Abstract

BackgroundMalignant adenomyoepithelioma and adenoid cystic carcinoma are both rare malignant epithelial-myoepithelial tumors of the breast. We report a very rare case with a malignant adenomyoepithelioma combining with adenoid cystic carcinoma in a single mass.Case presentationA 61-year-old female presented with a palpable painless mass in the right breast. Mammography revealed a large irregular dense shadow without obvious internal calcification. A simple lumpectomy was performed, and a 1.6cm well-circumscribed pale-tan nodule was presented. Histologically, the nodule was composed of two obscure lobules. One exhibited typical histological image of adenoid cystic carcinoma, the other one showed the image of epithelial-myoepithelial carcinoma of salivary gland, and support the diagnosis of biphasic malignant adenomyoepithelioma. Transition between the two lobules was gradual. Immunohistochemically, CK18 and P63 highlighted the epithelial and myoepithelial cells respectively in both lesions. CD117 was positive in the epithelial cells of adenoid cystic carcinoma, but was totally negative in malignant adenomyoepithelioma.ConclusionThis report is, to our knowledge, the first case that combines these two tumors in a single mass. In addition, we present a review of the literature. The histogenesis of these tumors is also discussed.Virtual SlidesThe virtual slide(s) for this article can be found here: http://med.motic.com/MoticGallery/Slide?id=D562817E-23C2-4F72-9823-86EF6DA40005&user=2C69F0D6-A478-4A2B-ABF0-BB36763E8025 and http://med.motic.com/MoticGallery/Slide?id=38BB7126-6FFB-4B66-A208-B8C0F528DCA8&user=2C69F0D6-A478-4A2B-ABF0-BB36763E8025

Highlights

  • Malignant adenomyoepithelioma and adenoid cystic carcinoma are both rare malignant epithelial-myoepithelial tumors of the breast

  • * Correspondence: bianli1976@sina.com †Equal contributors 1Department of Pathology, The First Affiliated Hospital of Kunming Medical University, 295, Xichang road, Kunming, Yunnan, China Full list of author information is available at the end of the article cystic carcinoma arising within an adenomyoepithelioma so far [3].To the best of our knowledge, what we present is the first case that combines malignant adenomyoepithelioma and adenoid cystic carcinoma in a single mass

  • According to the growth pattern of malignant components, the cases reported in the literatures can be divided into two types: 1) an area of obvious malignant component arising in a typical low-grade adenomyoepithelioma, which may be more appropriately named “adenomyoepithelioma with carcinoma” as defined in the 4th edition of WHO Classification of Tumours of the Breast [4], or “carcinoma arising in adenomyoepithelioma”; 2) an adenomyoepithelioma with apparent cytological atypia, increased mitotic activity (>5/10HPFs), invasive growth pattern, and/or presence of necrosis in both epithelial and myoepithelial cells as the histological image of the current case, reminiscent of epithelial-myoepithelial carcinoma of salivary glands [5,6]

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Summary

Introduction

Malignant adenomyoepithelioma and adenoid cystic carcinoma are both rare malignant epithelial-myoepithelial tumors of the breast. We report a very rare case with a malignant adenomyoepithelioma combining with adenoid cystic carcinoma in a single mass. The nodule was composed of two obscure lobules. One exhibited typical histological image of adenoid cystic carcinoma, the other one showed the image of epithelial-myoepithelial carcinoma of salivary gland, and support the diagnosis of biphasic malignant adenomyoepithelioma. CK18 and P63 highlighted the epithelial and myoepithelial cells respectively in both lesions. CD117 was positive in the epithelial cells of adenoid cystic carcinoma, but was totally negative in malignant adenomyoepithelioma. Malignant adenomyoepithelioma (AME) and adenoid cystic carcinoma (ACC) are both rare malignant epithelialmyoepithelial lesions of the breast. Composed of epithelial and myoepithelial cells, these two salivary gland-like tumors of the breast are morphologically close.

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