Abstract Background Isolated sphenoid sinus disease (ISSD) is a rare, often misdiagnosed condition of the paranasal sinus. If left untreated, it can lead to complications involving pituitary gland, cavernous sinus, neurologic, and vascular structures nearby. Case report We present case 60-year-old female with history severe left-sided headache, facial pain, diplopia, lateral rectus palsy. She w...