血清CEAとCA19-9異常高値にて発見された臼後部に生じたIntraductal carcinomaの1例

Intraductal carcinoma (IDC) is a rare salivary gland tumor, classified according to the 4th WHO Classification of Head and Neck Tumors in 2017. The tumor affects the parotid gland much more frequently than the minor salivary glands. It’s often asymptomatic. We report a case of IDC of the left retrom...

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Published in日本口腔外科学会雑誌 Vol. 67; no. 5; pp. 292 - 296
Main Authors 上田, 順宏, 桐田, 忠昭, 山川, 延宏, 有川, 翔, 河上, 真緒, 髙橋, 佑佳
Format Journal Article
LanguageJapanese
Published 社団法人 日本口腔外科学会 20.05.2021
Subjects
CA19-9
CEA
導管内癌
小唾液腺
腫瘍マーカー
Online AccessGet full text
ISSN0021-5163
2186-1579
DOI10.5794/jjoms.67.292

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Abstract Intraductal carcinoma (IDC) is a rare salivary gland tumor, classified according to the 4th WHO Classification of Head and Neck Tumors in 2017. The tumor affects the parotid gland much more frequently than the minor salivary glands. It’s often asymptomatic. We report a case of IDC of the left retromolar trigon occurring in an 81-year-old woman. She was diagnosed with high serum CEA and CA19-9 at a medical examination. Examinations of her gastrointestinal tract, lungs, and mammary glands were conducted, resulting in no abnormal findings. FDG-PET showed abnormal uptake only in the left retromolar trigon, and a painless mass was observed. A biopsy of this tumor indicated it was likely a low-grade salivary gland tumor. The tumor was completely removed, along with sufficient margins. Histological examination revealed that the lesion was composed of alveolar nests showing an expanded duct-like structure surrounded by an intact myoepithelial layer, with no evidence of infiltration. The final diagnosis was IDC. The postoperative course was uneventful and the patient’s serum CEA and CA19-9 levels decreased to within normal range. We believe that serum CEA and CA19-9 levels could be used for the diagnosis and evaluation of therapeutic effect for this tumor.
AbstractList Intraductal carcinoma (IDC) is a rare salivary gland tumor, classified according to the 4th WHO Classification of Head and Neck Tumors in 2017. The tumor affects the parotid gland much more frequently than the minor salivary glands. It’s often asymptomatic. We report a case of IDC of the left retromolar trigon occurring in an 81-year-old woman. She was diagnosed with high serum CEA and CA19-9 at a medical examination. Examinations of her gastrointestinal tract, lungs, and mammary glands were conducted, resulting in no abnormal findings. FDG-PET showed abnormal uptake only in the left retromolar trigon, and a painless mass was observed. A biopsy of this tumor indicated it was likely a low-grade salivary gland tumor. The tumor was completely removed, along with sufficient margins. Histological examination revealed that the lesion was composed of alveolar nests showing an expanded duct-like structure surrounded by an intact myoepithelial layer, with no evidence of infiltration. The final diagnosis was IDC. The postoperative course was uneventful and the patient’s serum CEA and CA19-9 levels decreased to within normal range. We believe that serum CEA and CA19-9 levels could be used for the diagnosis and evaluation of therapeutic effect for this tumor.
Author 山川, 延宏
上田, 順宏
有川, 翔
髙橋, 佑佳
桐田, 忠昭
河上, 真緒
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References 13) Palicelli A : Intraductal carcinomas of the salivary glands : systematic review and classification of 93 published cases. APMIS 128: 191-200, 2020.
8) Cheuk W, Miliauskas JR, et al : Intraductal carcinoma of the oral cavity : A case report and a reappraisal of the concept of pure ductal carcinoma in situ in salivary duct carcinoma. Am J Surg Pathol 28: 266-270, 2004.
12) Kimura M, Mii S, et al : Low-grade cribriform cystadenocarcinoma arising from a minor salivary gland : a case report. J Oral Sci 58: 145-149, 2016.
17) Lu H, Graham RP, et al : Intraductal carcinoma of salivary glands harboring TRIM27-RET fusion with mixed low grade and apocrine types. Head Neck Pathol 14: 239-245, 2020.
21) 渡邊昭仁， 片山昭公：高CEA血症で発見された舌下腺粘表皮腫瘍例．耳鼻臨床 91: 281-284, 1998.
11) Stevens TM, Kovalovsky AO, et al : Mammary analog secretory carcinoma, low-grade salivary duct carcinoma, and mimickers : a comparative study. Mod Pathol 28: 1084-1100, 2015.
22) 岸本麻子， 堤　俊之， 他：CEAが高値を示した耳下腺papillary cystadenocarcinoma例． 耳鼻臨床 45: 341-345, 1999.
2) El-Naggar AK, Chan JKC, et al : WHO Classification of Head and Neck Tumors. 4th ed, IARC Press, Lyon, 2017, p170-171.
3) Nojima J, Nakahira M, et al : An extremely rare salivary gland tumor : intraductal carcinoma of the buccal mucosa. Int Cancer Conf J 5: 53-56, 2016.
7) Tatemoto Y, Ohno A, et al : Low malignant intraductal carcinoma on the hard palate : a variant of salivary duct carcinoma? Eur J Cancer B Oral Oncol 32: 275-277, 1996.
4) Skálová A, Ptáková N, et al : NCOA4-RET and TRIM27-RET are characteristic gene fusions in salivary intraductal carcinoma, including invasive and metastatic tumors : is“ intraductal” correct? Am J Surg Pathol 43: 1303-1313, 2019.
18) Anderson C, Muller R, et al : Intraductal carcinoma of major salivary gland. Cancer 69: 609-614, 1992.
9) Ide F, Mishima K, et al : Circumscribed salivary duct carcinoma of the palate : a non-threatening variant. Histopathology 45: 89-91, 2004.
14) Justin AB, Jeffrey G, et al : Low-grade Apocrine Intraductal Carcinoma : Expanding the Morphologic and Molecular Spectrum of an Enigmatic Salivary Gland Tumor. Head Neck Pathol 14: 869-875, 2020.
20) Kuhel WI, Chow H, et al : Elevated carcinoembryonic antigen levels correlating with disease recurrence in patient with adenoid carcinoma. Head Neck 17: 431-436, 1995.
23) 篠原尚吾， 八木伸也， 他：CA19-9が高値を示した耳下腺腫瘍例．耳鼻臨床 87: 365-369, 1994.
10) Kokabu S, Nojima J, et al : Low-grade cribriform cystadenocarcinoma of the palatal gland : A case report. Oncol Lett 10: 2453-2457, 2015.
1) Seethala RR and Stenman G : Update from the 4th edition of the world health organization classification of Head and neck tumours : tumors of the salivary gland. Head Neck Pathol 11: 55-67, 2017.
15) Sheng CL, Ren TK, et al : Intraductal Carcinoma of Salivary Gland Originating from an Intraparotid Lymph Node : A Case Report. Malays J Pathol 41: 207-211, 2019.
19) Roh JL, Ryu CH, et al : Clinical utility of 18F-FDG PET for patients with salivary gland malignancies. J Nucl Med 48: 240-246, 2007.
6) Watatani K, Shirasuna K, et al : Intraductal carcinoma of the tongue : report of a case. Int J Oral Maxillofac Surg 20: 175-176, 1991.
16) Jeong JY, Ahn D, et al : Fine-needle aspiration cytology of low-grade cribriform cystadenocarcinoma with many psammoma bodies of the salivary gland. Korean J Pathol 47: 481-485, 2013.
5) Chen KT : Intraductal carcinoma of the minor salivary gland. J Laryngol Otol 97: 189-191, 1983.
References_xml – reference: 9) Ide F, Mishima K, et al : Circumscribed salivary duct carcinoma of the palate : a non-threatening variant. Histopathology 45: 89-91, 2004.
– reference: 14) Justin AB, Jeffrey G, et al : Low-grade Apocrine Intraductal Carcinoma : Expanding the Morphologic and Molecular Spectrum of an Enigmatic Salivary Gland Tumor. Head Neck Pathol 14: 869-875, 2020.
– reference: 17) Lu H, Graham RP, et al : Intraductal carcinoma of salivary glands harboring TRIM27-RET fusion with mixed low grade and apocrine types. Head Neck Pathol 14: 239-245, 2020.
– reference: 19) Roh JL, Ryu CH, et al : Clinical utility of 18F-FDG PET for patients with salivary gland malignancies. J Nucl Med 48: 240-246, 2007.
– reference: 23) 篠原尚吾， 八木伸也， 他：CA19-9が高値を示した耳下腺腫瘍例．耳鼻臨床 87: 365-369, 1994.
– reference: 16) Jeong JY, Ahn D, et al : Fine-needle aspiration cytology of low-grade cribriform cystadenocarcinoma with many psammoma bodies of the salivary gland. Korean J Pathol 47: 481-485, 2013.
– reference: 8) Cheuk W, Miliauskas JR, et al : Intraductal carcinoma of the oral cavity : A case report and a reappraisal of the concept of pure ductal carcinoma in situ in salivary duct carcinoma. Am J Surg Pathol 28: 266-270, 2004.
– reference: 15) Sheng CL, Ren TK, et al : Intraductal Carcinoma of Salivary Gland Originating from an Intraparotid Lymph Node : A Case Report. Malays J Pathol 41: 207-211, 2019.
– reference: 21) 渡邊昭仁， 片山昭公：高CEA血症で発見された舌下腺粘表皮腫瘍例．耳鼻臨床 91: 281-284, 1998.
– reference: 12) Kimura M, Mii S, et al : Low-grade cribriform cystadenocarcinoma arising from a minor salivary gland : a case report. J Oral Sci 58: 145-149, 2016.
– reference: 6) Watatani K, Shirasuna K, et al : Intraductal carcinoma of the tongue : report of a case. Int J Oral Maxillofac Surg 20: 175-176, 1991.
– reference: 2) El-Naggar AK, Chan JKC, et al : WHO Classification of Head and Neck Tumors. 4th ed, IARC Press, Lyon, 2017, p170-171.
– reference: 3) Nojima J, Nakahira M, et al : An extremely rare salivary gland tumor : intraductal carcinoma of the buccal mucosa. Int Cancer Conf J 5: 53-56, 2016.
– reference: 7) Tatemoto Y, Ohno A, et al : Low malignant intraductal carcinoma on the hard palate : a variant of salivary duct carcinoma? Eur J Cancer B Oral Oncol 32: 275-277, 1996.
– reference: 22) 岸本麻子， 堤　俊之， 他：CEAが高値を示した耳下腺papillary cystadenocarcinoma例． 耳鼻臨床 45: 341-345, 1999.
– reference: 10) Kokabu S, Nojima J, et al : Low-grade cribriform cystadenocarcinoma of the palatal gland : A case report. Oncol Lett 10: 2453-2457, 2015.
– reference: 5) Chen KT : Intraductal carcinoma of the minor salivary gland. J Laryngol Otol 97: 189-191, 1983.
– reference: 18) Anderson C, Muller R, et al : Intraductal carcinoma of major salivary gland. Cancer 69: 609-614, 1992.
– reference: 11) Stevens TM, Kovalovsky AO, et al : Mammary analog secretory carcinoma, low-grade salivary duct carcinoma, and mimickers : a comparative study. Mod Pathol 28: 1084-1100, 2015.
– reference: 4) Skálová A, Ptáková N, et al : NCOA4-RET and TRIM27-RET are characteristic gene fusions in salivary intraductal carcinoma, including invasive and metastatic tumors : is“ intraductal” correct? Am J Surg Pathol 43: 1303-1313, 2019.
– reference: 13) Palicelli A : Intraductal carcinomas of the salivary glands : systematic review and classification of 93 published cases. APMIS 128: 191-200, 2020.
– reference: 1) Seethala RR and Stenman G : Update from the 4th edition of the world health organization classification of Head and neck tumours : tumors of the salivary gland. Head Neck Pathol 11: 55-67, 2017.
– reference: 20) Kuhel WI, Chow H, et al : Elevated carcinoembryonic antigen levels correlating with disease recurrence in patient with adenoid carcinoma. Head Neck 17: 431-436, 1995.
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Snippet Intraductal carcinoma (IDC) is a rare salivary gland tumor, classified according to the 4th WHO Classification of Head and Neck Tumors in 2017. The tumor...
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SubjectTerms CA19-9
CEA
導管内癌
小唾液腺
腫瘍マーカー
Title 血清CEAとCA19-9異常高値にて発見された臼後部に生じたIntraductal carcinomaの1例
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