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Primary Cutaneous Adenomyoepithelioma Ex Spiradenoma With Malignant Histologic Features, Epithelial-Myoepithelial Carcinoma Type: A First Case Report With Molecular Studies.

Authors
  • Tran, Tien Anh N1
  • Bridge, Julia A2
  • Deharvengt, Sophie J3
  • Green, Donald C3
  • Linos, Konstantinos3
  • 1 Advent Health Orlando, Orlando, FL, USA.
  • 2 Nebraska Medical Center, Omaha, NE, USA.
  • 3 Dartmouth-Hitchcock Medical Center, Lebanon, NH, USA. , (Lebanon)
Type
Published Article
Journal
International journal of surgical pathology
Publication Date
Dec 09, 2019
Identifiers
DOI: 10.1177/1066896919888579
PMID: 31813296
Source
Medline
Keywords
Language
English
License
Unknown

Abstract

Adenomyoepithelioma is an extremely rare primary cutaneous neoplasm. Although there is ample evidence on the existence of malignant adenomyoepithelioma in the breast, a malignant counterpart in the skin has not been documented. We report a primary cutaneous adenomyoepithelioma (pcAME) with malignant features arising from a spiradenoma in a 39-year-old female patient. The tumor was solid-cystic in appearance and entirely located in the subcutaneous tissue. Histologically, the tumor displayed foci of adenomatous changes and adenomyoepitheliomatous hyperplasia adjacent to a minute spiradenoma. Gradual increase of architectural complexity, cytologic atypia, mitotic activity, and infiltrative growth were observed in a significant portion of the neoplasm, indicative of transformation to adenomyoepithelioma and subsequently low- to high-grade salivary-type epithelial-myoepithelial carcinoma (EMCA). The intimate dual populations of ductal and myoepithelial cells were highlighted by a panel of immunohistochemical stains in all different components of the tumor. Molecular studies revealed a PIKCA3 mutation, a genetic aberration that has been documented in EMCA, particularly of breast origin. The current case documents for the first time a pcAME with malignant features arising from a spiradenoma and suggests adenomyoepithelioma ex spiradenoma as a possible tumorigenesis pathway of this rare cutaneous tumor.

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