OBJECTIVE: In this study we examine the temporal connection between periodic leg movements (PLMs) and cortical arousals, as well as the treatment effect of pramipexole, in a clinical case with spinal cord lesion. METHODS: A patient with complete cervical spinal cord injury and PLMs during sleep underwent two baseline sleep recordings, one recording with dopaminergic treatment, and one recording with adaptive servoventilation. RESULTS: The PLMs were temporally dissociated from cortical arousals as well as from respiratory or heart rate events. PLMs were suppressed by pramipexole and persisted after treatment of apnea. CONCLUSION: The disconnection of PLMs from arousals supports a spinal generator or peripheral trigger mechanism for PLMs. The suppression of movements by a dopamine agonist suggests that its site of action is caudal to the cervical lesion and outside of the brain. Our observation provides significant new knowledge about the pathogenesis of PLMs and warrants studies in larger populations. ; Aaro V. Salminen, Mauro Manconi, Ville Rimpilä, Teemu M. Luoto, Eerika Koskinen, Raffaele Ferri, Juha Öhman, Olli Polo