We report on a 35-year old patient with multiple sclerosis (MS) with clinically and electrophysiologically typical facial myokymia, appearing during an acute bout of the disease. Magnetic resonance imaging (MRI) disclosed two new pontine lesions. During a follow-up period of eight months facial myokymia subsided, but five months later central fever with good therapeutic response to carbamazepine developed. In this report we discuss the potential mechanisms of myokymia and central temperature dysregulation in MS.