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Malignant adenomyoepithelioma combined with adenoid cystic carcinoma of the breast: a case report and literature review.

Yang Y, Wang Y, He J, Pan G, Tuo X, Jiang A, Bian L - Diagn Pathol (2014)

Bottom Line: Histologically, the nodule was composed of two obscure lobules.Immunohistochemically, CK18 and P63 highlighted the epithelial and myoepithelial cells respectively in both lesions.The histogenesis of these tumors is also discussed.

View Article: PubMed Central - PubMed

Affiliation: Department of Pathology, The First Affiliated Hospital of Kunming Medical University, 295, Xichang road, Kunming, Yunnan, China. bianli1976@sina.com.

ABSTRACT

Background: 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.

Case presentation: A 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.

Conclusion: This 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 slides: The 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.

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High-power examination to the malignant adenomyoepithelioma. Atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (arrow). HE, ×200.
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Fig3: High-power examination to the malignant adenomyoepithelioma. Atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (arrow). HE, ×200.

Mentions: A 61-year-old female was admitted to The First Affiliated Hospital of Kunming Medical University, Yunnan, China, complaining of a lump in the right breast which had appeared 2 days earlier and had been increasing in tenderness for a week. During the physical examination, the patient’s breasts were found to be bilaterally symmetrical, without any skin retraction. The two nipples were on the same horizontal line without discharge nor retraction. A mass was palpable in the upper-outer quadrant about 5 cm away from the right nipple, with an approximate size of 1.5 cm × 1 cm. The mass was tough in texture, irregular in shape, unclear in boundary and slightly adhesive to the surrounding tissues. There were no positive findings in the left breast. The superficial lymph nodes were not palpable in the bilateral axillary and clavicular fossa. The patient had no history of smoking or alcohol consumption and there was no family history of any types of tumor.Ultrasonography revealed an ill-defined mass in the right breast, and its internal echo was non-homogenous. Mammography revealed a small irregular dense shadow in the upper outer quadrant of the right breast (Figure 1). The boundary of the shadow was unclear and the glands around were gathered without obvious internal calcification. The emission computed tomography (ECT) of whole-body bone imaging and the CT of brain, liver and lung imaging showed no signs of tumor metastasis. Evaluation of tumor markers showed that serum carbohydrate antigen (CA) CA 15–3 was 37.6 U/ml (the normal range is 0–35 U/ml). Other tumor markers were within normal ranges. A simple lumpectomy was performed in the hospital.Grossly, the well-circumscribed pale-tan nodule measured 1.6cm × 1.2cm × 0.5cm and lacked a distinct capsule. Histologically, the nodule was composed of two obscure lobules without evidence of a capsule on low-power examination (Figure 2). One of the lobules consisted of small tubular ducts formed by two phenotypically distinct cell layers. The inner epithelial cells exhibited eosinophilic cytoplasm and the outer myoepithelial cells were clear. Tubular structures predominated in this lobule and formed an expansile circumscribed, partially-encapsulated mass, although focally infiltrated into adjacent fat tissue (Figure 2). A papillary component was also noted in some areas. In addition, atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (11/10HPF) (Figure 3). These histological features support the diagnosis of malignant adenomyoepithelioma.The other lobule of the mass exhibited typical histological image of adenoid cystic carcinoma which consisted of tubular and cribriform structure and had infiltrative borders. The epithelial and myoepithelial cells, whose difference was not as obvious as in adenomyoepithelioma, were polarized around two types of spaces: true glandular spaces (contain mucoid material) and pseudolumens (contain basement membrane material) (Figure 4). The myoepithelial cells of adenoid cystic carcinoma were smaller, had a more hyperchromatic and basaloid character and had much less cytoplasm than those of adenomyoepithelioma, and the invagination of stroma was frequently present. Transition from adenomyoepithelioma to adenoid cystic carcinoma was gradual. And we can also see adenomyoepithelioma and adenoid cystic carcinoma combined in some areas.Immunohistochemistry can distinguish the two different cell types. In both lesions, the myoepithelial cells were highlighted by CK5/6, smooth muscle actin (SMA) and p63, while the epithelial cells were positive for CK18 (Figure 5). Immunostains for estrogen (ER) and progesterone (PR) were negative in AME and ACC but positive in surrounding normal ductal epithelial, and human epidermal growth factor receptor 2 (Her2) were also negative in this case. Nevertheless, CD117 expression was found in adenoid cystic carcinoma in contrast to adenomyoepithelioma, where CD117 was not expressed (Figure 5).Figure 1


Malignant adenomyoepithelioma combined with adenoid cystic carcinoma of the breast: a case report and literature review.

Yang Y, Wang Y, He J, Pan G, Tuo X, Jiang A, Bian L - Diagn Pathol (2014)

High-power examination to the malignant adenomyoepithelioma. Atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (arrow). HE, ×200.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4222729&req=5

Fig3: High-power examination to the malignant adenomyoepithelioma. Atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (arrow). HE, ×200.
Mentions: A 61-year-old female was admitted to The First Affiliated Hospital of Kunming Medical University, Yunnan, China, complaining of a lump in the right breast which had appeared 2 days earlier and had been increasing in tenderness for a week. During the physical examination, the patient’s breasts were found to be bilaterally symmetrical, without any skin retraction. The two nipples were on the same horizontal line without discharge nor retraction. A mass was palpable in the upper-outer quadrant about 5 cm away from the right nipple, with an approximate size of 1.5 cm × 1 cm. The mass was tough in texture, irregular in shape, unclear in boundary and slightly adhesive to the surrounding tissues. There were no positive findings in the left breast. The superficial lymph nodes were not palpable in the bilateral axillary and clavicular fossa. The patient had no history of smoking or alcohol consumption and there was no family history of any types of tumor.Ultrasonography revealed an ill-defined mass in the right breast, and its internal echo was non-homogenous. Mammography revealed a small irregular dense shadow in the upper outer quadrant of the right breast (Figure 1). The boundary of the shadow was unclear and the glands around were gathered without obvious internal calcification. The emission computed tomography (ECT) of whole-body bone imaging and the CT of brain, liver and lung imaging showed no signs of tumor metastasis. Evaluation of tumor markers showed that serum carbohydrate antigen (CA) CA 15–3 was 37.6 U/ml (the normal range is 0–35 U/ml). Other tumor markers were within normal ranges. A simple lumpectomy was performed in the hospital.Grossly, the well-circumscribed pale-tan nodule measured 1.6cm × 1.2cm × 0.5cm and lacked a distinct capsule. Histologically, the nodule was composed of two obscure lobules without evidence of a capsule on low-power examination (Figure 2). One of the lobules consisted of small tubular ducts formed by two phenotypically distinct cell layers. The inner epithelial cells exhibited eosinophilic cytoplasm and the outer myoepithelial cells were clear. Tubular structures predominated in this lobule and formed an expansile circumscribed, partially-encapsulated mass, although focally infiltrated into adjacent fat tissue (Figure 2). A papillary component was also noted in some areas. In addition, atypia was obvious in both epithelial and myoepithelial cells with moderate degree of nuclear pleomorphism, prominent nucleoli, high nuclear cytoplasmic ratio and increased mitotic figures (11/10HPF) (Figure 3). These histological features support the diagnosis of malignant adenomyoepithelioma.The other lobule of the mass exhibited typical histological image of adenoid cystic carcinoma which consisted of tubular and cribriform structure and had infiltrative borders. The epithelial and myoepithelial cells, whose difference was not as obvious as in adenomyoepithelioma, were polarized around two types of spaces: true glandular spaces (contain mucoid material) and pseudolumens (contain basement membrane material) (Figure 4). The myoepithelial cells of adenoid cystic carcinoma were smaller, had a more hyperchromatic and basaloid character and had much less cytoplasm than those of adenomyoepithelioma, and the invagination of stroma was frequently present. Transition from adenomyoepithelioma to adenoid cystic carcinoma was gradual. And we can also see adenomyoepithelioma and adenoid cystic carcinoma combined in some areas.Immunohistochemistry can distinguish the two different cell types. In both lesions, the myoepithelial cells were highlighted by CK5/6, smooth muscle actin (SMA) and p63, while the epithelial cells were positive for CK18 (Figure 5). Immunostains for estrogen (ER) and progesterone (PR) were negative in AME and ACC but positive in surrounding normal ductal epithelial, and human epidermal growth factor receptor 2 (Her2) were also negative in this case. Nevertheless, CD117 expression was found in adenoid cystic carcinoma in contrast to adenomyoepithelioma, where CD117 was not expressed (Figure 5).Figure 1

Bottom Line: Histologically, the nodule was composed of two obscure lobules.Immunohistochemically, CK18 and P63 highlighted the epithelial and myoepithelial cells respectively in both lesions.The histogenesis of these tumors is also discussed.

View Article: PubMed Central - PubMed

Affiliation: Department of Pathology, The First Affiliated Hospital of Kunming Medical University, 295, Xichang road, Kunming, Yunnan, China. bianli1976@sina.com.

ABSTRACT

Background: 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.

Case presentation: A 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.

Conclusion: This 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 slides: The 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.

Show MeSH
Related in: MedlinePlus