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Small cell carcinoma (SmCC) is a type of neuroendocrine tumor commonly originating in the lung, with only about 2-4% of cases arising at extrapulmonary sites. Extrapulmonary SmCC of the head and neck has a poor prognosis and a high rate of distant metastasis. The paranasal sinus is a rare location for extrapulmonary SmCC and only a few related papers have been published to date. We report a rare case of SmCC originating from the sphenoid sinus in a patient with a recurrent pituitary tumor with a literature review.