Case Reports in Pathology

Case Reports in Pathology / 2012 / Article

Case Report | Open Access

Volume 2012 |Article ID 305858 |

Ahlam A. Awamleh, Mihir Gudi, Sami Shousha, "Malignant Adenomyoepithelioma of the Breast with Lymph Node Metastasis: A Detailed Immunohistochemical Study", Case Reports in Pathology, vol. 2012, Article ID 305858, 4 pages, 2012.

Malignant Adenomyoepithelioma of the Breast with Lymph Node Metastasis: A Detailed Immunohistochemical Study

Academic Editor: J. Kulka
Received26 Sep 2011
Accepted19 Oct 2011
Published18 Jan 2012


Malignant adenomyoepithelioma of the breast is a rare tumour with around 30 cases reported in the literature. Metastases associated with these tumours are usually haematogenous. Axillary lymph node metastases are thought to be unusual, and it has been recently suggested that axillary node dissection is not indicated unless clinically palpable. We here present a case of a 63-year-old woman, who developed a malignant adenomyoepithelioma with axillary lymph node metastasis, that included epithelial and myoepithelial elements, in spite of the absence of clinically enlarged nodes. We suggest that histological examination of axillary sentinel node(s) or node sampling may be worthwhile in this condition.

Adenomyoepithelioma of the breast is a benign neoplasm characterised by biphasic proliferation of epithelial and myoepithelial cells [1]. It resembles adenomyoepithelioma of salivary glands as first described by Hamperl [2]. Malignant change can occur rarely in one or both cellular components [3] and can be either a pure myoepithelial carcinoma or a combined malignant adenomyoepithelioma [1]. Around 30 cases of malignant adenomyoepithelioma have been reported in the literature [1, 35].

A 63-year-old woman presented with a mass in the left breast. A core biopsy showed intraductal papilloma with atypical hyperplasia (B3). This was removed by wide local excision. Grossly, the biopsy included two small greyish white soft nodules, each measuring 1 cm in diameter. Microscopic examination showed multiple intraductal papillary lesions. In some areas, the papillae were covered by a single layer of epithelial cells with underlying several layers of myoepithelial cells which stained positively for smooth muscle actin, p63, and CD10, appearances which were interpreted as adenomyoepithelioma (Figure 1). The epithelial cells were ER negative and many were CK5 and 14 positive, indicating that they are of basal-like rather than luminal type. Other areas of the lesion consisted of solid proliferation of a mixture of these epithelial and myoepithelial cells and showed abundant mitotic figures and marked nuclear pleomorphism (Figure 2(a)), with evidence of peripheral invasion (Figure 2(b)). The features were interpreted as a malignant adenomyoepithelioma developing in continuity with a benign adenomyoepithelioma. The lesions reached the excision margins and reexcision was recommended.

A mastectomy was carried out with axillary lymph node sampling. Pathological examination showed a partly cystic and partly solid tumour measuring  cm, that showed similar features to those seen previously and consisted of a mixture of epithelial and myoepithelial cells arranged in a benign adenomyoepithelioma fashion in some areas, which merged with areas showing malignant features as those described above with invasion of adjacent breast tissue. One of the two dissected lymph nodes showed a 1.8 mm metastatic focus which was positive for CK8/18, CK19, AE1/AE3, CK5/6, CK14, SMA, and CD10, indicating the presence of epithelial and myoepithelial elements (Figure 3).

Malignant adenomyoepithelioma is generally heralded by a long history of a stable breast mass followed by rapid growth phase [1]. Grossly, the tumour is usually nodular and the nodules may show cystic changes as well as necrosis and foci of calcification. Histological features of malignant transformation include nuclear atypia, increased mitotic activity, necrosis, and infiltrative growth pattern [3, 4]. In our case, all these features were present, except for necrosis, and the dual cellular nature was established by immunohistochemistry in the primary lesion and lymph node metastasis.

Malignant adenomyoepithelioma has the potential for distant metastases. These typically occur in lesions larger than 2 cm [3] and in those with high-grade malignant component [1]. Distal metastasis were described in 8 (32%) out of 25 cases reviewed by two authors [1, 3], involving lungs, brain, and soft tissues. Other authors described metastasis in the liver, bone, thyroid gland, and mediastinal lymph nodes [68].

Axillary lymph node involvement is thought to be unusual, hence a recent review article has suggested that axillary node dissection is not indicated unless there is clinically involved lymph nodes [1]. However, metastases to axillary nodes have been reported in 2 previous cases of malignant adenomyoepithelioma [9, 10], in addition to the current case where no palpable lymph nodes were present. Thus, we would suggest that examination of sentinel node(s) or axillary node sampling may be worthwhile in this condition.


  1. M. M. Hayes, “Adenomyoepithelioma of the breast: a review stressing its propensity for malignant transformation,” Journal of Clinical Pathology, vol. 64, no. 6, pp. 477–484, 2011. View at: Publisher Site | Google Scholar
  2. H. Hamperl, “The myothelia (myoepithelial cells). Normal state; regressive changes; hyperplasia; tumors,” Current Topics in Pathology, vol. 53, pp. 161–220, 1970. View at: Google Scholar
  3. A. A. Ahmed and D. S. Heller, “Malignant adenomyoepithelioma of the breast with malignant proliferation of epithelial and myoepithelial elements: a case report and review of the literature,” Archives of Pathology and Laboratory Medicine, vol. 124, no. 4, pp. 632–636, 2000. View at: Google Scholar
  4. S. Y. Choi, J. S. Kim, S. J. Kim, Y. J. Kim, L. Kim, and U. C. Young, “Malignant adenomyoepithelioma of the breast presenting as a large mass that grew slowly without metastasis,” Journal of Breast Cancer, vol. 12, no. 3, pp. 219–222, 2009. View at: Publisher Site | Google Scholar
  5. A. Khurana and Y. Jalpota, “Myoepithelial carcinoma arising in an adenomyoepithelioma of the breast: a case report of a rare entity,” Indian Journal of Pathology and Microbiology, vol. 53, no. 2, pp. 310–312, 2010. View at: Publisher Site | Google Scholar
  6. I. I. Takahashi, H. Tashiro, K. Wakasugi et al., “Malignant adenomyoepithelioma of the breast: a case with distant metastases,” Breast Cancer, vol. 6, no. 1, pp. 73–77, 1999. View at: Google Scholar
  7. P. Bult, J. M. M. Verwiel, T. Wobbes, M. M. Kooy-Smits, J. Biert, and R. Holland, “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 Archiv, vol. 436, no. 2, pp. 158–166, 2000. View at: Google Scholar
  8. C. Jones, R. Tooze, and S. R. Lakhani, “Malignant adenomyoepithelioma of the breast metastasizing to the liver,” Virchows Archiv, vol. 442, no. 5, pp. 504–506, 2003. View at: Google Scholar
  9. P. C. Chen, C. K. Chen, A. D. Nicastri, and R. B. Wait, “Myoepithelial carcinoma of the breast with distant metastasis and accompanied by adenomyoepitheliomas,” Histopathology, vol. 24, no. 6, pp. 543–548, 1994. View at: Google Scholar
  10. V. S. Suresh Attili, K. Saini, K. C. Lakshmaiah, U. Batra, M. Malathi, and C. Ramachandra, “Malignant adenomyoepithelioma of the breast,” Indian Journal of Surgery, vol. 69, no. 1, pp. 14–16, 2007. View at: Google Scholar

Copyright © 2012 Ahlam A. Awamleh et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Related articles

No related content is available yet for this article.
 PDF Download Citation Citation
 Download other formatsMore
 Order printed copiesOrder

Related articles

No related content is available yet for this article.

Article of the Year Award: Outstanding research contributions of 2021, as selected by our Chief Editors. Read the winning articles.