Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 16, 2022; 10(11): 3630-3638
Published online Apr 16, 2022. doi: 10.12998/wjcc.v10.i11.3630
Pleomorphic adenoma of the left lacrimal gland recurred and transformed into myoepithelial carcinoma after multiple operations: A case report
Wen-Peng Huang, Li-Ming Li, Jian-Bo Gao
Wen-Peng Huang, Li-Ming Li, Jian-Bo Gao, Department of Radiology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, Henan Province, China
Author contributions: Huang WP contributed to the acquisition and analysis of the work, drafted the manuscript, imaging data collection and analysis; Li LM contributed to the manuscript editing; Gao JB wrote the review and editing; all authors met the requirements for authorship for the submitted version and agreed to its submission.
Informed consent statement: Participant gave written consent to participate in the study.
Conflict-of-interest statement: The authors declare that they have no competing interests.
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: Jian-Bo Gao, PhD, Academic Research, Chairman, Chief Doctor, Instructor, Department of Radiology, The First Affiliated Hospital of Zhengzhou University, No. 1 East Jianshe Road, Zhengzhou 450052, Henan Province, China. jianbogaochina@163.com
Received: December 14, 2021
Peer-review started: December 14, 2021
First decision: January 25, 2022
Revised: February 2, 2022
Accepted: February 27, 2022
Article in press: February 27, 2022
Published online: April 16, 2022
Abstract
BACKGROUND

Myoepithelial carcinoma (MC) is a clinically rare malignancy, there is controversy regarding its etiology and its biological behavior is not fully elucidated. Extensive surgical resection is the main treatment method. We describe a case of pleomorphic adenoma (PA) with multiple postoperative recurrences after malignant transformation, and the history of the disease in this patient was more than 20 years. Complete resection during the first surgery of PA and long-term postoperative follow-up is necessary.

CASE SUMMARY

A 34-year-old male with PA and a history of 5 postoperative recurrences over 21 years, each surgically removed, presented 15 d ago with headache, nasal congestion, protrusion of the right eyeball and loss of vision in the right eye, with progressively worsening symptoms. The patient underwent surgery, and MC was confirmed by pathology examination. A small PA component was locally visible under light microscope. The patient had a recurrence of the tumor 2 mo after surgery and underwent surgical resection.

CONCLUSION

During the first operation for PA, care should be taken not to rupture the envelope to prevent tumor cell implantation, and when complete resection is not possible due to the anatomical site, postoperative radiotherapy is necessary to control the lesion and prevent infiltration and malignant transformation of the tumor to MC. Computed tomography and magnetic resonance imaging is important for establishing diagnosis and developing a treatment plan.

Keywords: Myoepithelial carcinoma, Pleomorphic adenoma, Magnetic resonance imaging, Treatment, X-ray computed tomography, Case report

Core Tip: This case illustrates that pleomorphic adenoma should be completely removed during the first operation to prevent capsule rupture and tumor cell implantation. When complete resection is not possible due to the anatomical site, postoperative radiotherapy should be performed to control the lesion and prevent infiltration and malignant transformation to myoepithelial carcinoma. Postoperative follow-up, especially long-term follow-up for recurrent cases, is necessary and systemic examination should be undertaken to prevent distant metastasis.