Sphenoid sinus mucocele is an infrequent but well-recognized entity in sinus pathology. The symptoms produced by it are related to the anatomical surroundings of the sphenoid sinus. We describe a case of sphenoid sinus mucocele presenting with ipsilateral oculomotor nerve palsy 10 years after endoscopic sphenoidotomy. The patient underwent emergency endoscopic decompression of the sphenoid sinus with marsupialization of the mucocele, resulting in immediate remission of his symptoms. We conclude that the otorhinolaryngologist should be aware of such a potential complication when counselling the patient prior to endoscopic sinus surgery.