To describe a case of sympathetic ophthalmia due to corneal perforation caused by exposure keratitis in a patient with recurrent sphenoid wing meningioma.

A 34-year-old female patient presented with proptosis in her left eye caused by left sphenoid greater wing meningioma despite tumor debulking surgery and radiation treatment. The cornea was perforated with prolapsed iris due to exposure keratitis, thus enucleation of the left eye was performed. After 2 weeks, an inflammatory reaction occurred in both eyes, keratic precipitates on corneal endothelium, exudative retinal detachment, and multiple granulomatous nodules on the right eye retina. The patient was diagnosed with sympathetic ophthalmia, thus enucleation of the left eye and debulking of the tumor were performed followed by a high-dose intravenous steroid therapy. At 5 months postoperatively, slit lamp biomicroscope showed no chamber reaction; improved disc swelling and exudative retinal detachment in the right eye were observed.

Despite conservative treatment for exposure keratitis due to proptosis caused by malignant sphenoid meningioma, corneal perforation can develop. Because sympathetic ophthalmia can occur, the other eye should be monitored.