A Case of Solid-type Adenoid Cystic Carcinoma in the Upper Trachea Found by Diplopia
Background. Adenoid cystic carcinoma (ACC) of the trachea is rare. ACCs are categorized into cribriform, tubular and solid types, and solid type is known to have the poorest prognosis. Case. A 57-year-old Japanese man who had suffered from diplopia for 2 months visited a hospital. Head magnetic reso...
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| Published in | The Journal of the Japan Society for Respiratory Endoscopy Vol. 41; no. 5; pp. 479 - 484 |
|---|---|
| Main Authors | , , , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
The Japan Society for Respiratory Endoscopy
25.09.2019
特定非営利活動法人 日本呼吸器内視鏡学会 |
| Subjects | |
| Online Access | Get full text |
| ISSN | 0287-2137 2186-0149 |
| DOI | 10.18907/jjsre.41.5_479 |
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| Abstract | Background. Adenoid cystic carcinoma (ACC) of the trachea is rare. ACCs are categorized into cribriform, tubular and solid types, and solid type is known to have the poorest prognosis. Case. A 57-year-old Japanese man who had suffered from diplopia for 2 months visited a hospital. Head magnetic resonance imaging (MRI) showed a right intraorbital tumor and multiple brain tumors, and chest computed tomography (CT) revealed multiple bilateral pulmonary nodules. He was introduced to our hospital for further examinations, and chest CT at the first visit showed a thickened tracheal wall in the right upper ventral trachea and multiple bilateral pulmonary nodules. Positron emission tomography (PET)-CT demonstrated a mild fluorodeoxyglucose (FDG) uptake (maximum standardized uptake value 5.4) in the tracheal lesion. Bronchoscopic findings revealed a submucosal tumor with vascular hyperplasia in the right upper ventral trachea with magenta coloring on autofluorescence imaging. Histopathologically, the biopsied specimen obtained from the lesion was diagnosed as solid-type ACC arising in the trachea. Conclusion. Our patient with solid-type ACC of the trachea had accompanying diplopia but showed no respiratory symptoms. ACC of the trachea sometimes show asymptomatic intramural progression without any respiratory symptoms, so physicians should be alert for this disease. |
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| AbstractList | Background. Adenoid cystic carcinoma (ACC) of the trachea is rare. ACCs are categorized into cribriform, tubular and solid types, and solid type is known to have the poorest prognosis. Case. A 57-year-old Japanese man who had suffered from diplopia for 2 months visited a hospital. Head magnetic resonance imaging (MRI) showed a right intraorbital tumor and multiple brain tumors, and chest computed tomography (CT) revealed multiple bilateral pulmonary nodules. He was introduced to our hospital for further examinations, and chest CT at the first visit showed a thickened tracheal wall in the right upper ventral trachea and multiple bilateral pulmonary nodules. Positron emission tomography (PET)-CT demonstrated a mild fluorodeoxyglucose (FDG) uptake (maximum standardized uptake value 5.4) in the tracheal lesion. Bronchoscopic findings revealed a submucosal tumor with vascular hyperplasia in the right upper ventral trachea with magenta coloring on autofluorescence imaging. Histopathologically, the biopsied specimen obtained from the lesion was diagnosed as solid-type ACC arising in the trachea. Conclusion. Our patient with solid-type ACC of the trachea had accompanying diplopia but showed no respiratory symptoms. ACC of the trachea sometimes show asymptomatic intramural progression without any respiratory symptoms, so physicians should be alert for this disease.
背景．気管原発腺様囊胞癌は稀な腫瘍である．病理学的には篩状型や管状型，充実型に分類されるが，充実型は予後不良とされる．症例．57歳，男性．2か月前からの複視を主訴に近医を受診した．頭部MRIで右眼窩内腫瘍と多発脳腫瘍，胸部CTで両肺に多発結節影を指摘され，精査のために当科を受診した．胸部CTで両肺の多発結節影に加え，上部気管の右腹側に壁肥厚とpositron emission tomography（PET）-CTでfluorodeoxyglucose（FDG）の軽度の集積亢進像（standardized uptake value（SUV）max 5.4）を認めた．気管支鏡検査では，上部気管の右腹側に表面平滑で血管増生に富んだ粘膜下腫瘍を認め，自家蛍光気管支鏡で同部位はマゼンタ色を示した．同部位の生検から，組織学的に充実型腺様囊胞癌と診断された．結論．複視を契機に診断した充実型気管原発腺様囊胞癌の1例を経験した．気管原発腺様囊胞癌は，腫瘍細胞が壁内に浸潤性に増殖・進展し，呼吸器症状を伴わないこともあり，見逃さないようにする必要がある． Background. Adenoid cystic carcinoma (ACC) of the trachea is rare. ACCs are categorized into cribriform, tubular and solid types, and solid type is known to have the poorest prognosis. Case. A 57-year-old Japanese man who had suffered from diplopia for 2 months visited a hospital. Head magnetic resonance imaging (MRI) showed a right intraorbital tumor and multiple brain tumors, and chest computed tomography (CT) revealed multiple bilateral pulmonary nodules. He was introduced to our hospital for further examinations, and chest CT at the first visit showed a thickened tracheal wall in the right upper ventral trachea and multiple bilateral pulmonary nodules. Positron emission tomography (PET)-CT demonstrated a mild fluorodeoxyglucose (FDG) uptake (maximum standardized uptake value 5.4) in the tracheal lesion. Bronchoscopic findings revealed a submucosal tumor with vascular hyperplasia in the right upper ventral trachea with magenta coloring on autofluorescence imaging. Histopathologically, the biopsied specimen obtained from the lesion was diagnosed as solid-type ACC arising in the trachea. Conclusion. Our patient with solid-type ACC of the trachea had accompanying diplopia but showed no respiratory symptoms. ACC of the trachea sometimes show asymptomatic intramural progression without any respiratory symptoms, so physicians should be alert for this disease. |
| Author | Nakamura, Midori Tachiwada, Takashi Noguchi, Shingo Yamasaki, Kei Higashi, Yasuyuki Kawanami, Toshinori Nemoto, Kazuki Yatera, Kazuhiro Uchimura, Keigo Morimoto, Toshiki |
| Author_FL | 森本 俊規 内村 圭吾 立和田 隆 野口 真吾 山﨑 啓 川波 敏則 東 泰幸 中村 碧 根本 一樹 矢寺 和博 |
| Author_FL_xml | – sequence: 1 fullname: 東 泰幸 – sequence: 2 fullname: 内村 圭吾 – sequence: 3 fullname: 森本 俊規 – sequence: 4 fullname: 中村 碧 – sequence: 5 fullname: 根本 一樹 – sequence: 6 fullname: 立和田 隆 – sequence: 7 fullname: 野口 真吾 – sequence: 8 fullname: 山﨑 啓 – sequence: 9 fullname: 川波 敏則 – sequence: 10 fullname: 矢寺 和博 |
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| References | 4. 新井 良, 石井芳樹. 腺様嚢胞癌. 呼吸. 2015;34:603-606. 5. Maziak DE, Todd TR, Keshavjee SH, et al. Adenoid cystic carcinoma of the airway: thirty-two-year experience. J Thorac Cardiovasc Surg. 1996;112:1522-1532. 9. 草地信也, 倉重真澄, 鈴木秀明, ほか. 気管, 気管支および肺のAdenoid Cystic Carcinomaの浸潤形態に関する検討-本邦報告例の文献的検討-. 肺癌. 1986;26:745-751. 6. Suzuki J, Ikeya T, Takahashi N, et al. A case of solid type pulmonary adenoid cystic carcinoma of the lung. J Jpn Soc Respir Endoscopy. 2014;36:288-292. 13. 道場昭太郎, 藤田秀人, 川村泰一, ほか. 右肺底幹気管支に発生したと思われる腺様嚢胞癌の1例. 胸部外科. 2003;56:977-980. 14. Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results. Ann Thorac Surg. 1990;49:69-77. 3. Kanematsu T, Yohena T, Uehara T, et al. Treatment outcome of resected and nonresected primary adenoid cystic carcinoma of the lung. Ann Thorac Cardiovasc Surg. 2002;8:74-77. 17. Bhattacharyya T, Bahl A, Kapoor R, et al. Primary adenoid cystic carcinoma of lung: a case report and review of the literature. J Cancer Res Ther. 2013;9:302-304. 1. Turnbull AD, Huvos AG, Goodner JT, et al. The malignant potential of bronchial adenoma. Ann Thorac Surg. 1972;14:453-464. 16. Song Z, Wu W, Zhang Y. Effective treatment with icotinib in primary adenoid cystic carcinoma of the lung with liver metastasis. J Thorac Oncol. 2014;9:e67-e69. 15. Misiukiewicz KJ, Camille N, Tishler R, et al. Organ preservation for adenoid cystic carcinoma of the larynx. Oncologist. 2013;18:579-583. 8. Nomori H, Kobayashi K, Ishihara T, et al. CT, bronchoscopic and pathological findings in adenoid cystic carcinoma of trachea and main stem bronchus. J Jpn Soc Bronchol. 1989;11:415-421. 12. Font RL, Ferry AP. Carcinoma metastatic to the eye and orbit III. A clinicopathologic study of 28 cases metastatic to the orbit. Cancer. 1976;38:1326-1335. 7. Nomori H, Kaseda S, Kobayashi K, et al. Adenoid cystic carcinoma of the trachea and main-stem bronchus. A clinical, histopathologic, and immunohistochemical study. J Thorac Cardiovasc Surg. 1988;96:271-277. 2. Travis WD, Travis LB, Devesa SS. Lung cancer. Cancer. 1995;75 (Suppl):191-202. 10. Huo Z, Meng Y, Wu H, et al. Adenoid cystic carcinoma of the tracheobronchial tree: clinicopathologic and immunohistochemical studies of 21 cases. Int J Clin Exp Pathol. 2014;7:7527-7535. 11. 雨宮次生, 林田祐彦, 嵩 義則. 日本における転移性眼窩腫瘍-文献的考察-. 眼科. 1998;40:307-328. |
| References_xml | – reference: 15. Misiukiewicz KJ, Camille N, Tishler R, et al. Organ preservation for adenoid cystic carcinoma of the larynx. Oncologist. 2013;18:579-583. – reference: 8. Nomori H, Kobayashi K, Ishihara T, et al. CT, bronchoscopic and pathological findings in adenoid cystic carcinoma of trachea and main stem bronchus. J Jpn Soc Bronchol. 1989;11:415-421. – reference: 7. Nomori H, Kaseda S, Kobayashi K, et al. Adenoid cystic carcinoma of the trachea and main-stem bronchus. A clinical, histopathologic, and immunohistochemical study. J Thorac Cardiovasc Surg. 1988;96:271-277. – reference: 13. 道場昭太郎, 藤田秀人, 川村泰一, ほか. 右肺底幹気管支に発生したと思われる腺様嚢胞癌の1例. 胸部外科. 2003;56:977-980. – reference: 16. Song Z, Wu W, Zhang Y. Effective treatment with icotinib in primary adenoid cystic carcinoma of the lung with liver metastasis. J Thorac Oncol. 2014;9:e67-e69. – reference: 17. Bhattacharyya T, Bahl A, Kapoor R, et al. Primary adenoid cystic carcinoma of lung: a case report and review of the literature. J Cancer Res Ther. 2013;9:302-304. – reference: 4. 新井 良, 石井芳樹. 腺様嚢胞癌. 呼吸. 2015;34:603-606. – reference: 3. Kanematsu T, Yohena T, Uehara T, et al. Treatment outcome of resected and nonresected primary adenoid cystic carcinoma of the lung. Ann Thorac Cardiovasc Surg. 2002;8:74-77. – reference: 5. Maziak DE, Todd TR, Keshavjee SH, et al. Adenoid cystic carcinoma of the airway: thirty-two-year experience. J Thorac Cardiovasc Surg. 1996;112:1522-1532. – reference: 11. 雨宮次生, 林田祐彦, 嵩 義則. 日本における転移性眼窩腫瘍-文献的考察-. 眼科. 1998;40:307-328. – reference: 10. Huo Z, Meng Y, Wu H, et al. Adenoid cystic carcinoma of the tracheobronchial tree: clinicopathologic and immunohistochemical studies of 21 cases. Int J Clin Exp Pathol. 2014;7:7527-7535. – reference: 9. 草地信也, 倉重真澄, 鈴木秀明, ほか. 気管, 気管支および肺のAdenoid Cystic Carcinomaの浸潤形態に関する検討-本邦報告例の文献的検討-. 肺癌. 1986;26:745-751. – reference: 2. Travis WD, Travis LB, Devesa SS. Lung cancer. Cancer. 1995;75 (Suppl):191-202. – reference: 12. Font RL, Ferry AP. Carcinoma metastatic to the eye and orbit III. A clinicopathologic study of 28 cases metastatic to the orbit. Cancer. 1976;38:1326-1335. – reference: 1. Turnbull AD, Huvos AG, Goodner JT, et al. The malignant potential of bronchial adenoma. Ann Thorac Surg. 1972;14:453-464. – reference: 14. Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results. Ann Thorac Surg. 1990;49:69-77. – reference: 6. Suzuki J, Ikeya T, Takahashi N, et al. A case of solid type pulmonary adenoid cystic carcinoma of the lung. J Jpn Soc Respir Endoscopy. 2014;36:288-292. |
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| Snippet | Background. Adenoid cystic carcinoma (ACC) of the trachea is rare. ACCs are categorized into cribriform, tubular and solid types, and solid type is known to... |
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| SubjectTerms | Adenoid cystic carcinoma Solid type Tracheal neoplasm 充実型 気管腫瘍 腺様囊胞癌 |
| Title | A Case of Solid-type Adenoid Cystic Carcinoma in the Upper Trachea Found by Diplopia |
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