Different forms of symptomatic dystonia have been reported in patients with multiple sclerosis, but the appearance of spasmodic torticollis is extremely rare. We present the case of a 24-year-old man with postural and intentional left hand tremor and leftward spasmodic torticollis. The duration of the disease is 6 years, without remissions. The cerebrospinal fluid examination revealed oligoclonal (two bands) hypergammaglobulinemia. Auditory and visual evoked potentials showed altered sensory afferentation. On magnetic resonance imaging brain scan, multiple T2 high signal lesions involving the white matter of both cerebral hemispheres, mainly the periventricular regions, were found. There were also multiple lesions in the brain stem and some in the cerebellar hemispheres. Thus the diagnosis of multiple sclerosis could be considered as definite. In conclusion, extremely rarely, multiple sclerosis may appear with progressive spasmodic torticollis and rubral tremor.