Clinical and pathological characteristics in malignant adenomyoepithelioma of the breast: a systematic review of case reports

Introduction

Background

Breast adenomyoepithelioma (AME) is a rare biphasic neoplasm. First described by Hamperl in 1970, it is characterised by the proliferation of both epithelial and myoepithelial cells (1-4). Whilst most cases are benign, AME has the potential to become invasive when one or both cell types undergo malignant transformation.

Malignant adenomyoepithelioma (MAME) was first described by Tavassoli in 1991 (5) and officially characterised by the World Health Organisation in 2002 (6). Histological features indicative of malignancy include nuclear pleomorphism, necrosis, infiltrative borders, invasive growth, and high mitotic index (2,3,7-9).

Rationale and knowledge gap

Diagnosing MAME through pre-operative investigations is challenging due to its biphasic origin. Excisional biopsy is currently recommended to rule out malignant differentiation even if a fine needle aspirate (FNA) or core needle biopsy (CNB) suggests benign disease because of the tumour’s morphological heterogeneity (3). Due to its rarity, the biological behaviour of MAME is not well understood and there is no published guideline or consensus on its management. Current literature is limited to case reports and small case series.

Objective

This is the first systematic review on this topic that aims to address current knowledge gaps and to identify the clinical and pathological characteristics that may be associated with recurrent or metastatic potential which in turn may be used to guide clinicians in managing this rare cohort of patients. We present this article in accordance with the PRISMA reporting checklist (available at https://abs.amegroups.com/article/view/10.21037/abs-25-4/rc) (10).

Methods

Study selection

A systematic literature search was conducted using the Embase, MEDLINE, Cochrane, and PubMed databases from inception to the time of the last search on 5 September 2023. The search strategy was carried out using the keywords adenomyoepithelioma, malignant adenomyoepithelioma, breast cancer/neoplasm/malignancy/carcinoma, mammary cancer; Medical Subject Headings [MeSH] terms including “adenomyoepithelioma” and “breast neoplasms”; and Embase plus Medline subject headings such as “adenomyoepithelioma of the breast” and “breast adenomyoepithelioma”.

An advanced search with the combination of keywords, MeSH terms and subject headings using Boolean operators (‘AND’ ‘OR’) was performed (Table 1). Two authors (N.C. and N.S.) performed the systematic literature search. Furthermore, the references from all included papers were manually searched for additional publications.

Selection criteria and data extraction

Studies were included if they: (I) reported on a case of MAME; (II) were published in English; (III) had the full text article available. Studies were excluded if (I) the pathology was a pure myoepithelioma not associated with AME; (II) the article included only conference/meeting abstracts; (III) had insufficient case data for inclusion. For the purposes of this review, papers were not grouped by study design.

Titles and abstracts of all articles were initially screened to determine their relevance to the topic. Duplicates were excluded in this stage. Full texts of relevant articles were then retrieved and reviewed to determine their inclusion. Articles that did not meet the inclusion criteria or met exclusion criteria were excluded. To limit bias, title and abstract screening were conducted by two authors (T.D. and A.B.) and discrepancies were resolved by discussion with a third author (M.M.). Full-text articles were reviewed by two different authors (B.C. and N.S.) and discrepancies were resolved with a third (N.C.). The references of all included papers after full text review were manually searched for additional titles (X.R.W.). New articles found during reference review were subjected to the same process (Figure 1).

Figure 1 PRISMA flow chart diagram of study selection process. AME, adenomyoepithelioma; PRISMA, Preferred Reporting Items for Systematic Reviews and Meta-Analyses.

A narrative synthesis rather than a formal meta-analysis was selected due to the heterogeneity of the publications discovered. We anticipated substantial variability in reporting quality across studies. As our primary aim was to provide a comprehensive synthesis of all available published data on this topic, excluding lower-quality reports could have led to a much less complete review. We therefore chose not to formally assess risk of bias or exclude studies based on methodological quality, though we acknowledge this as a limitation. Additionally, we excluded abstracts and non-peer-reviewed material to ensure that only fully published data were included.

Data extracted from the selected studies, where it was available, include (I) patient demographics such as age, gender, laterality of disease and presenting symptoms; (II) investigations such as pre-operative FNA or CNB findings; (III) histopathological findings such as size of tumour, malignant component, microscopic features, architecture subtype, infiltrative border, nuclear atypia, necrosis, nuclear pleomorphism, lymphovascular invasion (LVI), perineural invasion (PNI), calcifications, mitotic rate, Ki-67, hormone receptor markers and margins; (IV) management details including type of surgery, lymph node (LN) assessment, neoadjuvant and adjuvant therapy, timing and management of recurrent or metastatic disease if applicable, follow-up period and death if reported. Data was extracted by a single researcher independently for each report and entered into a database, which was reviewed and verified by two researchers in collaboration (N.C. and M.M.).

A protocol was not registered prior to commencing. No automated tools were used in data extraction or analysis. No assumptions were made about missing information.

Results

A total of 167 cases of MAME were identified from 92 publications within the search period (Table 2). Table 3 displays clinical and pathological characteristics of the cases.

Clinical findings

Where sex was reported 99.3% (n=135) were female, with one male patient (11). The average age at presentation was 61.1 years old, ranging from 20 (12) to 93 years old (7,13). The average follow-up period was 35.7 (range, 3–210) months. Where mortality data was reported, 20 patients died from the disease (17.9%, 20 of 112), all of whom had metastases. Death occurred at an average of 34.9 months from the time of diagnosis (reported in 16 cases, range, 3–154 months).

The duration of symptoms preceding diagnosis was reported in 34 cases. The median duration was 12 months, ranging from one week in a patient with a painful breast lump (14) to over 20 years in a patient with a painless 15-centimetre ulcerating mass that had been slowly increasing in size (15).

Pathological findings

The size of the MAME lesion was reported in 129 cases, ranging from 4 (3) to 170 mm (16). The average size of all lesions was 35.1 mm with a median of 25 mm. Among the non-metastatic cases, the average size was 29.6 mm. The malignant component of the tumour was reported in 105 cases: in 44.8% (n=47) there were malignant changes in both epithelial and myoepithelial components, in 36.2% (n=38) there were malignant features only in the myoepithelial component, and in 19.0% (n=20) of cases, only the epithelial component demonstrated features of malignancy.

Of the 121 cases with increased mitotic activity, 89 reported numerical mitotic counts. Among these 89 the average mitotic rate was 11.4 figures per 10 high power fields (HPF) with a median of 10, ranging from 1 to 62 figures per 10 HPF (13). Three cases reported “rare” mitotic figures (97), one case reported “low” (17), and one case reported “few” (18). Only one case was reported to have no mitotic figures (19). Of these 89 cases, 13 were in metastatic patients and 76 were in non-metastatic patients. The average reported mitotic rate amongst the 76 non-metastatic cases was 10.9 figures per 10 HPF.

Ki-67 proliferation index was reported for 49 cases, one of which did not specify a number. The average Ki-67 was 36.3%, with a median of 30%, ranging from 10% (3,15,20,21) to 90% (22). For the 41 non-metastatic cases, the average Ki-67 was 35.1%.

Reporting of hormone receptor status was variable with estrogen receptor (ER), progesterone receptor (PR) and human epidermal receptor 2 (HER2) status reported in only 93 of 167 cases (55.7%). Among these, 34 (36.6%) were triple negative, 35 (37.6%) were ER-negative, 24 (25.8%) were ER-positive, and 5 (5.4%) were HER2-positive. All 5 HER2-positive cases were ER/PR-negative. Overall, triple-negative and ER-negative subtypes predominated, accounting for 74.2% (69 of 93) of reported cases.

Management

Table 4 details the initial surgical management that was reported. One hundred and twenty-nine patients underwent surgery, but 13 patients (10%) underwent surgery without specified detail. 62 of the 129 patients (48.1%) underwent breast conservation surgery (BCS) as their primary intervention which included management reported as wide local excision, lumpectomy, quadrantectomy and partial mastectomy. BCS was followed immediately by either total mastectomy or radical mastectomy in an additional 14 cases (10.9%). Forty patients (31%) had an upfront total or radical mastectomy. An additional 10 patients who had BCS initially, underwent a mastectomy or radical mastectomy for local recurrence (LR), bringing the total number of cases undergoing mastectomy to 64 (49.6%).

Tumour margins were reported in 51 cases, of which 98% (n=50) had negative margins. One case reported a positive margin for a patient that later developed LR without metastasis (23). Ten cases had a margin of 2 mm or less, and of these, three had LR (3,24,25) and two developed metastatic disease (3,25). LN management was described in 92 cases. Node status was reported in 80 of these cases and 15% of these (n=12) had LN involvement. Four cases had nodal involvement without distant metastatic disease.

There was only one reported use of neoadjuvant therapy in a 34-year-old female who received adriamycin and cyclophosphamide prior to mastectomy but later developed lung metastases during follow-up at 18 months (3). Adjuvant chemotherapy was administered in 12 cases. The most common regimen, reported in four cases, was adriamycin and cyclophosphamide (3,11,12). Other regimens included one case of fluorouracil with epirubicin and cyclophosphamide (26) and one case of epirubicin with cyclophosphamide and docetaxel (25). Six cases underwent adjuvant chemotherapy but did not specify the regimen (7,27-30). One patient (31) underwent palliative chemotherapy using cyclophosphamide with methotrexate and fluorouracil but did not respond, leading to the addition of local radiotherapy and adriamycin. Endocrine therapy was described in 10 cases, seven of which had ER-positive tumours. One case involved a triple negative tumour (3) and two did not specify hormone receptor status (32,33).

Sub analysis on metastatic cases

A sub-analysis of patients with metastatic MAME was performed. The clinicopathological features and management of this cohort are summarised in Table 5. A total of 35 patients with metastatic MAME were found. The average age at presentation amongst these cases was 58.8 years old, ranging 20 (12) to 91 years old (7).

Time to first diagnosis of metastatic disease was reported in 28 cases, with an average time from first encounter to diagnosis of metastatic disease of 29.3 months (median 24, range, 0.8–144 months). The average follow-up for metastatic cases where it was reported (n=34) was 38.9 months (range, 3–162 months). Mortality status was available for 30 cases, with death reported in 66.7% (n=20). The average time to death was 34.9 months among the 16 cases which reported the timing, ranging from 3 (34) to 154 months (31).

The size of the primary lesion was reported in 30 cases, with an average size of 53.4 mm, median of 40 mm and range of 10 (7,35) to 170 mm (16). The malignant component of the MAME lesion was reported in 25 patients. Of these, 14 (56%) had malignancy in both myoepithelial and epithelial components, 8 (32%) in the myoepithelial component alone and 3 (12%) in the epithelial component alone.

Most patients with metastatic disease had tumours with infiltrative borders (75%, 9 of 12), nuclear atypia (100%, 19 of 19), necrosis (93.8%, 15 of 16) and nuclear pleomorphism (100%, 8 of 8). All 22 cases which commented on mitotic figures reported their presence but only 13 reported a numeric count. Of these 13 there was an average mitotic rate of 14.6 figures per 10 HPF, ranging from 4 (33) to 45 figures per 10 HPF (36). The Ki-67 proliferation index was reported in 20% of cases (n=7), averaging 43.4% and ranging from 20% (26) to 90% (36). Hormone status was reported in 20 of 35 cases (57.1%). Among these, 6 cases (30%) were triple-negative, 8 were ER-negative (40%), and 6 were ER-positive (30%). HER2 status was described in 11 cases, with 2 being HER2-positive, both of which were ER/PR-negative.

Table 6 shows the site and frequency of distant metastases. LN surgery was reported in 24 patients—2 had sentinel lymph node biopsy (SLNB), 17 had axillary clearance and 5 did not undergo axillary sampling. 2 metastatic cases with LN involvement did not report on the method of LN sampling (37,38). The LN status was reported in 25 patients, of which 6 cases (24%) showed positive LN involvement (12,16,25,37-39).

Sub analysis of local recurrence

A sub analysis was performed for the 29 patients with LR. The average age of all patients with LR was 57.7 years old (median 58, range, 20–84 years old) (12,39). Time to first recurrence was described in 26 cases with an average time of 20.2 months (median 6, range, 1–180 months) (23,24). Table 7 lists the 13 patients who had LR, but no associated metastasis. Among these 13, the two cases which provided immunohistochemistry data were ER-positive but PR/HER2-negative (39) and triple negative (24). Of the 11 cases (5,9,23,24,40-43) providing information on malignant component, 6 (54.5%) showed proliferation in both the myoepithelial and epithelial components, 3 (27.3%) in the myoepithelial alone and 2 (18.2%) in the epithelial alone. Four patients experienced a second recurrence without metastasis (24,41,42). The average follow-up in patients who had LR without metastasis was 48 months, with no reported deaths.

Discussion

Key findings

This systematic review analysed 167 cases of MAME of the breast and represents the most comprehensive synthesis to date. The average size of MAME was 35.1 mm, which is consistent with 87.5% (84 of 96) of patients presenting with a clinically appreciable mass and only a few asymptomatic patients being identified during routine imaging (10.4%, 10 of 96). The largest reported MAME was 170 mm, in a 54-year-old female who presented with a 6-month history of a palpable breast mass. She had axillary LN involvement and died after 6 months with multiple bone metastases (16).

Most MAMEs involved proliferation of either both the myoepithelial and epithelial components (44.8%, 47 of 105) or the myoepithelial component alone (36.2%, 38 of 105). Only a minority involved the epithelial component alone (19%, 20 of 105). A considerable proportion of the cases were ER-negative (37.6%, 35 of 93), or triple-negative (36.6%, 34 of 93). Given that ER-negative and triple-negative breast cancers are typically more aggressive and associated with a higher likelihood of metastasis (98,99), this finding suggests that the aggressive behaviour observed in MAME, including the 35 cases of metastatic disease, could be partly linked to these hormone receptor characteristics.

In patients with metastatic MAME, the average tumour size was 53.4 mm, compared to 29.6 mm in the rest of the cohort, suggesting that larger tumours are associated with increased risk of metastasis. The average mitotic rate and Ki-67 in metastatic cases was 14.6 figures per 10 HPF and 43.4% respectively, compared to 10.9 figures per 10 HPF and 35.1% in non-metastatic cases. This may indicate that higher mitotic and Ki-67 proliferation rates may be associated with a greater risk of metastasis, however more data is required to draw a meaningful conclusion as only seven metastatic patients had reported Ki-67 figures (3,18,22,25,26,29,31).

Of the 29 patients with LR, 16 also had metastatic disease. The average time to LR and metastasis was 20.2 and 29.3 months from time of diagnosis respectively, suggesting that these events tend to occur within the commonly recommended 5-year clinical and radiological surveillance of breast cancer (100,101).

LN involvement was reported in 12 cases (5,12,16,25,27,37-39,44-46), and a minority of the total patient cohort had LVI (8.3%) or PNI (22.2%). This supports the theory that MAME spreads haematogeneously (18,20,47,48). There are currently many publications which suggest SLNB may not be required (5,7). However, despite only 25 metastatic cases reporting on LN status, LN involvement was higher in patients with metastatic disease, present in 24% of the cases (6 of 25). As there were only 12 cases of LN involvement in the study, this means that 50% (6 of 12) of cases with LN involvement also had metastatic disease. Of the six cases without metastatic disease (5,27,37,44-46), one patient had LR (5).

The histological component which undergoes malignant transformation may influence the biological behaviour of MAME. In 10 of the 12 cases with LN involvement, malignant proliferation involved both myoepithelial and epithelial components (n=7) or myoepithelial component alone (n=3), with no cases affecting epithelial proliferation alone. Similarly, of the cases with LR which reported on the malignant component, 52.4% (11 of 21) reported proliferation in both components: 33.3% (7 of 21) in the myoepithelial component alone and 14.3% (3 of 21) in the epithelial component alone. In the 35 cases who had metastatic disease and reported the malignant component, 56% (14 of 25) had proliferation in both components, 32% (8 of 25) in the myoepithelial component alone and only 12% (3 of 25) in the epithelial component alone. This suggests that tumours with either combined or isolated myoepithelial proliferation may be more aggressive, indicating the potential need for SLNB or closer surveillance in such cases.

Histopathological diagnostic modality was reported in 45.5% (76 of 167) of cases, with most undergoing FNA, CNB or surgical biopsy. Of the reported cases, 46.6% (n=34) had a pre-operative differential diagnosis suggestive of malignancy, but only 8.2% (n=6) of these were diagnosed as MAME. In contrast, 53.4% (n=39) of MAME cases had a pre-operative diagnosis of benign or atypical lesions. This highlights the diagnostic challenge in differentiating between benign, atypical, and malignant AME on limited sampling, which underscores the importance of excisional biopsy due to the lesion’s morphological heterogeneity.

Strengths and limitations

This is the first systematic review published on MAME and fills a significant dearth in the literature by providing a comprehensive synthesis of existing data to provide insight for healthcare providers managing patients with this rare disease. However, many publications had incomplete or unreported data which introduces limitations to our study. The retrospective, anecdotal, and heterogeneous nature of many of the case reports impacts generalisability and may introduce publication and selection bias, as only unusual cases are typically published. In an area where high-quality data is limited, however, excluding these may have led to the loss of valuable insights.

For this reason, we have refrained from a statistical analysis or drawing confident conclusions. We encourage case reports and case series to be published in accordance with the CARE (102) and PROCESS (103) guidelines respectively to ensure transparency and standardisation of quality for better usefulness.

Comparison with similar research

Haque et al. conducted a large analysis of 110 patients with MAME or myoepithelioma using data from the National Cancer Database in the United States (104). Unfortunately, the data from this analysis could not be included in our systematic review due to the inclusion of malignant myoepithelioma. Compared to our findings, Haque’s cohort had a larger proportion of patients who underwent adjuvant chemotherapy and radiotherapy. In our study, adjuvant chemotherapy and radiotherapy were only reported in 12 and 25 cases respectively, compared to Haque’s population where 29 patients underwent chemotherapy, 40 patients underwent adjuvant radiotherapy, and 13 received both. Despite this, Haque et al. concluded that neither surgical technique, radiotherapy nor chemotherapy was associated with improved overall survival in their population.

Explanation of findings

The predominance of ER-negative and triple negative disease may explain the relatively high rates of recurrence and metastasis, as these subtypes are associated with more aggressive behaviour (45,46). Similarly, the association between larger tumour size, higher mitotic rate, and elevated Ki-67 with metastatic disease suggests a biologically aggressive tumour phenotype. The finding that malignant transformation of the myoepithelial component (alone or in combination) correlates with worse outcomes may reflect differences in tumour biology that are not yet fully understood. Difficulty in diagnosing MAME pre-operatively stems from its morphological overlap with benign adenomyoepithelioma and other biphasic tumours. This emphasises the need for improved awareness among clinicians and pathologists and highlights the limitations of core needle and fine needle biopsies in this context.

Implications and actions needed

Our findings suggest that excisional biopsy should be strongly considered when AME is suspected, even if initial FNA or CNB suggests benign pathology. Given the tendency for tumours with combined or myoepithelial malignant proliferation to behave more aggressively, wider excision margins, LN assessment, and closer surveillance may be appropriate in such cases.

Most MAME cases do not express hormone receptors, limiting the role of endocrine therapy. Although LN involvement is rare, it was associated with a higher risk of metastasis, suggesting that LN evaluation may still be warranted, especially in larger or more aggressive tumours.

Further research, including prospective registries and high-quality case series, is needed to improve understanding of MAME and develop evidence-based management strategies. In the meantime, heightened clinical suspicion and careful pathological assessment are essential to guide management.

Conclusions

MAME of the breast is an extremely rare tumour and there is limited understanding of its biological behaviour. Most cases occur in post-menopausal females with a median age of 61.1 years. Pre-operative diagnosis of MAME is challenging, as is differentiating between malignant and benign AME. Excisional biopsy is recommended due to the pathological heterogeneity and the limitations of pre-operative biopsies. Most MAME cases do not express hormone receptors. LN involvement is rare but may be associated with higher risk of metastatic disease. Larger tumours may also be associated with higher risk of metastasis. Tumours with both myoepithelial and epithelial proliferation, or myoepithelial proliferation alone, may be more aggressive.

Acknowledgments

Our abstract has been accepted for a poster presentation at the Australasian Society for Breast Disease 14th Scientific Meeting, to be held on Broadbeach, Gold Coast, Australia, from October 9th to 11th, 2025.

Footnote

Reporting Checklist: The authors have completed the PRISMA reporting checklist. Available at https://abs.amegroups.com/article/view/10.21037/abs-25-4/rc

Peer Review File: Available at https://abs.amegroups.com/article/view/10.21037/abs-25-4/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://abs.amegroups.com/article/view/10.21037/abs-25-4/coif). The authors have no conflicts of interest to declare.

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.

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 non-commercial 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). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Hamperl H. The myothelia (myoepithelial cells). Normal state; regressive changes; hyperplasia; tumors. Curr Top Pathol 1970;53:161-220.
2. Ahmadi N, Negahban S, Aledavood A, et al. Malignant adenomyoepithelioma of the breast: a review. Breast J 2015;21:291-6. [Crossref] [PubMed]
3. Alqudaihi HMA, Lee SB, Son BH, et al. Clinicopathological characteristics and outcomes of malignant adenomyoepithelioma of the breast: a single institution's experience. World J Surg Oncol 2022;20:128. [Crossref] [PubMed]
4. Rakha E, Tan PH, Ellis I, et al. Adenomyoepithelioma of the breast: a proposal for classification. Histopathology 2021;79:465-79. [Crossref] [PubMed]
5. Tavassoli FA. Myoepithelial lesions of the breast. Myoepitheliosis, adenomyoepithelioma, and myoepithelial carcinoma. Am J Surg Pathol 1991;15:554-68. [Crossref] [PubMed]
6. Tavassoli FA. Peritoneal tumours. Pathology and Genetics of Tumours of the Breast and Female Genital Organs. IARC; 2003:197-202.
7. Hayes MM. Adenomyoepithelioma of the breast: a review stressing its propensity for malignant transformation. J Clin Pathol 2011;64:477-84. [Crossref] [PubMed]
8. Moritz AW, Wiedenhoefer JF, Profit AP, et al. Breast adenomyoepithelioma and adenomyoepithelioma with carcinoma (malignant adenomyoepithelioma) with associated breast malignancies: A case series emphasizing histologic, radiologic, and clinical correlation. Breast 2016;29:132-9. [Crossref] [PubMed]
9. Oda G, Nakagawa T, Mori M, et al. Adenomyoepithelioma of the breast with malignant transformation and repeated local recurrence: A case report. World J Clin Cases 2021;9:8864-70. [Crossref] [PubMed]
10. Page MJ, McKenzie JE, Bossuyt PM, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ 2021;372: [Crossref] [PubMed]
11. Gafton B, Scripcariu V, Prutianu I, et al. Challenges in management of male breast adenomioepithelioma with malignant behavior: Case report. Medicine (Baltimore) 2019;98:e17587. [Crossref] [PubMed]
12. Suresh Attili VS, Saini K, Lakshmaiah KC, et al. Malignant adenomyoepithelioma of the breast. Indian Journal of Surgery 2007;69:14-6.
13. Hungermann D, Buerger H, Oehlschlegel C, et al. Adenomyoepithelial tumours and myoepithelial carcinomas of the breast--a spectrum of monophasic and biphasic tumours dominated by immature myoepithelial cells. BMC Cancer 2005;5:92. [Crossref] [PubMed]
14. Yang Y, Wang Y, He J, et al. Malignant adenomyoepithelioma combined with adenoid cystic carcinoma of the breast: a case report and literature review. Diagn Pathol 2014;9:148. [Crossref] [PubMed]
15. Choi SY, Kim JS, Kim SJ, et al. Malignant adenomyoepithelioma of the breast presenting as a large mass that grew slowly without metastasis. Journal of Breast Cancer 2009;12:219-22.
16. Chen PC, Chen CK, Nicastri AD, et al. Myoepithelial carcinoma of the breast with distant metastasis and accompanied by adenomyoepitheliomas. Histopathology 1994;24:543-8. [Crossref] [PubMed]
17. Samanta DR, Senapati SN, Sharma PK, et al. Adenomyoepithelioma of the breast. Hematol Oncol Stem Cell Ther 2009;2:364-6. [Crossref] [PubMed]
18. Moro K, Sakata E, Nakahara A, et al. Malignant adenomyoepithelioma of the breast. Surg Case Rep 2020;6:118. [Crossref] [PubMed]
19. Hempenstall LE, Saxena M, Donaldson E. Malignant adenomyoepithelioma with multifocal adenosquamous carcinoma of the breast: A case report. Breast J 2019;25:731-2. [Crossref] [PubMed]
20. Jones M, Fletcher J. Malignant adenomyoepithelioma of the breast. Pathology 2017;49:322-5. [Crossref] [PubMed]
21. Petrozza V, Pasciuti G, Pacchiarotti A, et al. Breast adenomyoepithelioma: a case report with malignant proliferation of epithelial and myoepithelial elements. World J Surg Oncol 2013;11:285. [Crossref] [PubMed]
22. Lee S, Oh SY, Kim SH, et al. Malignant Adenomyoepithelioma of the Breast and Responsiveness to Eribulin. J Breast Cancer 2015;18:400-3. [Crossref] [PubMed]
23. Rasbridge SA, Millis RR. Adenomyoepithelioma of the breast with malignant features. Virchows Arch 1998;432:123-30. [Crossref] [PubMed]
24. Ha MI, Seo BK, Choi JW. Rapid Local Recurrence of Breast Myoepithelial Carcinoma Arising in Adenomyoepithelioma: A Case Report. Taehan Yongsang Uihakhoe Chi 2020;81:207-12. [Crossref] [PubMed]
25. Yuan Z, Qu X, Zhang ZT, et al. Lessons From Managing the Breast Malignant Adenomyoepithelioma and the Discussion on Treatment Strategy. World J Oncol 2017;8:126-31. [Crossref] [PubMed]
26. Maffini F, Renne G, Olivadese R, et al. A rare case of lung metastasis from a malignant adenomyoepithelioma of the breast: histological features and therapeutic implications. Ecancermedicalscience 2013;7:372. [Crossref] [PubMed]
27. Baum JE, Sung KJ, Tran H, et al. Mammary Epithelial-Myoepithelial Carcinoma: Report of a Case With HRAS and PIK3CA Mutations by Next-Generation Sequencing. Int J Surg Pathol 2019;27:441-5. [Crossref] [PubMed]
28. Foschini MP, Pizzicannella G, Peterse JL, et al. Adenomyoepithelioma of the breast associated with low-grade adenosquamous and sarcomatoid carcinomas. Virchows Arch 1995;427:243-50. [Crossref] [PubMed]
29. Honda Y, Iyama K. Malignant adenomyoepithelioma of the breast combined with invasive lobular carcinoma. Pathol Int 2009;59:179-84. [Crossref] [PubMed]
30. Joyon N, Guillaume Z, Ouafi L, et al. Malignant Adenomyoepithelioma of the Breast: An Unexpected Malignancy in a Lynch Syndrome Patient. Int J Surg Pathol 2023;31:572-5. [Crossref] [PubMed]
31. Bult P, Verwiel JM, 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. Virchows Arch 2000;436:158-66. [Crossref] [PubMed]
32. Simpson RH, Cope N, Skálová A, et al. Malignant adenomyoepithelioma of the breast with mixed osteogenic, spindle cell, and carcinomatous differentiation. Am J Surg Pathol 1998;22:631-6. [Crossref] [PubMed]
33. Jones C, Tooze R, Lakhani SR. Malignant adenomyoepithelioma of the breast metastasizing to the liver. Virchows Arch 2003;442:504-6. [Crossref] [PubMed]
34. Kihara M, Yokomise H, Irie A, et al. Malignant adenomyoepithelioma of the breast with lung metastases: report of a case. Surg Today 2001;31:899-903. [Crossref] [PubMed]
35. Lubin D, Toorens E, Zhang PJ, et al. Adenomyoepitheliomas of the Breast Frequently Harbor Recurrent Hotspot Mutations in PIK3-AKT Pathway-related Genes and a Subset Show Genetic Similarity to Salivary Gland Epithelial-Myoepithelial Carcinoma. Am J Surg Pathol 2019;43:1005-13. [Crossref] [PubMed]
36. Fang ZM, Tse RV, Marjoniemi VM, et al. Radioresistant malignant myoepithelioma of the breast with high level of ataxia telangiectasia mutated protein. J Med Imaging Radiat Oncol 2009;53:234-9. [Crossref] [PubMed]
37. Ginter PS, McIntire PJ, Kurtis B, et al. Adenomyoepithelial tumors of the breast: molecular underpinnings of a rare entity. Mod Pathol 2020;33:1764-72. [Crossref] [PubMed]
38. Oka K, Sando N, Moriya T, et al. Malignant adenomyoepithelioma of the breast with matrix production may be compatible with one variant form of matrix-producing carcinoma: a case report. Pathol Res Pract 2007;203:599-604. [Crossref] [PubMed]
39. Bièche I, Coussy F, El-Botty R, et al. HRAS is a therapeutic target in malignant chemo-resistant adenomyoepithelioma of the breast. J Hematol Oncol 2021;14:143. [Crossref] [PubMed]
40. Cameron HM, Hamperl H, Warambo W. Leiomyosarcoma of the breast originating from myothelium (myoepithelium). J Pathol 1974;114:89-92. [Crossref] [PubMed]
41. Pauwels C, De Potter C. Adenomyoepithelioma of the breast with features of malignancy. Histopathology 1994;24:94-6. [Crossref] [PubMed]
42. Qureshi A, Kayani N, Gulzar R. Malignant adenomyoepithelioma of the breast: a case report with review of literature. BMJ Case Rep 2009;2009:bcr01.2009.1442.
43. Sugano I, Nagao T, Tajima Y, et al. Malignant adenomyoepithelioma of the breast: a non-tubular and matrix-producing variant. Pathol Int 2001;51:193-9. [Crossref] [PubMed]
44. Logie N, Hugh J, Paulson K, et al. Radiotherapy in the Multidisciplinary Management of Adenomyoepithelioma of the Breast with an Axillary Lymph Node Metastasis: A Case Report and Review of the Literature. Cureus 2017;9:e1380. [Crossref] [PubMed]
45. Awamleh AA, Gudi M, Shousha S. Malignant adenomyoepithelioma of the breast with lymph node metastasis: a detailed immunohistochemical study. Case Rep Pathol 2012;2012:305858. [Crossref] [PubMed]
46. Damiani S, Riccioni L, Pasquinelli G, et al. Poorly differentiated myoepithelial cell rich carcinoma of the breast. Histopathology 1997;30:542-8. [Crossref] [PubMed]
47. Kim MJ, Kim CS, Ju MJ, et al. Malignant adenomyoepithelioma of the breast: A rare case report. Int J Surg Case Rep 2019;59:111-4. [Crossref] [PubMed]
48. Howlett DC, Mason CH, Biswas S, et al. Adenomyoepithelioma of the breast: spectrum of disease with associated imaging and pathology. AJR Am J Roentgenol 2003;180:799-803. [Crossref] [PubMed]
49. Ahmed AA, Heller DS. Malignant adenomyoepithelioma of the breast with malignant proliferation of epithelial and myoepithelial elements: a case report and review of the literature. Arch Pathol Lab Med 2000;124:632-6. [Crossref] [PubMed]
50. Antonelli MS, Mallel G, Pecoraro A, et al. Adenomyoepithelioma of the breast: case report and literature review. G Chir 2018;39:255-7.
51. Baraban E, Zhang PJ, Jaffer S, et al. MYB rearrangement and immunohistochemical expression in adenomyoepithelioma of the breast: a comparison with adenoid cystic carcinoma. Histopathology 2018;73:897-903. [Crossref] [PubMed]
52. Bojja VKR, Bipte S, Reelkar AV, et al. Malignant Adenomyoepithelial Tumour of the Breast - a Rare Diagnosis - Case Report. Indian J Surg Oncol 2023;14:18-20. [Crossref] [PubMed]
53. Bui CM, Bose S. Primary mucoepidermoid carcinoma of the breast arising in adenomyoepithelioma. BMJ Case Rep 2022;15:e247281. [Crossref] [PubMed]
54. Chen F, Feng H, Wu H, et al. Malignant adenomyoepithelioma of the breast with contralateral apocrine ductal carcinoma in situ: A rare case. J Clin Ultrasound 2023;51:857-9. [Crossref] [PubMed]
55. Cheung KL, Wong AW, Parker H, et al. Pathological features of primary breast cancer in the elderly based on needle core biopsies--a large series from a single centre. Crit Rev Oncol Hematol 2008;67:263-7. [Crossref] [PubMed]
56. Ericson-Lindquist K, Johansson A, Levéen P, et al. Targeted sequencing may facilitate differential diagnostics of pulmonary tumours: a case series. Diagn Pathol 2017;12:31. [Crossref] [PubMed]
57. Fan F, Smith W, Wang X, et al. Myoepithelial carcinoma of the breast arising in an adenomyoepithelioma: mammographic, ultrasound and histologic features. Breast J 2007;13:203-4. [Crossref] [PubMed]
58. Gandhi JS, Mehta A, Gupta G, et al. Malignant adenomyoepithelioma: a tumor of low malignant potential despite basal phenotype. Indian J Pathol Microbiol 2011;54:230-2. [Crossref] [PubMed]
59. Grodecka-Gazdecka S, Graja T, Kurzawa P. Rare malignacies of the breast diagnosed and treated in the year 2001 at the Chair and Clinic of Oncology of the Poznan University of Medical Sciences. Nowotwory. Journal of Oncology 2004;54:34-40.
60. Han B, Mori I, Nakamura M, et al. Myoepithelial carcinoma arising in an adenomyoepithelioma of the breast: case report with immunohistochemical and mutational analysis. Pathol Int 2006;56:211-6. [Crossref] [PubMed]
61. Harigopal M, Park K, Chen X, et al. Pathologic quiz case: a rapidly increasing breast mass in a postmenopausal woman. Malignant adenomyoepithelioma. Arch Pathol Lab Med 2004;128:235-6. [Crossref] [PubMed]
62. Hegyi L, Thway K, Newton R, et al. Malignant myoepithelioma arising in adenomyoepithelioma of the breast and coincident multiple gastrointestinal stromal tumours in a patient with neurofibromatosis type 1. J Clin Pathol 2009;62:653-5. [Crossref] [PubMed]
63. Jameel Z, Kiluk J, Rosa M. Malignant Adenomyoepithelioma of the Breast and Associated Epithelial-Myoepithelial Carcinoma; A Rare Case Report. Int J Surg Pathol 2022;30:569-73. [Crossref] [PubMed]
64. Kakkar A, Jangra K, Kumar N, et al. Epithelial-myoepithelial carcinoma of the breast: A rare type of malignant adenomyoepithelioma. Breast J 2019;25:1273-5. [Crossref] [PubMed]
65. Khurana A, Jalpota Y. Myoepithelial carcinoma arising in an adenomyoepithelioma of the breast: a case report of a rare entity. Indian J Pathol Microbiol 2010;53:310-2. [Crossref] [PubMed]
66. Kiaer H, Nielsen B, Paulsen S, et al. Adenomyoepithelial adenosis and low-grade malignant adenomyoepithelioma of the breast. Virchows Arch A Pathol Anat Histopathol 1984;405:55-67. [Crossref] [PubMed]
67. Korolczuk A, Amarowicz M, Bąk K, et al. Adenomyoepithelioma of the breast with late pulmonary metastases - case report and review of the literature. J Cardiothorac Surg 2016;11:121. [Crossref] [PubMed]
68. Kurashina M. Fine-needle aspiration cytology of benign and malignant adenomyoepithelioma: report of two cases. Diagn Cytopathol 2002;26:29-34. [Crossref] [PubMed]
69. Leung TK, Chu JS, Huang PJ, et al. Breast MRI for monitoring images of an "adenomyoepithelioma with malignant features", before, during, and after chemotherapy. Breast J 2010;16:652-3. [Crossref] [PubMed]
70. Loose JH, Patchefsky AS, Hollander IJ, et al. Adenomyoepithelioma of the breast. A spectrum of biologic behavior. Am J Surg Pathol 1992;16:868-76. [Crossref] [PubMed]
71. Luo X, She J, Xu T, et al. Establishment and characterization of organoids from a patient with adenomyoepithelioma of the breast. Bioengineered 2021;12:11578-85. [Crossref] [PubMed]
72. Marian C, Boila A, Soanca D, et al. Malignant transformation of adenomyoepithelioma of the breast by a monophasic population: a report of two cases and review of literature. APMIS 2013;121:272-9. [Crossref] [PubMed]
73. Michal M, Baumruk L, Burger J, et al. Adenomyoepithelioma of the breast with undifferentiated carcinoma component. Histopathology 1994;24:274-6. [Crossref] [PubMed]
74. Noël JC, Simon P, Aguilar SF. Malignant myoepithelioma arising in cystic adenomyoepithelioma. Breast J 2006;12:386. [Crossref] [PubMed]
75. Nomura K, Fukunaga M, Uchida K, et al. Adenomyoepithelioma of the breast with exaggerated proliferation of epithelial cells: report of a case. Pathol Int 1996;46:1011-4. [Crossref] [PubMed]
76. Parikh P, Jameel Z, Falcon S, et al. Adenomyoepithelioma of the breast: Case series and literature review. Clin Imaging 2021;75:157-64. [Crossref] [PubMed]
77. Reis-Filho JS, Fulford LG, Crebassa B, et al. Collagenous spherulosis in an adenomyoepithelioma of the breast. J Clin Pathol 2004;57:83-6. [Crossref] [PubMed]
78. Robinson A, Mayne J, Boroumand N, et al. Malignant adenomyoepithelioma: a rare entity. Breast J 2014;20:94-6. [Crossref] [PubMed]
79. Spyrou A, Katsourakis A, Chytas D, et al. Combination of Adenomyoepithelioma and Adenoid Cystic Carcinoma of the Breast: A Case Report of an Uncommon Histopathological Entity. Am J Case Rep 2022;23:e934391. [Crossref] [PubMed]
80. Trojani M, Guiu M, Trouette H, et al. Malignant adenomyoepithelioma of the breast. An immunohistochemical, cytophotometric, and ultrastructural study of a case with lung metastases. Am J Clin Pathol 1992;98:598-602. [Crossref] [PubMed]
81. Uchida M, Gatica C, Hasson D, et al. Breast adenomyoepithelioma from a radiologic perspective. Radiologia (Engl Ed) 2022;64:37-43. [Crossref] [PubMed]
82. Van Dorpe J, De Pauw A, Moerman P. Adenoid cystic carcinoma arising in an adenomyoepithelioma of the breast. Virchows Arch 1998;432:119-22. [Crossref] [PubMed]
83. Van Hoeven KH, Drudis T, Cranor ML, et al. Low-grade adenosquamous carcinoma of the breast. A clinocopathologic study of 32 cases with ultrastructural analysis. Am J Surg Pathol 1993;17:248-58. [Crossref] [PubMed]
84. Watanabe S, Otani T, Iwasa T, et al. A Case of Metastatic Malignant Breast Adenomyoepithelioma With a Codon-61 Mutation of HRAS. Clin Breast Cancer 2019;19:e589-92. [Crossref] [PubMed]
85. Wiens N, Hoffman DI, Huang CY, et al. Clinical characteristics and outcomes of benign, atypical, and malignant breast adenomyoepithelioma: a single institution's experience. Am J Surg 2020;219:651-4. [Crossref] [PubMed]
86. Xu J, Tang X, Iida Y, et al. Adenomyoepithelioma with carcinoma of the breast: A report of two cases and a review of the literature. Pathol Res Pract 2016;212:130-4. [Crossref] [PubMed]
87. Zhang L, Qin G, He Z, et al. The mammography and MRI manifestations of adenomyoepithelioma of the breast. Clin Radiol 2016;71:235-43. [Crossref] [PubMed]
88. Zhang Z, Wang Y, Xie X, et al. Malignant adenomyoepithelioma of the breast: A case report. Medicine (Baltimore) 2021;100:e24461. [Crossref] [PubMed]
89. Zizi-Sermpetzoglou A, Vasilakaki T, Grammatoglou X, et al. Malignant adenomyoepithelioma of the breast--case report. Eur J Gynaecol Oncol 2009;30:234-6.
90. Febres-Aldana CA, Mejia-Mejia O, Krishnamurthy K, et al. Malignant transformation in a Breast Adenomyoepithelioma Caused by Amplification of c-MYC: A Common pathway to Cancer in a Rare Entity. J Breast Cancer 2020;23:93-9. [Crossref] [PubMed]
91. Ito R, Ota D, Ando S, et al. A case of adenomyoepithelioma with myoepithelial carcinoma of the breast. Clin Case Rep 2019;7:930-4. [Crossref] [PubMed]
92. Lari EA, Lari AA, Alsaeed T. Malignant adenomyoepithelioma of the breast: A case report. Int J Surg Case Rep 2020;72:56-8. [Crossref] [PubMed]
93. Rossetti C, Azzalis LA, Junqueira VBC, et al. Adenomyoepithelioma with malignant transformation diagnosed by immunohistochemical analysis: case report. Jornal Brasileiro de Patologia e Medicina Laboratorial 2011;47.
94. Shah AK, Venkatesan S, Katoch R, et al. Epithelial-Myoepithelial Carcinoma Ex Pleomorphic Adenoma in Breast: An Exotic Rarity. Med J Armed Forces India 2011;67:74-6. [Crossref] [PubMed]
95. Takahashi I I, Tashiro H, Wakasugi K, et al. Malignant Adenomyoepithelioma of the Breast:A Case with Distant Metastases. Breast Cancer 1999;6:73-7. [Crossref] [PubMed]
96. Kalyani R, Srinivas Murthy V. Malignant adenomyoepithelioma of breast masquerading as soft tissue lytic lesion of right iliac bone: a rare entity. J Clin Diagn Res 2014;8:FD03-4. [Crossref] [PubMed]
97. Foschini MP, Krausz T. Salivary gland-type tumors of the breast: a spectrum of benign and malignant tumors including "triple negative carcinomas" of low malignant potential. Semin Diagn Pathol 2010;27:77-90. [Crossref] [PubMed]
98. Anders C, Carey LA. Understanding and treating triple-negative breast cancer. Oncology (Williston Park) 2008;22:1233-9; discussion 1239-40, 1243.
99. Aysola K, Desai A, Welch C, et al. Triple Negative Breast Cancer - An Overview. Hereditary Genet 2013;2013:001.
100. Runowicz CD, Leach CR, Henry NL, et al. American Cancer Society/American Society of Clinical Oncology Breast Cancer Survivorship Care Guideline. J Clin Oncol 2016;34:611-35. [Crossref] [PubMed]
101. De Wilde RL, Devassy R, Torres-de la Roche LA, et al. Guidance and Standards for Breast Cancer Care in Europe. J Obstet Gynaecol India 2020;70:330-6. [Crossref] [PubMed]
102. Riley DS, Barber MS, Kienle GS, et al. CARE guidelines for case reports: explanation and elaboration document. J Clin Epidemiol 2017;89:218-35. [Crossref] [PubMed]
103. Agha RA, Sohrabi C, Mathew G, et al. The PROCESS 2020 Guideline: Updating Consensus Preferred Reporting Of CasESeries in Surgery (PROCESS) Guidelines. Int J Surg 2020;84:231-5. [Crossref] [PubMed]
104. Haque W, Verma V, Suzanne Klimberg V, et al. Clinical presentation, national practice patterns, and outcomes of breast adenomyoepithelioma. Breast J 2020;26:653-60. [Crossref] [PubMed]