Background: Sarcomatoid salivary duct carcinoma (sarcomatoid SDC) is a rare subtype of SDC. We encountered 1 case of sarcomatoid SDC that developed from a submandibular gland pleomorphic adenoma, and we herein report our findings.

Case: A 42-year-old female had tumentia and pain in the right submental area, and therefore she underwent a close examination, wherein a right submandibular gland neoplasm was detected. Surgery for removing the right submandibular gland was performed in addition to neck dissection. Cytology showed typical findings of conventional SDC. Moreover, isolated and scattered large spindle cells and multinucleated cells were also detected along with atypical epidermal cell clumps showing keratinization. From a histologic perspective, it appeared to be sarcomatoid SDC that developed from a pleomorphic adenoma and also involved a squamous cell carcinoma component.

Conclusion: This is the first report on the cytologic findings ofa case of sarcomatoid SDC with a squamous cell carcinoma component. When sarcomatoid cells appear in the cytology along with findings of typical SDC, sarcomatoid SDC should thus be considered. Furthermore, squamous cell carcinoma component may be involved as in the present case, so it is necessary to be aware of the possibility thereof.

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