Background: Myoclonic jerks are due to sudden, brief, involuntary muscle contractions, positive myoclonus, or brief cessation of ongoing muscular activity, negative myoclonus, and may be difficult to recognize. Case Report: We describe an immunocompetent, adult, male patient with sleep-related, multifocal, myoclonic jerks and neurotoxoplasmosis with abnormal cerebrospinal fluid but normal brain imaging. There was complete resolution of the myoclonus with antitoxoplasmosis therapy after 1 week, and no relapse after 1 year. Discussion: Neurotoxoplasmosis may be subtle in presentation, difficult to diagnose, and more common than realized, and it is being increasingly implicated in epileptogenesis in humans.