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Ganglioneuromas may occur in the small and large intestine as either solitary lesions or, more commonly, as multiple lesions (ganglioneuromatosis). The former are very rare, whereas ganglioneuromatosis may be associated with von Recklinghausen's disease and multiple endocrine neoplasia (MEN) type II B. We described the clinicopathologic features of a case of solitary polypoid ganglioneuroma of the ileocecal valve. The lesion was endoscopically diagnosed in a 27 year old man, with a history of abdominal pain. No association with von Reckling-hausen's disease or MEN was identified. Mutational analysis for RET was negative. Microscopically, the tumor consisted of a proliferation of well differentiated Schwann cells and ganglion cells in the lamina propria. The solitary polipoid ganglioneuroma is invariably benign. It shows no evidence of recurrence after total excision.