Malignant adenomyoepithelioma of breast: a case report

Malignant adenomyoepithelioma of breast: a case report | Authorea try { document.documentElement.classList.add('js'); } catch (e) { } var _gaq = _gaq || []; _gaq.push(['_setAccount', 'G-8VDV14Y67G']); _gaq.push(['_trackPageview']); (function() { var ga = document.createElement('script'); ga.type = 'text/javascript'; ga.async = true; ga.src = ('https:' == document.location.protocol ? 'https://ssl' : 'http://www') + '.google-analytics.com/ga.js'; var s = document.getElementsByTagName('script')[0]; s.parentNode.insertBefore(ga, s); })(); Skip to main content Preprints Collections Wiley Open Research IET Open Research Ecological Society of Japan All Collections About About Authorea FAQs Contact Us Quick Search anywhere Search for preprint articles, keywords, etc. Search Search ADVANCED SEARCH SCROLL This is a preprint and has not been peer reviewed. Data may be preliminary. 11 January 2025 V1 Latest version Share on Malignant adenomyoepithelioma of breast: a case report Authors : Gengyu Chen 0000-0002-9254-1773 , Yiqiang Yin , and Tong Li [email protected] Authors Info & Affiliations https://doi.org/10.22541/au.173657721.16912852/v1 151 views 83 downloads Contents Abstract Information & Authors Metrics & Citations View Options References Figures Tables Media Share Abstract Malignant adenomyoepithelioma (MAME) of breast is an uncommon neoplasm, comprising merely 1% of primary breast malignancies. This case report presents a 66-year-old Chinese female presented to the clinic in August 2024 with an observed right breast mass that had been noticed 8 months prior, then the patient underwent a modified radical mastectomy and axillary lymph node dissection, with histopathology demonstrating malignant adenomyoepithelioma. This investigation aims to scrutinize the clinicopathological characteristics of a singular case of MAME while reviewing pertinent literature to augment comprehension regarding this condition. Introduction： Adenomyoepithelioma of the breast (AME) is a rare biphasic tumor composed of glandular epithelium and hypertrophic myoepithelium. Malignant transformation of either one or both components is referred to as malignant adenomyoepithelioma of the breast (MAME). Due to its complex histological manifestations and heterogeneous tissue configuration pattern, misdiagnosis can easily occur. This paper presents a case study on malignant AME, analyzing its clinical and pathological features in order to enhance understanding of the disease and provide evidence for clinical treatment. CASE PRESENTATION A 66-year-old Chinese female presented to the clinic in August 2024 with an observed right breast mass that had been noticed 8 months prior. Upon clinical examination, it was noted that the right breast nipple was not visible and a lump measuring approximately 10cm ×8cm in size was evident at the center of the breast. The lump exhibited a firm texture and vegetative appearance, along with multiple surface ruptures, limited mobility, indistinct boundaries, tenderness, and no palpable swollen lymph nodes in both axilla and clavicle regions. Ultrasonography revealed a solid-cystic mass measuring 10.0×8.0 cm within the right breast. CT scan results indicated no evidence of distant metastasis. The patient underwent a modified radical mastectomy and axillary lymph node dissection. Histopathology revealed the presence of malignant adenomyoepithelioma. Macroscopically, the tumor measured 10x8cm and appeared as a lobulated grey-white mass with irregular margins. Microscopic examination demonstrated invasion of the epidermal and dermal tissues by the tumor. The tumor cell nests were predominantly composed of proliferating myoepithelial cells, with a small number of irregular glandular cavities observed. These myoepithelial cells exhibited abundant cytoplasm or eosinophilic cells, indistinct cell membranes, round or oval nuclei with thick nuclear membranes, small nucleoli, moderate atypia, cuboid glandular epithelium, and red cytoplasm. The proliferating myoepithelial cells formed a ”double cannula”-like structure surrounding the glandular epithelium. The glandular epithelial cells were located near the coelomic side and displayed cuboidal morphology with ambiguous boundaries, subround nuclei that stained deeply, eosinophilic cytoplasmic content; while myoepithelial cells surrounded them from afar on the coelomic side. Fusiform-shaped transparent tumors showed invasive growth patterns characterized by significant cellular atypia and comedo-like necrosis (Figure 1). Immunohistochemical results showed (B5 slices): CK5/6 (+), CD10 (+), CK7 (local +), CK8/18 (+), ER (-), PR (-), Her- 2 (0), CD117 (-), GFAP (-), S - 100 (-), AR (-), GATA - 3 (-). (B4 slices): GATA - 3 (-), Mammaglobin (+), P63(+), CR (+), CK (+), PAX - 8 (-), CK5/6 (+), Calponin (+), SMA (+), EMA (local +), Ki - 67 about 20% of (+), Syn(-). Special dyeing:PAS (+), D-PAS (-) (Figure 2). Eighteen lymphnodes were isolated, none of which showed tumor deposit. The patient did not take follow-up treatment after surgery. The patient has been followed up for 3 months without significant recurrence. Discussion Adenomyoepithelioma of breast is a rare breast tumor with epithelial-myoepithelial differentiation, which was first reported by Hamperl in 1970. Malignant transformation can occur in either or both components of adenomyoepithelioma. Adenomyoepithelioma accompanied by cancer is defined as malignant adenomyoepithelioma by WHO (2019) classification of breast tumors[6]. The incidence of MAME is exceedingly rare in clinical practice, predominantly observed among elderly females aged between 26 and 86 years. Tumor sizes ranged from 0.6 to 9.0 cm, with approximately half occurring in the outer upper quadrant. In this case, the patient is a 66-year-old female with a tumor located in the middle and upper quadrant of the lateral breast, which aligns closely with existing literature reports. Currently, there is no standardized diagnostic criteria for malignant adenomyoepithelioma (MAME). The reference criteria reported in the literature are as follows[9]: (1) Invasion of the tumor leading to destruction of breast structure or surrounding adipose tissue; (2) Significant proliferation of glandular epithelium or myoepithelial cells with evident atypia, enlarged nuclei, irregularities, and visible nucleoli; (3) Presence of more than 5 pathological mitotic images per 10 high-power fields; (4) Occurrence of necrosis within the tumor; (5) Analysis revealing DNA polyploidy indicating aneuploidy in tumor cells. In this case, the tumor exhibited invasive growth by infiltrating both epidermis and dermis. Immunohistochemical markers confirmed expression of glandular epithelial cells and myoepithelial cells. The myoepithelial cytoplasm appeared transparent with an invasive growth pattern characterized by round or oval nuclei, thick nuclear membranes, small nucleoli, prominent cellular atypia, and comedo-like necrosis. This diagnosis aligns with simultaneous malignant transformation from two types of adenomyoepithelioma into malignant adenomyoepithelioma. The distinction between MAME and malignant myoepithelioma, low-grade adenosquamous carcinoma, papillary sarcoma, and metaplastic carcinoma needs to be made. When considering the differential diagnosis, particular attention should be given to immunohistochemical findings, especially myoepithelial markers. MAME exhibits the expression of basal cell myoepithelial markers such as CK5/6, p63, and SMMHC. The epithelial cell markers include CK7 and CK8/18 while ER, PR, and Her-2 are not expressed[11]. In this case study, positive immunohistochemical staining for the myoepithelial marker CK5/6 and epithelial cell marker CK7 was observed while ER, PR, and Her-2 were negative; these findings are consistent with the aforementioned research results. The MAME condition demonstrates heightened invasiveness, susceptibility to recurrence, and a propensity for metastasis. Blood metastases are more prevalent than lymphatic metastases, with distant spread occurring readily. Common sites of metastasis include the lungs and brain[13], while the risk of distant dissemination is positively correlated with tumor size[14]. Currently, there is no standardized approach for treating MAME. In clinical practice, extended resection combined with sentinel lymph node dissection remains the preferred treatment option. Post-surgical local radiotherapy is employed followed by systemic chemotherapy based on pathological findings. Due to negative ER and PR status along with ineffective endocrine therapy, patients experiencing recurrence and metastasis face an extremely poor prognosis; most succumb within a few years after initial treatment. MAME is a rare neoplasm of breast shows spectrum of histological pattern due to which it causes diagnostic confusion. The diagnosis of this neoplasm can be challenging, histological features and immunophenotype are the key to diagnosis. Funding No funding. Conflicts of Interest All authors have completed the ICMJE niform disclosure form. The authors have no conflicts of interest to declare. Registry and the Registration No. of the study/trial. N/A. Animal Studies. N/A. Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was provided by the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal. Open Access Statement : This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the noncommercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). References [1] WHO classification of tumors editorial board.WHO classification of tumors[M]．5th ed.Lyon: IARCP Press,2019:46-48 [2] Bult P, Verwiel J M M, Wobbes T, et al.Malignant adenomyoepithelioma of the breast with metastasis in the thyroid gland 12 years after excision of the primary tumor. Case report and review of the literature.[J].Virchows Archiv, 2000, 436(2):158-166.DOI:10.1007/PL00008216. [3] Papazian M, Kalantzis I, Galanopoulos G, Mani I, Tzaida O, Iacovidou I, Ziras N. Malignant myoepithelioma of the breast: A case report and review of the literature. Mol Clin Oncol. 2016 May;4(5):723-727. doi: 10.3892/mco.2016.808. Epub 2016 Mar 7. PMID: 27123270; PMCID: PMC4840516. [4] Bult P, Verwiel J M M, Wobbes T,et al.Malignant adenomyoepithelioma of the breast with metastasis in the thyroid gland 12 years after excision of the primary tumor. Case report and review of the literature.[J].Virchows Archiv, 2000, 436(2):158-166.DOI:10.1007/PL00008216. [5] Petrozza V, Pasciuti G, Pacchiarotti A,et al.Breast adenomyoepithelioma: A case report with malignant proliferation of epithelial and myoepithelial elements[J].World Journal of Surgical Oncology, 2013, 11(1):285.DOI:10.1186/1477-7819-11-285. Figure 1 A: The tumor cells invaded the epidermis with comedo-like necrosis. B: The tumor cells invaded the dermal tissue. C:The proliferating myoepithelial cells formed a ”double sleeve” -like structure around the glandular epithelium. The myoepithelial cytoplasmic transparent tumor showed infiltrative growth with obvious cell atypia and comedo-like necrosis. Figure 2 Immunohistochemical A: CK5/6(+) B：CK7（+） Information & Authors Information Version history V1 Version 1 11 January 2025 Copyright This work is licensed under a Non Exclusive No Reuse License. Authors Affiliations Gengyu Chen 0000-0002-9254-1773 Fourth People's Hospital of Jinan View all articles by this author Yiqiang Yin Fourth People's Hospital of Jinan View all articles by this author Tong Li [email protected] Fourth People's Hospital of Jinan View all articles by this author Metrics & Citations Metrics Article Usage 151 views 83 downloads .FvxKWukQNSOunydq8rnd { width: 100px; } Citations Download citation Gengyu Chen, Yiqiang Yin, Tong Li. Malignant adenomyoepithelioma of breast: a case report. Authorea . 11 January 2025. 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