Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 6, 2019; 7(17): 2623-2629
Published online Sep 6, 2019. doi: 10.12998/wjcc.v7.i17.2623
Carcinoma ex pleomorphic adenoma of the trachea: A case report
Heng-Xing Gao, Quan Li, Wen-Li Chang, Ya-Long Zhang, Xiao-Zhi Wang, Xue-Xue Zou
Heng-Xing Gao, Xiao-Zhi Wang, Respiratory Department, Binzhou Medical University Hospital, Binzhou 256603, Shandong Province, China
Quan Li, Xue-Xue Zou, Department of Radiology, Binzhou Medical University Hospital, Binzhou 256603, Shandong Province, China
Wen-Li Chang, Ya-Long Zhang, Department of Pathology, Binzhou Medical University Hospital, Binzhou 256603, Shandong Province, China
Author contributions: Gao HX and Li Q drafted the first manuscript. Chang WL and Zhang YL contributed to the histological diagnosis. Wang XZ searched the literature data and was responsible for the clinical care. Zou XX supervised the manuscript revision and provided imaging materials.
Informed consent statement: Informed written consent was obtained from the patient.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
Corresponding author: Xue-Xue Zou, MD, Doctor, Department of Radiology, Binzhou Medical University Hospital, The Yellow River Road, Binzhou 256603, Shandong Province, China.
Telephone: +86-543-3256590
Received: March 21, 2019
Peer-review started: March 23, 2019
First decision: July 30, 2019
Revised: August 6, 2019
Accepted: August 20, 2019
Article in press: August 20, 2019
Published online: September 6, 2019

Carcinoma ex pleomorphic adenoma (CXPA) is defined as a malignant salivary gland tumor arising from a primary or recurrent pleomorphic adenoma. Only three cases of CXPA of the trachea have been reported in the literature.


We report a case of tracheal CXPA in a 55-year-old woman, who presented with a more than 3-mo history of progressive dyspnea. Computed tomography of the neck and thorax revealed an inhomogeneous, broad-based lesion arising from the tracheal wall on the right side. Endoscopy revealed a subglottic neoplasm causing up to 90% luminal stenosis. The tumor was resected using a high-frequency electrosurgical snare combined with argon plasma coagulation. Histopathology and immunohistochemistry revealed that the tumor was a CXPA of the trachea.


We report the fourth case of tracheal CXPA, and present the first instance of resection of CXPA using high-frequency electrosurgical snare and laser ablation. We also discuss the pathogenesis, diagnosis, histopathology, and systemic therapy of this rare disease.

Keywords: Case report, Carcinoma ex pleomorphic adenoma, Trachea, Pleomorphic adenoma

Core tip: After extensive search of literature in English, there are only 3 previous reports of primary Carcinoma ex pleomorphic adenoma of the trachea. In our case, there are many unique features. In imaging examination, there is no calcification can be seen in the primary tracheal CXPA of the previous cases, but ours can. Given the rare incidence of CXPA in trachea, no standard systemic therapy options have been established. We successfully performed endobronchial resection of the tumor with a high-frequency electrosurgical snare combined with argon plasma coagulation with less injury and quick recovery.