• Zhihao Zhang, Yueyuan Wang, Xiao Xie, Jingyu Peng, Jinghui Hong, Lirong Bi, and Ming Yang.
• Department of Breast Surgery.
• Medicine (Baltimore). 2021 Feb 5; 100 (5): e24461e24461.

RationaleAdenomyoepithelioma (AME) is a rare biphasic tumor consisting of epithelial and Myoepithelial cell. Most of the AME is benign, and only a few will progress to malignancy, Here, we report a case of low-grade malignant adenomyoepithelioma, and review the related literature, in a bid to investigate its clinical and pathological features and thus, enhance our understanding of this tumor.Patient ConcernsA 64-year-old woman visited our hospital with a 1-year history of a painless mass in her left breast. Physical examination revealed a palpable painless mass, measuring approximately 4.5 cm, in the left breast.DiagnosisHistological examination confirmed the diagnosis of malignant adenomyoepithelioma.InterventionsThe patient underwent local excision of the mass, with frozen section analysis revealing ductal carcinoma in situ. Mastectomy and sentinel lymph node biopsy were then performed.OutcomesWe conducted a one-year follow-up, and relapse was not observed.LessonsTreatment of AME remains controversial owing to the lack of high volume data and absence of prospective studies. Simple mastectomy is an acceptable treatment of this tumor.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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