Cystadenocarcinoma (papillary cystadenocarcinoma) of the salivary gland is a rare malignant neoplasm. Major locations of this neoplasm are the parotid gland, the sublingual gland, and minor salivary glands, while occurrence in the submandibular gland is extremely rare. As far as we know, only one report, written in the French language, has been published concerning cystadenocarcinoma of the submandibular gland, but no report is available in the English language. In this report, we describe a case of cystadenocarcinoma arising from the submandibular gland of a 54-year-old male patient. The neoplasm is a low-grade carcinoma, and pre-operative examination may not show typical characteristics of malignant neoplasms. Therefore, as was true in this case, the differential diagnosis from benign lesions is sometimes difficult. This is the first report on cystadenocarcinoma of the submandibular gland in the English language and the first to show a computed tomography (CT) scan and magnetic resonance imaging (MRI) of this neoplasm in the submandibular gland.
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