We report the clinical and pathologic features of a small cell neuroendocrine carcinoma arising in the oral cavity of a 63-year-old man. The tumor stained positively with neuron-specific enolase and had dense core granules ultrastructurally. Chemotherapy ordinarily given for pulmonary small cell carcinoma induced a clinical remission. When the tumor recurred 8 mo later, focal squamous differentiation was present within the neuroendocrine carcinoma. The patient died 2.5 yr after presentation.
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