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Abstract Carcinoma ex pleomorphic adenoma (CXPA) is a rare epithelial malignancy arising from benign pleomorphic adenoma. Cases with two distinct malignant components are exceedingly rare. Nonspecific clinical and imaging features and heterogeneous histology hinder differentiation from other salivary gland tumors, risking misdiagnosis and suboptimal therapy. We report two CXPA cases with dual malignant components and review the literature.
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