The reversible potassium channel blocker 4-aminopyridine is effective in the treatment of numerous cerebellar dysfunctions, such as episodic ataxia type 2 and downbeat nystagmus syndrome. In 2011, its sustained release form, dalfampridine, was admitted in Europe for the treatment of walking difficulties in patients with multiple sclerosis (MS). Here we report the case of a 44-year old patient with a progressive MS whose upper limb tremor was markedly reduced under treatment with 4-aminopyridine, as documented in a Tremor Activities of Daily Living questionnaire and in the 9-Hole Peg test. Hand accelerations decreased in the left hand from 10.9 m/sec(2) to 2.2 m/sec(2) and in the right hand from 4.2 m/sec(2) to 0.9 m/sec(2). This case report indicates for the first time that 4-aminopyridine might be effective in the symptomatic treatment of tremor entities in patients with MS. The finding calls for further prospective studies to determine the usefulness of 4-aminopyridine or its sustained-release form dalfampridine in treating patients with tremor and MS.