PURPOSE: To report a case of ocular neuromyotonia occurring after cavernous sinus thrombosis secondary to mucormycosis. METHODS: Case report. We performed serial comprehensive neuro-ophthalmologic examinations. RESULTS: Fifteen months after initial total ophthalmoplegia of the right eye and complete right upper eyelid ptosis, isolated ocular neuromyotonia, characterized by episodic upward jerking movements of the right upper eyelid, was noted. CONCLUSION: Ocular neuromyotonia, which usually manifests in patients with a history of intracranial tumors and cranial radiation, may also be secondary to infectious cavernous sinus thrombosis.