Background: Mucoceles occur most commonly in frontal sinus and rarely in sphenoid sinus. The common presentations in sphenoid sinus mucoceles are deep-seated headache and visual disturbance. Case description: We present the case of a 58-year-old lady who came to us with headache, nose block, and reduced vision in the left eye since 2 years. She was diagnosed with an expansile lesion compressing the optic nerve. She underwent endoscopic drainage multiple times. The histopathological examination (HPE) was suggestive of aspergillus and she was treated with voriconazole. In spite of medical and surgical treatment, her symptoms were persistent. Eventually, she presented to our hospital with worsening of symptoms and deterioration of vision. Following MRI, we did drainage of mucocele with marsupialization. The clival bone was drilled out widening the entire face of sphenoid sinus. Fungal culture was suggestive of aspergillus and the patient was given voriconazole for 3 months. She has been on regular follow-up since then. Conclusion: In cases of recurrent sphenoid sinus mucoceles, complete marsupialization of the mucocele, drilling of bony barriers, adequate medical management, and diligent follow-up are necessary for the successful outcome. Clinical significance: Recurrent mucoceles of the sphenoid sinus represent a difficult entity to treat and are ridden with complications. Not only do they require aggressive surgical management but also meticulous postoperative medical management and repeated endoscopic follow-ups.