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Carcinoma ex pleomorphic adenoma is a rare neoplasm of the salivary gland. This lesion occurs when a malignant tumor arises in the epithelial component of a pleomorphic adenoma. The patient was a 35-year-old male with ovoid shaped and protruding cytologic finding by fine needle aspiration biopsy showed highly malignant cells. After submandibular gland extirpation and supraomohyoid neck disection, the histologic examination revealed that tumor was composed partly of a benign pleomorphic adenoma and partly of an adenoid cystic carcinoma component with areas of calcification, capsular invasion, and perineural invasion. The adenoid cystic carcinoma component showed a cribriform pattern, also known as Swiss cheese appearance. We adenoid cystic carcinoma in the submandibular gland.