Malignant adenomyoepithelioma of the breast

Abstract Background Adenomyoepithelioma (AME) of the breast is a very rare tumor and is generally considered to be benign. However, some show malignant transformation, which results in local recurrences or distant metastases. The morphological features of AME that might predict malignant potential h...

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Main Authors: Kazuki Moro, Eiko Sakata, Asa Nakahara, Hideki Hashidate, Emmanuel Gabriel, Haruhiko Makino
Format: Article
Language:English
Published: Japan Surgical Society 2020-05-01
Series:Surgical Case Reports
Subjects:
Online Access:http://link.springer.com/article/10.1186/s40792-020-00881-2
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author Kazuki Moro
Eiko Sakata
Asa Nakahara
Hideki Hashidate
Emmanuel Gabriel
Haruhiko Makino
author_facet Kazuki Moro
Eiko Sakata
Asa Nakahara
Hideki Hashidate
Emmanuel Gabriel
Haruhiko Makino
author_sort Kazuki Moro
collection DOAJ
description Abstract Background Adenomyoepithelioma (AME) of the breast is a very rare tumor and is generally considered to be benign. However, some show malignant transformation, which results in local recurrences or distant metastases. The morphological features of AME that might predict malignant potential have not been elucidated. Moreover, there is also no established multidisciplinary treatment for malignant AME aside from complete excision at an early stage. Case presentation A 64-year-old female diagnosed with AME of the left breast underwent lumpectomy. The surgical margins were negative. Six months after the operation, however, malignant AME recurred locally in the left breast. MRI showed multiple masses, which invaded the skin. A left mastectomy with axillary lymph node dissection was performed. Additional areas of AME were found in about one third of the entire breast. Eight months after the mastectomy, lung metastases were detected. She underwent chemotherapy with fluorouracil, epirubicin, and cyclophosphamide (FEC) for 9 cycles with little response. Lung metastasectomy was performed. Nine months after lung metastasectomy, the metastases were widespread to the brain, heart, and kidney; she subsequently died 2 months later. Conclusions Malignant AME has various morphological features, and in this report, we characterize new findings from both imaging and pathology/autopsy. Malignant potency is related to the tumor size, tumor appearance, and mitoses, even if only a few. Given that ductal spread is one of the morphological features of malignant AME, it is of paramount importance to assess the surgical margins.
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spelling doaj-art-7b859c45cfd74c30afbf928e0bee27e62025-08-20T03:33:58ZengJapan Surgical SocietySurgical Case Reports2198-77932020-05-01611610.1186/s40792-020-00881-2Malignant adenomyoepithelioma of the breastKazuki Moro0Eiko Sakata1Asa Nakahara2Hideki Hashidate3Emmanuel Gabriel4Haruhiko Makino5Department of Breast Surgery, Niigata City General HospitalDepartment of Breast Surgery, Niigata City General HospitalDepartment of Pathology, Niigata City General HospitalDepartment of Pathology, Niigata City General HospitalDepartment of Surgery, Mayo ClinicDepartment of Breast Surgery, Niigata City General HospitalAbstract Background Adenomyoepithelioma (AME) of the breast is a very rare tumor and is generally considered to be benign. However, some show malignant transformation, which results in local recurrences or distant metastases. The morphological features of AME that might predict malignant potential have not been elucidated. Moreover, there is also no established multidisciplinary treatment for malignant AME aside from complete excision at an early stage. Case presentation A 64-year-old female diagnosed with AME of the left breast underwent lumpectomy. The surgical margins were negative. Six months after the operation, however, malignant AME recurred locally in the left breast. MRI showed multiple masses, which invaded the skin. A left mastectomy with axillary lymph node dissection was performed. Additional areas of AME were found in about one third of the entire breast. Eight months after the mastectomy, lung metastases were detected. She underwent chemotherapy with fluorouracil, epirubicin, and cyclophosphamide (FEC) for 9 cycles with little response. Lung metastasectomy was performed. Nine months after lung metastasectomy, the metastases were widespread to the brain, heart, and kidney; she subsequently died 2 months later. Conclusions Malignant AME has various morphological features, and in this report, we characterize new findings from both imaging and pathology/autopsy. Malignant potency is related to the tumor size, tumor appearance, and mitoses, even if only a few. Given that ductal spread is one of the morphological features of malignant AME, it is of paramount importance to assess the surgical margins.http://link.springer.com/article/10.1186/s40792-020-00881-2DifferentiationDuctal spreadHematogenous spreadMalignant adenomyoepitheliomaSurgical margin
spellingShingle Kazuki Moro
Eiko Sakata
Asa Nakahara
Hideki Hashidate
Emmanuel Gabriel
Haruhiko Makino
Malignant adenomyoepithelioma of the breast
Surgical Case Reports
Differentiation
Ductal spread
Hematogenous spread
Malignant adenomyoepithelioma
Surgical margin
title Malignant adenomyoepithelioma of the breast
title_full Malignant adenomyoepithelioma of the breast
title_fullStr Malignant adenomyoepithelioma of the breast
title_full_unstemmed Malignant adenomyoepithelioma of the breast
title_short Malignant adenomyoepithelioma of the breast
title_sort malignant adenomyoepithelioma of the breast
topic Differentiation
Ductal spread
Hematogenous spread
Malignant adenomyoepithelioma
Surgical margin
url http://link.springer.com/article/10.1186/s40792-020-00881-2
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AT asanakahara malignantadenomyoepitheliomaofthebreast
AT hidekihashidate malignantadenomyoepitheliomaofthebreast
AT emmanuelgabriel malignantadenomyoepitheliomaofthebreast
AT haruhikomakino malignantadenomyoepitheliomaofthebreast