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A 51-year-old woman presented with a gradually enlarging left breast mass; imaging showed a 3.6 cm lesion. Core biopsy demonstrated biphasic proliferation with myoepithelial atypia and high mitotic activity. Wide local excision with oncoplastic reduction achieved negative margins, and pathology confirmed pure M-AME (pT2 pN0), triple-negative by receptor testing, with Ki-67 35%–40%. Adjuvant radiotherapy followed, and disease-free status persisted at 1 year.",{"@graph":69,"@context":122},[70,84,105],{"@type":71,"itemListElement":72},"BreadcrumbList",[73,77,79,82],{"item":74,"name":75,"@type":76,"position":8},"https://docshare.wps.com","Home","ListItem",{"item":78,"name":9,"@type":76,"position":14},"https://docshare.wps.com/document/",{"item":80,"name":35,"@type":76,"position":81},"https://docshare.wps.com/document/healthcare/",3,{"item":83,"name":65,"@type":76,"position":19},"https://docshare.wps.com/document/malignant-adenomyoepithelioma-of-the-breast-a-rare-case-report-and-literature-review/440687/",{"url":83,"name":65,"@type":85,"image":86,"author":91,"headline":65,"publisher":94,"fileFormat":97,"inLanguage":63,"description":67,"dateModified":98,"datePublished":99,"encodingFormat":97,"isAccessibleForFree":100,"interactionStatistic":101},"DigitalDocument",{"url":87,"@type":88,"width":89,"height":90},"https://docshare.wps.com/thumbnails/malignant-adenomyoepithelioma-of-the-breast-a-rare-case-report-and-literature-review/440687.png","ImageObject",300,407,{"name":92,"@type":93},"moss~","Person",{"url":74,"name":95,"@type":96},"DocShare","Organization","application/pdf","2026-11-04","2026-09-29",true,{"@type":102,"interactionType":103,"userInteractionCount":52},"InteractionCounter",{"@type":104},"ViewAction",{"@type":106,"mainEntity":107},"FAQPage",[108,114,118],{"name":109,"@type":110,"acceptedAnswer":111},"What is malignant adenomyoepithelioma (M-AME) of the breast?","Question",{"text":112,"@type":113},"It is a rare malignant transformation of adenomyoepithelioma, a typically biphasic tumor composed of epithelial and myoepithelial elements.","Answer",{"name":115,"@type":110,"acceptedAnswer":116},"How was the diagnosis established in this case?",{"text":117,"@type":113},"Imaging identified a 3.6 cm lesion, and core biopsy showed biphasic proliferation with myoepithelial atypia and high mitotic activity; final histopathology confirmed pure M-AME with receptor and Ki-67 assessment.",{"name":119,"@type":110,"acceptedAnswer":120},"What treatment was performed and what was the short-term outcome?",{"text":121,"@type":113},"The patient underwent wide local excision with oncoplastic reduction to achieve negative margins, followed by adjuvant radiotherapy; the patient remained disease-free at 1 year.","https://schema.org",{"og:url":83,"og:type":124,"og:title":65,"og:site_name":95,"og:description":67},"article",{"robots":126,"canonical":83},"index,follow",{"doc_id":128,"site_id":62},440687,1790922318,{"code":4,"msg":5,"data":131},{"doc_id":128,"user_id":132,"nickname":92,"user_avatar":133,"doc_module":4,"category_id":34,"category_name":35,"doc_title":65,"doc_description":67,"doc_content":134,"file_id":135,"file_url":136,"file_type":137,"file_size":138,"view_count":52,"is_deleted":4,"is_public":8,"is_downloadable":8,"audit_status":8,"page_count":19,"language":139,"language_code":63,"site_id":62,"html_lang":63,"table_of_contents":140,"faqs":141,"seo_title":142,"seo_description":67,"update_tm":143,"read_time":52,"preview_page_count":4},962090895452,"https://ap-avatar.wpscdn.com/davatar_a8503ba1806abce46bf441b54a3ca4cd","Journal of Surgical Case Reports, 2026, 1, rjaf1039  \n[https://doi.org/10.1093/jscr/rjaf1039](https://doi.org/10.1093/jscr/rjaf1039)  \nCase Report  \nCase Report  \nMalignant adenomyoepithelioma of the breast: a rare case report and literature review  \nGalia Jadkarim1 , Raghad H. Alsharif2 , Fatmah Kahtani2 , Meral Altayeb3 , Faiz Zagzoog3 , Shaza Alsharif4 , 5 , 6 , Rana Ajabnoor 7 , *  \n1 Department of Breast Surgical Oncology, King Abdulaziz Medical City, National Guard Hospital, Ministry of the National Guard-Health Affairs, Jeddah 21423, Saudi Arabia  \n2 Department of General Surgery, King Abdulaziz Medical City, National Guard Hospital, Ministry of the National Guard-Health Affairs, Jeddah 21423, Saudi Arabia  \n3 Department of Surgery, King Abdulaziz Hospital, Jeddah 22421, Saudi Arabia  \n4 College of Applied Medical Sciences, King Saud Bin Abdulaziz University for Health Sciences, Jeddah 21423, Saudi Arabia  \n5 King Abdullah International Medical Research Center, Jeddah 21423, Saudi Arabia  \n6 Department of Radiology, King Abdulaziz Medical City, National Guard Hospital, Ministry of the National Guard-Health Affairs, Jeddah 21423, Saudi Arabia  \n7 Department of Anatomical Pathology, Faculty of Medicine, King Abdulaziz University, Jeddah 21589, Saudi Arabia  \n*Corresponding author. Department of Anatomical Pathology, King Abdulaziz University, Jeddah 21589, Saudi Arabia. E-mail: [rajabnoor@kau.edu.sa](rajabnoor@kau.edu.sa)  \nAbstract  \nAdenomyoepithelioma (AME) of the breast is a rare biphasic tumor composed of epithelial and myoepithelial elements, with malignant transformation being exceptionally uncommon. We report a case of pure malignant adenomyoepithelioma (M-AME) in a 51-year-old woman who presented with a gradually enlarging left breast mass. Imaging revealed a 3.6 cm lesion, and core biopsy showed biphasic proliferation with myoepithelial atypia and high mitotic activity. Wide local excision with oncoplastic reduction achieved negative margins, and histopathology confirmed pure M-AME (pT2 pN0), triple-negative for estrogen, progesterone, and Human Epidermal growth factor Receptor 2, with a Ki-67 index of 35%–40% . The patient received adjuvant radiotherapy and remains disease-free at 1 year. This case highlights the diagnostic challenges of M-AME, underscores the importance of immunohistochemistry and complete excision with negative margins, and adds to the limited literature on this exceptionally rare breast neoplasm.  \nKeywords: malignant adenomyoepithelioma; oncoplastic mammoplasty; adjuvant radiotherapy, follow up  \nIntroduction  \nAdenomyoepithelioma (AME) of the breast is an uncommon biphasic tumor composed of both epithelial and myoepithelial elements. Although typically benign, AME has the potential for malignant transformation in one or both cellular components, giving rise to malignant adenomyoepithelioma (M-AME) [1 , 2] .  \nAccording to a recent systematic review by Chen et al. [3], only 167 cases of M-AME have been documented in the literature, underscoring its extreme rarity, and the limited understanding of its biological behavior [4] . Because of this scarcity, establishing accurate diagnosis and optimal management strategies remains difficult. Current evidence is mainly derived from individual case reports and small series.  \nSurgical excision with negative margins continues to be the cornerstone of treatment, as lymph-node involvement and distant metastasis are relatively uncommon [1 , 5 , 6] .  \nHere, we present a case of pure malignant adenomyoepithelioma, not associated with conventional invasive ductal carcinoma of no special type, incidentally, detected during  \nradiologic evaluation performed prior to reduction mammoplasty. The aim of reporting this case is to contribute to the limited body of literature on this exceptionally rare tumor and to share our experience in broadening the understanding of its clinicopathological characteristics.  \nCase presentation  \nA 51-year-old multiparous woman,","cbCaivP3tzXvaExr","https://ap.wps.com/l/cbCaivP3tzXvaExr","pdf",798692,"English","# Abstract\n# Introduction\n# Case presentation\n## Clinical presentation and comorbidities\n## Imaging findings\n## Diagnosis and treatment\n# Discussion\n# Conclusion","[{\"question\":\"What is malignant adenomyoepithelioma (M-AME) of the breast?\",\"answer\":\"It is a rare malignant transformation of adenomyoepithelioma, a typically biphasic tumor composed of epithelial and myoepithelial elements.\"},{\"question\":\"How was the diagnosis established in this case?\",\"answer\":\"Imaging identified a 3.6 cm lesion, and core biopsy showed biphasic proliferation with myoepithelial atypia and high mitotic activity; final histopathology confirmed pure M-AME with receptor and Ki-67 assessment.\"},{\"question\":\"What treatment was performed and what was the short-term outcome?\",\"answer\":\"The patient underwent wide local excision with oncoplastic reduction to achieve negative margins, followed by adjuvant radiotherapy; the patient remained disease-free at 1 year.\"}]","Malignant adenomyoepithelioma of the breast - a rare case report and literature review | PDF",1790692963]