Abstract
BACKGROUND: Ocular neuromyotonia (ONM) is a rare ocular motility disorder characterized by involuntary paroxysmal extraocular muscle contraction and is caused by radiation therapy, vascular compression, and inflammatory disease. This study includes a rare case of ONM caused by a recurrent meningioma. CASE DESCRIPTION: A 56-year-old man presented with diplopia due to the right oculomotor nerve palsy caused by a sphenoidal atypical meningioma, with improved symptoms after initial surgery. During the next 7 years, he underwent local radiation therapy, second surgery, and Gamma Knife radiosurgery to control the tumor's repetitive recurrence around the right anterior clinoid process. After these treatments, residual tumor was controlled for the next 3 years. However, 3 months after his last visit, he started to suffer from the right ONM and visual disturbance. The magnetic resonance imaging results revealed a rapid growth of the posterior part of the residual tumor, involving the right oculomotor nerve. The third tumor resection was performed to prevent further aggravation of the symptoms. Decompression of the right oculomotor nerve was achieved, and ONM disappeared immediately after surgery. CONCLUSION: If nerve compression by the tumor is clearly indicated with the neuroradiological assessment, surgical intervention is the treatment of choice to improve ONM.