Carcinoma ex pleomorphic adenoma (CXPA) arising from primary or recurrent pleomorphic adenoma is characterized by the presence of longstanding palpable mass with growth spurt mainly in the parotid region. Overall, patients with CXPA have a poor prognosis. Pleural effusion involved by CXPA is precedingly rare, compared to that common dissemination from pulmonary or mammary origin. Here we reported a metastatic CXPA in pleural effusion from an 82-year-old male. The clinical presentation, cytological morphology, and immunohistochemistry confirmed the diagnosis. When dealing patients with prolonged history of pleomorphic adenoma, underwent recent rapid growth of malignant transformation and malignant cells presented in the pleural effusion, metastatic CXPA should be seriously considered.
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