A rare case of oncocytic Schneiderian papilloma originating from the sphenoid sinus characterised, for 3 years, by non-specific symptoms of severe headache, a block of nasal breathing, and deprecating sense of smell was presented by an elderly female patient. Sphenoid sinus functional endoscopic sinus surgery (FESS), with a one-block tumour excision, through an endonasal approach, with a histological study of removed tumour masses, were performed on the patient. Long observation in the post-operative period was necessary, considering the risk of recurrence and malignancy of oncocytic Schneiderian papilloma (OSP). Although the oncocytic papilloma of the sphenoid sinus is rare, non-specific symptoms make this pathology easily misdiagnosed. Thus, any isolated unilateral process in the paranasal sinuses with long-existing symptoms must be given careful attention due to the chance of this process being an inverted papilloma with malignization. CT scan indicating a unilateral opacification of paranasal sinuses with local calcifications is a typical manifestation, and endoscopic sphenoidotomy can be recommended as a treatment of choice.
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