Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. May 16, 2022; 10(14): 4648-4653
Published online May 16, 2022. doi: 10.12998/wjcc.v10.i14.4648
Diagnosis of an extremely rare case of malignant adenomyoepithelioma in pleomorphic adenoma: A case report
Wei-Tong Zhang, Ya-Bing Wang, Yi Ang, Hui-Zhen Wang, Yong-Xiang Li
Wei-Tong Zhang, Hui-Zhen Wang, Yong-Xiang Li, Department of General Surgery, The First Affiliated Hospital of Anhui Medical University, Hefei 23000, Anhui Province, China
Ya-Bing Wang, Yi Ang, Department of Breast Surgery, Yijishan Hospital of Wannan Medical College, Wuhu 241000, Anhui Province, China
Author contributions: Zhang WT and Wang YB were the patient’s surgeon, reviewed the literature and contributed to manuscript drafting; Zhang WT and Ang Y reviewed the literature and contributed to manuscript drafting; Wang HZ analyzed and interpreted the imaging findings; Li YX was responsible for the revision of the manuscript for important intellectual content; all authors issued final approval for the version to be submitted.
Supported by National Natural Science Foundation of China, No. 81874063; and Natural Science Foundation of Anhui Province, No. 2008085QH408.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Yong-Xiang Li, MD, Chief Doctor, Professor, Surgeon, Department of General Surgery, The First Affiliated Hospital of Anhui Medical University, No. 218 Jixi Road, Hefei 23000, Anhui Province, China. liyongxiang@ahmu.edu.cn
Received: December 8, 2021
Peer-review started: December 8, 2021
First decision: January 25, 2022
Revised: January 29, 2022
Accepted: March 26, 2022
Article in press: March 26, 2022
Published online: May 16, 2022
Processing time: 156 Days and 0.1 Hours
Abstract
BACKGROUND

Pleomorphic adenoma (PA) is the most common type of salivary gland tumor, and its common sites are parotid gland, sinus, nasal septum and cleft palate. PA is an uncommon benign type of tumor occurring in the breast, and there are few reports of cases in Asia.

CASE SUMMARY

An 84-year-old woman found a mass in the upper outer quadrant of the right breast > 1 year ago. The patient underwent a right breast lumpectomy and sentinel lymph node biopsy. The pathological diagnosis was PA in the upper outer quadrant of the right breast, and the malignant component was malignant adenomyoepithelioma. The postoperative course was uneventful, and no chemotherapy was administered. At 18 mo of follow-up, the patient is alive and well, with no evidence of recurrent disease.

CONCLUSION

Patients with breast PA should first undergo extended excision of breast masses followed by pathological examination. If malignancy is confirmed or the surgical margin is positive, modified radical mastectomy should be performed.

Keywords: Pleomorphic adenoma of breast; Diagnosis; Malignant adenomyoepithelioma; Salivary gland tumors; PLAG1; HMGA2; Case report

Core Tip: Pleomorphic adenoma (PA) is an uncommon benign type of tumor occurring in the breast, and there are few reports of cases in Asia. We here present an extremely rare case of malignant adenomyoepithelioma in PA. We performed a detailed pathological examination, immunohistochemistry and fluorescence in situ hybridization (including detection of PLAG1 and HMGA2 gene rearrangement). We also summarize the pathological differential diagnosis of PA.