Abstract Introduction A 38-year-old man with severe head trauma complicated by paroxysmal severe intracranial pressure elevation associated with tachypnea, tachycardia, diaphoresis, and extensor posturing was diagnosed as suffering from paroxysmal autonomic instability with dystonia (PAID). These events were unresponsive to standard medical therapy, which included morphine, fentanyl,labetalol, lorazepam, metoprolol, and clonidine.Methods A trial treatment with dexmedetomidine, a central acting alpha2-agonist, to control symptoms of PAID was initiated 12 days after injury. PAID-related events subsided during the 72-h infusion protocol of 0.2–0.7 mcg/kg/h. No further events were noted after termination of the 72-h infusion.Conclusions Dexmedetomidine may be a novel pharmacologic agent to aid in abrogating PAID. Keywords Dexmedetomidine Sympathetic storming Paroxysmal autonomic instability with dystonia Diencephalic seizure Dysautonomia Traumatic brain injury |