We report axial myoclonic jerks causing flexion of the trunk, neck, left shoulder, hips and knees in a 28-years-old HIV positive patient. The clinical and electromyographic features of the jerks were consistent with a spinal origin and corresponded to the new concept of propriospinal myoclonus. No structural lesion was identified in this patient. Neurological examination was otherwise normal. HIV specific antibodies were detected in CSF, suggesting central nervous system infection. Spinal myoclonus should be considered an unusual and early manifestation of central nervous system HIV infection.