Ocular neuromyotonia caused by a recurrent sphenoidal ridge meningioma

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.

The Most rapid recurrent meningioma: Case report and literature review

“…No recurrence in 5 years. Read and Williams, 2017 [15] Cervicomedullary junction Neck pain and rapid onset of arm and leg weakness 5 years 1,4 cm enhancing mass at cervicomedullary junction WHO grade I Systemic doxorubicin n/a Sadiya et al , 2019 [16] Left frontal Slurred speech 1 year 2,9 × 2,5 × 2,4 cm solid cyctic tumor of left frontal dural base with invasion of brain parenchyma WHO grade II-III Subtotal resection and radiotherapy n/a Shingai et al , 2021 [17] Sphenoid Intermittent diplopia 7 years Tumor mass in the posterior part of sphenoid with right oculomotor invasion WHO grade II Resection and gamma knife radiotherapy n/a Thomas et al , 2019 [18] Right middle fossa Generalized tonic-clonic seizures 7 years, 2 years, 5 years, and 2 years 2,4 × 2,9 × 1,5 cm right middle fossa with foramen ovale invasion WHO grade II Surgical resection, bevacizumab and everolimus n/a Wang et al , 2015 [19] Right orbitofrontal Serious Headache, Gradually impaired vision of the right eye, and vomiting 3 years, 2 years, and 5 months 1. Small local reccurence tumor 2.…”

Ocular neuromyotonia: a review of diagnosis and treatment

“…Other causes of ONM as reported in the literature include hypovitaminosis D [3], congenital [22], alcohol ingestion [23], Paget disease of bone [24], myelogram with thorium toxicity [25], cavernous sinus thrombosis secondary to mucormycosis [26], intramedullary stroke [27], chronic inflammatory demyelinating disease [28], botulinum toxin injection [29], brainstem demyelination [30], Tolosa-Hunt syndrome [31], idiopathic [32], intracranial tumor [33], and as a presenting sign of meningioma [34].…”

“…In cases of ONM due to nerve compression, neurosurgical microvascular decompression can achieve resolution of symptoms. In a review of eight ONM cases with a clear relationship to nerve compression, Shingai et al found that placing a sponge between the nerve and the vessel resulted in improvement for all three of its cases while carbamazepine only resulted in improvement for two of its five cases [33].…”