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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

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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.

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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

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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.

Funding

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

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Authors and Affiliations

  1. Pathology Division, Shizuoka Cancer Center, 1007 Shimonagakubo, Nagaizumi-cho, Sunto-gun, Shizuoka, 411-8777, Japan

    Kimihide Kusafuka, Takuya Kawasaki, Takashi Nakajima & Takashi Sugino

  2. Division of Head and Neck Surgery, Shizuoka Cancer Center, Shizuoka, Japan

    Tetsuro Onitsuka, Noriko Hamaguchi, Kotaro Morita, Takashi Mukaigawa, Yukio Nishiya, Tomoyuki Kamijo & Yoshiyuki Iida

Authors
  1. Kimihide Kusafuka
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  2. Takuya Kawasaki
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  3. Tetsuro Onitsuka
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  4. Noriko Hamaguchi
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  5. Kotaro Morita
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  6. Takashi Mukaigawa
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  8. Tomoyuki Kamijo
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  9. Yoshiyuki Iida
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  10. Takashi Nakajima
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  11. Takashi Sugino
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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.

Corresponding author

Correspondence to Kimihide Kusafuka.

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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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Kusafuka, K., Kawasaki, T., Onitsuka, T. et al. 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. Head and Neck Pathol 14, 230–238 (2020). https://doi.org/10.1007/s12105-018-0987-2

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  • DOI: https://doi.org/10.1007/s12105-018-0987-2

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