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Acantholytic Squamous Cell Carcinoma and Salivary Duct Carcinoma Ex-pleomorphic Adenoma of the Submandibular Gland: A Report of Two Extremely Rare Cases with an Immunohistochemical Analysis

  • Kimihide KusafukaEmail author
  • Takuya Kawasaki
  • Tetsuro Onitsuka
  • Noriko Hamaguchi
  • Kotaro Morita
  • Takashi Mukaigawa
  • Yukio Nishiya
  • Tomoyuki Kamijo
  • Yoshiyuki Iida
  • Takashi Nakajima
  • Takashi Sugino
Case Report
  • 44 Downloads

Abstract

Carcinoma ex pleomorphic adenoma (CXPA) is a malignant tumor of the salivary gland that arises from pleomorphic adenoma (PA). Squamous cell carcinoma (SCC) is extremely rare in the salivary glands. We report two cases of acantholytic SCC (ASCC) ex PA. Case 1 involved a 72-year-old female, and case 2 involved a 67-year-old male. Histologically, both cases involved PA, and salivary duct carcinoma (SDC) components, which were positive for androgen receptor (AR) and gross cystic disease fluid protein (GCDFP)-15 but negative for HER2, were seen in the intracapsular regions. The invasive components consisted of ASCC, which were positive for cytokeratin 5/6 and p63 but negative for AR and GCDFP-15. The SDC and ASCC components were positive for the epidermal growth factor receptor. In both cases, the cytoplasmic localization or decreased expression of E-cadherin was observed in the ASCC. In the early phase, CXPA might emerge as SDC, and it might change into SCC as it invades beyond the capsule due to changes in microenvironment. Also, the aberrant expression of E-cadherin is related to acantholysis in SCC.

Keywords

Acantholysis Squamous cell carcinoma Salivary duct carcinoma Carcinoma ex pleomorphic adenoma E-cadherin 

Abbreviations

CXPA

Carcinoma ex pleomorphic adenoma

PA

Pleomorphic adenoma

SDC

Salivary duct carcinoma

AR

Androgen receptor

GCDFP-15

Gross cystic disease fluid protein-15

HER2

Human epidermal growth factor receptor 2

SCC

Squamous cell carcinoma

ASCC

Acantholytic squamous cell carcinoma

EvG

Elastica van Gieson

CK

Cytokeratin

EGFR

Epidermal growth factor receptor

MMG

Mammaglobin

PSA

Prostate specific antigen

ASMA

α-smooth muscle actin

EMT

Epithelial–mesenchymal transition

P/M

Polyclonal/monoclonal

AMACR

Alpha-methylacyl-CoA racemase

Cy

Cytoplasmic positivity

Myo

Myoepithelial cells

Duct

Ductal cells

Notes

Acknowledgements

The authors thank Mr. Isamu Hayashi, Mr. Kiyoshi Tone, Ms. Sachiyo Ono, Mr. Koji Muramatsu, Mr. Masato Abe, Mr. Hiroshi Tashiro, Mr. Masatake Honda, Ms. Chiho Tashiro, Ms. Shiori Masujima, Mr. Hiroyuki Shiiya, Mr. Shogo Fujii, Ms. Yoko Kosaka, and Ms. Miho Naka (the staff at the Pathology Division, Shizuoka Cancer Center, Shizuoka, Japan) for their excellent technical assistance. We are also grateful to Ms. Minako Ishii, (the secretary of the Pathology Division, Shizuoka Cancer Center, Shizuoka, Japan) for her valuable help during the preparation of this manuscript.

Author Contributions

KK designed and drafted the manuscript, and KK, TNa and TS made the histopathological diagnosis. TO, NH, KM, TM, TNi, TK, and YI belong to Division of Head and Neck Surgery, Shizuoka Cancer Center, Shizuoka, Japan.

Funding

The authors declare that they received no funding support for this study.

Compliance with Ethical Standards

Conflict of interest

The authors declare no conflict of interest in association with this study.

Ethics Approval and Consent to Participate

This study was approved by the institutional review board of Shizuoka Cancer Center (No. 19–49). All subjects signed informed consent forms to participate.

Consent for Publication

Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. A copy of the consent form is available for review by the Editor of this journal.

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Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  • Kimihide Kusafuka
    • 1
    Email author
  • Takuya Kawasaki
    • 1
  • Tetsuro Onitsuka
    • 2
  • Noriko Hamaguchi
    • 2
  • Kotaro Morita
    • 2
  • Takashi Mukaigawa
    • 2
  • Yukio Nishiya
    • 2
  • Tomoyuki Kamijo
    • 2
  • Yoshiyuki Iida
    • 2
  • Takashi Nakajima
    • 1
  • Takashi Sugino
    • 1
  1. 1.Pathology DivisionShizuoka Cancer CenterShizuokaJapan
  2. 2.Division of Head and Neck SurgeryShizuoka Cancer CenterShizuokaJapan

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