Tolosa-Hunt syndrome(THS) is a painful ophthalmoplegia caused by an idiopathic granulomatous in- flammation of the superior orbital fissure or cavernous sinus of unknown etiology. A 72-year-old woman developed right-sided ophthalmoplegia with the pupillary dilatation and ptosis, and hypesthesia in the territory of the ophthalmic division of the right trigerminal nerve in May 2003. The MRI showed enlar- gement of right cavernous sinus by an enhancing soft tissue mass extending to the sellar region. Prior to this presentation, in August 2002, MRI done at another institusion already revealed the same soft tissue mass in the right cavernous sinus. The patient underwent a transsphenoidal biopsy which revealed chronic granulomatous inflammation with necrosis. The patient was treated with steroids, followed by the resolution of the neurologic symptoms. We report this case of biopsy-proven THS in which radiologic abnormality preceded clinical presentation by at least 10 months. Korean Journal of Headache 5(2):153-157, 2004