学的進展度と予後との関連−現行T分類との比較検討−．頭頸部癌 38: 50-55, 2012.
7) Mitate E, Kawano S, et al : Carcinoma ex pleomorphic adenoma of the upper lip: a case of an unusual malignant component of squamous cell carcinoma. World J Surg Oncol. 231: 11, 2013. Available at https://doi.org/10.1186/1477-7819-11-234. Accessed Feb 23, 2021.
40) Gupta A, Koochakzadeh S, et al : Carcinoma ex pleomorphic adenoma: A review of incidence, demographics, risk factors, and survival. Am J Otolaryngol 40: 6, 2019.
12) Juan R : Surgical Pathology. 9th Ed, Mosby Co, Milan, 2004, p894-895.
29) 沼田　勉， 渋谷真理子， 他：耳下腺の多形腺腫内癌．JOHNS 22: 1067-1070, 2006.
37) Cheuk W and Chan JK : Advances in salivary gland pathology. Histopathology 51: 1-20, 2007.
15) Jaehne M, Roeser K, et al : Clinical and immunohistologic typing of salivary duct carcinoma: a report of 50 cases. Cancer 103: 2526-2533, 2005.
9) Seifert G : Histopathology of malignant salivary gland tumors. Eur J Cancer B Oral Oncol 28: 49-56, 1992.
1) Olsen KD, Lewis JE, et al : Carcinoma ex pleomorphic adenoma: a clinicopathologic review. Head Neck 23: 705-712, 2001.
8) Mariano FV, Rincon D, et al : Carcinoma ex-pleomorphic adenoma of upper lip showing copy number loss of tumor suppressor genes. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 116: 69-74, 2013.
34) 藤田芳史， 久保田彰， 他：顔面神経温存を優先した耳下腺癌治療の検討．日耳咽 113: 115-122, 2010.
6) Dyalram D, Huebner T, et al : Carcinoma ex pleomorphic adenoma of the upper lip. Int J Oral Maxillofac Surg 41: 364-367, 2012.
2) Ellis GL and Auclair PL : AFIP Atlas of Tumor Pathology: Tumors of the Salivary Glands. American Registry of Pathology, Washington, DC, 2008, p259-269.
36) Arihara Y, Murase K, et al : Trastuzumab-Based Combination Chemotherapy in Patients with Human Epidermal Growth Factor Receptor-2-Positive Metastatic Carcinoma ex Pleomorphic Adenoma. Case Rep Oncol 11: 835-841, 2018.
17) Luk PP, Weston JD, et al : Salivary duct carcinoma: clinicopathologic features, morphologic spectrum, and somatic mutations. Head Neck 38: E1838-1847, 2016.
41) Lewis JE, Olsen KD et al, et al : Carcinoma ex pleomorphic adenoma: pathologic analysis of 73 cases. Hum Pathol 32: 596-604, 2001.
19) Locati LD, Perrone F, et al : Treatment relevant target immunophenotyping of 139 salivary gland carcinomas (SGCs). Oral Oncol 45: 986-990, 2009.
31) 信澤愛子， 谷口明慧， 他：顎下腺に発生した多形腺腫由来癌の1例．日口診誌 30: 42-46, 2017.
43) Olsen KD and Lewis JE : Carcinoma ex pleomorphic adenoma: a clinicopathologic review. Head Neck 11: 705-712, 2001.
3) Gnepp DR, Brandwein-Gensler MS, et al : Carcinoma ex pleomorphic adenoma. In Barnes L, Eveson JW, eds ; WHO classification of Tumours, Pathology and Genetics of Head and Neck Tumours. IARC Press, Lyon, 2005, p242-243.
26) Matsuzaki H, Yanagi Y, et al : Minor salivary gland tumors in the oral cavity: diagnostic value of dynamic contrast-enhanced MRI. Eur J Radiol 81: 2684-2691, 2012.
10) 長尾俊孝：唾液腺腫瘍の組織分類と病理診断．口腔咽頭科 19: 143-152, 2007.
22) 丸川浩平， 小峰梨果， 他：小唾液腺由来と考えられる唾液腺癌の3例．日口診誌 30: 263-268, 2017.
14) Skálová A, Stárek I, et al : Expression of HER-2/neu gene and protein in salivary duct carcinomas of parotid gland as revealed by fluorescence in-situ hybridization and immunohistochemistry. Histopathology 42: 348-358, 2003.
42) Katabi N, Gomez D, et al : Prognostic factors of recurrence in salivary carcinoma ex pleomorphic adenoma, with emphasis on the carcinoma histologic subtype: a clinicopathologic study of 43 cases. Hum Pathol 11: 927-934, 2010.
18) Takase S, Kano S, et al : Biomarker immunoprofile in salivary duct carcinomas: clinicopathological and prognostic implications with evaluation of the revised classification. Oncotarget 29: 59023-59035, 2017.
23) 高野智誠， 安原一夫， 他： 耳下腺腫瘍におけるdynamic studyの有用性の検討．頭頸部外 30: 199-203, 2020.
25) 鳴瀬智史， 柳本惣市， 他：生検では癌腫が認められなかった多形腺腫由来癌の1例．日口診誌 29: 60-67, 2016.
35) 佐々木徹， 川端一嘉， 他：顎下腺癌の治療. 頭頸部癌 39: 281-286, 2013.
38) Ye P, Gao Y, et al : Carcinoma Ex Pleomorphic Adenoma: Is It a High-Grade Malignancy? J Oral Maxillofac Surg 74: 2093-2104, 2016.
3) Johnson C, Barry M, et al : Her-2/neu expression in salivary duct carcinoma: an immunohistochemical and chromogenic in situ hybridization study. Appl Immunohistochem Mol Morphol 16: 54-58, 2008.
33) 本多伸光， 小林泰輔， 他：頸部リンパ節転移により判明した軟口蓋多形腺腫由来癌例．耳鼻臨床 103: 737-742, 2010.
39) Zhao J, Wang J, et al : Prognostic factors affecting the clinical outcome of carcinoma ex pleomorphic adenoma in the major salivary gland. World J Surg Oncol 11 : 180, 2013. Available at https://doi.org/10.1186/1477-7819-11-180. Accessed May 30, 2021.
20) Martinez-Barba E, Cortes-Guardiola JA, et al : Salivary duct carcinoma: clinicopathological and immunohistochemical studies.J Craniomaxillofac Surg 25: 328-334, 1997.
4) Yong DJ, Mazlinada M, et al : Invasive carcinoma ex pleomorphic adenoma of submandibular gland: A case report. Asian J Oral Maxillofac Surg 23: 207-209, 2011.
21) 小村　健：唾液腺腫瘍の外科療法．口腔腫瘍 23: 73-81, 2011.
30) 大竹史浩， 田村隆行， 他：頰部に発生した多形腺腫由来癌の1例．口腔腫瘍 25: 207-212, 2013.
11) Lewis JE, McKinney BC, et al : Salivary duct carcinoma. Clinicopathologic and immunohistochemical review of 26 cases. Cancer 77: 223-230, 1996.
44) 木下浩二， 安田真也， 他：顎下部に発生した多形腺腫由来癌の1例．日口外誌 55: 424-427, 2009.
16) Masubuchi T, Tada Y, et al : Clinicopathological significance of androgen receptor, HER2, Ki-67 and EGFR expressions in salivary duct carcinoma. Int J Clin Oncol 20: 35-44, 2015.
24) 若盛　隼， 小川武則， 他：MRI で悪性腫瘍箇所を同定し得た耳下腺多形腺腫由来癌の1例．頭頸部外 26: 389-393, 2017.
5) McNamara ZJ, Batstone M, et al : Carcinoma ex pleomorphic adenoma in a minor salivary gland of the upper lip. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 108: 51-53, 2009.
28) 沼田　勉， 昼間　清， 他：悪性混合腫瘍（多形腺腫内癌）．癌と化療 31: 314-317, 2004.
References_xml – reference: 3) Gnepp DR, Brandwein-Gensler MS, et al : Carcinoma ex pleomorphic adenoma. In Barnes L, Eveson JW, eds ; WHO classification of Tumours, Pathology and Genetics of Head and Neck Tumours. IARC Press, Lyon, 2005, p242-243.
– reference: 19) Locati LD, Perrone F, et al : Treatment relevant target immunophenotyping of 139 salivary gland carcinomas (SGCs). Oral Oncol 45: 986-990, 2009.
– reference: 21) 小村　健：唾液腺腫瘍の外科療法．口腔腫瘍 23: 73-81, 2011.
– reference: 38) Ye P, Gao Y, et al : Carcinoma Ex Pleomorphic Adenoma: Is It a High-Grade Malignancy? J Oral Maxillofac Surg 74: 2093-2104, 2016.
– reference: 23) 高野智誠， 安原一夫， 他： 耳下腺腫瘍におけるdynamic studyの有用性の検討．頭頸部外 30: 199-203, 2020.
– reference: 28) 沼田　勉， 昼間　清， 他：悪性混合腫瘍（多形腺腫内癌）．癌と化療 31: 314-317, 2004.
– reference: 39) Zhao J, Wang J, et al : Prognostic factors affecting the clinical outcome of carcinoma ex pleomorphic adenoma in the major salivary gland. World J Surg Oncol 11 : 180, 2013. Available at https://doi.org/10.1186/1477-7819-11-180. Accessed May 30, 2021.
– reference: 22) 丸川浩平， 小峰梨果， 他：小唾液腺由来と考えられる唾液腺癌の3例．日口診誌 30: 263-268, 2017.
– reference: 41) Lewis JE, Olsen KD et al, et al : Carcinoma ex pleomorphic adenoma: pathologic analysis of 73 cases. Hum Pathol 32: 596-604, 2001.
– reference: 35) 佐々木徹， 川端一嘉， 他：顎下腺癌の治療. 頭頸部癌 39: 281-286, 2013.
– reference: 29) 沼田　勉， 渋谷真理子， 他：耳下腺の多形腺腫内癌．JOHNS 22: 1067-1070, 2006.
– reference: 42) Katabi N, Gomez D, et al : Prognostic factors of recurrence in salivary carcinoma ex pleomorphic adenoma, with emphasis on the carcinoma histologic subtype: a clinicopathologic study of 43 cases. Hum Pathol 11: 927-934, 2010.
– reference: 3) Johnson C, Barry M, et al : Her-2/neu expression in salivary duct carcinoma: an immunohistochemical and chromogenic in situ hybridization study. Appl Immunohistochem Mol Morphol 16: 54-58, 2008.
– reference: 15) 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: 16) Masubuchi T, Tada Y, et al : Clinicopathological significance of androgen receptor, HER2, Ki-67 and EGFR expressions in salivary duct carcinoma. Int J Clin Oncol 20: 35-44, 2015.
– reference: 2) Ellis GL and Auclair PL : AFIP Atlas of Tumor Pathology: Tumors of the Salivary Glands. American Registry of Pathology, Washington, DC, 2008, p259-269.
– reference: 33) 本多伸光， 小林泰輔， 他：頸部リンパ節転移により判明した軟口蓋多形腺腫由来癌例．耳鼻臨床 103: 737-742, 2010.
– reference: 44) 木下浩二， 安田真也， 他：顎下部に発生した多形腺腫由来癌の1例．日口外誌 55: 424-427, 2009.
– reference: 31) 信澤愛子， 谷口明慧， 他：顎下腺に発生した多形腺腫由来癌の1例．日口診誌 30: 42-46, 2017.
– reference: 18) Takase S, Kano S, et al : Biomarker immunoprofile in salivary duct carcinomas: clinicopathological and prognostic implications with evaluation of the revised classification. Oncotarget 29: 59023-59035, 2017.
– reference: 25) 鳴瀬智史， 柳本惣市， 他：生検では癌腫が認められなかった多形腺腫由来癌の1例．日口診誌 29: 60-67, 2016.
– reference: 1) Olsen KD, Lewis JE, et al : Carcinoma ex pleomorphic adenoma: a clinicopathologic review. Head Neck 23: 705-712, 2001.
– reference: 5) McNamara ZJ, Batstone M, et al : Carcinoma ex pleomorphic adenoma in a minor salivary gland of the upper lip. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 108: 51-53, 2009.
– reference: 40) Gupta A, Koochakzadeh S, et al : Carcinoma ex pleomorphic adenoma: A review of incidence, demographics, risk factors, and survival. Am J Otolaryngol 40: 6, 2019.
– reference: 4) Yong DJ, Mazlinada M, et al : Invasive carcinoma ex pleomorphic adenoma of submandibular gland: A case report. Asian J Oral Maxillofac Surg 23: 207-209, 2011.
– reference: 9) Seifert G : Histopathology of malignant salivary gland tumors. Eur J Cancer B Oral Oncol 28: 49-56, 1992.
– reference: 10) 長尾俊孝：唾液腺腫瘍の組織分類と病理診断．口腔咽頭科 19: 143-152, 2007.
– reference: 12) Juan R : Surgical Pathology. 9th Ed, Mosby Co, Milan, 2004, p894-895.
– reference: 17) Luk PP, Weston JD, et al : Salivary duct carcinoma: clinicopathologic features, morphologic spectrum, and somatic mutations. Head Neck 38: E1838-1847, 2016.
– reference: 6) Dyalram D, Huebner T, et al : Carcinoma ex pleomorphic adenoma of the upper lip. Int J Oral Maxillofac Surg 41: 364-367, 2012.
– reference: 43) Olsen KD and Lewis JE : Carcinoma ex pleomorphic adenoma: a clinicopathologic review. Head Neck 11: 705-712, 2001.
– reference: 36) Arihara Y, Murase K, et al : Trastuzumab-Based Combination Chemotherapy in Patients with Human Epidermal Growth Factor Receptor-2-Positive Metastatic Carcinoma ex Pleomorphic Adenoma. Case Rep Oncol 11: 835-841, 2018.
– reference: 37) Cheuk W and Chan JK : Advances in salivary gland pathology. Histopathology 51: 1-20, 2007.
– reference: 24) 若盛　隼， 小川武則， 他：MRI で悪性腫瘍箇所を同定し得た耳下腺多形腺腫由来癌の1例．頭頸部外 26: 389-393, 2017.
– reference: 7) Mitate E, Kawano S, et al : Carcinoma ex pleomorphic adenoma of the upper lip: a case of an unusual malignant component of squamous cell carcinoma. World J Surg Oncol. 231: 11, 2013. Available at https://doi.org/10.1186/1477-7819-11-234. Accessed Feb 23, 2021.
– reference: 32) Chen AM, Garcia J, et al : The role of postoperative radiation therapy in carcinoma ex pleomorphic adenoma of the parotid gland. Int J Radiat Oncol Biol Phys 67: 138-143, 2007.
– reference: 11) Lewis JE, McKinney BC, et al : Salivary duct carcinoma. Clinicopathologic and immunohistochemical review of 26 cases. Cancer 77: 223-230, 1996.
– reference: 30) 大竹史浩， 田村隆行， 他：頰部に発生した多形腺腫由来癌の1例．口腔腫瘍 25: 207-212, 2013.
– reference: 20) Martinez-Barba E, Cortes-Guardiola JA, et al : Salivary duct carcinoma: clinicopathological and immunohistochemical studies.J Craniomaxillofac Surg 25: 328-334, 1997.
– reference: 27) 橋本和樹， 山元英崇， 他：唾液腺多形腺腫由来癌における病理組織学的進展度と予後との関連−現行T分類との比較検討−．頭頸部癌 38: 50-55, 2012.
– reference: 34) 藤田芳史， 久保田彰， 他：顔面神経温存を優先した耳下腺癌治療の検討．日耳咽 113: 115-122, 2010.
– reference: 26) Matsuzaki H, Yanagi Y, et al : Minor salivary gland tumors in the oral cavity: diagnostic value of dynamic contrast-enhanced MRI. Eur J Radiol 81: 2684-2691, 2012.
– reference: 8) Mariano FV, Rincon D, et al : Carcinoma ex-pleomorphic adenoma of upper lip showing copy number loss of tumor suppressor genes. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 116: 69-74, 2013.
– reference: 14) Skálová A, Stárek I, et al : Expression of HER-2/neu gene and protein in salivary duct carcinomas of parotid gland as revealed by fluorescence in-situ hybridization and immunohistochemistry. Histopathology 42: 348-358, 2003.
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多形腺腫
多形腺腫由来癌
小唾液腺
Title 上唇に発生した多形腺腫由来癌の1例
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