Successful Intrathecal Baclofen Therapy for Intractable Paroxysmal Sympathetic Hyperactivity in Patient with Pontine Hemorrhage: A case report

Abstract

Paroxysmal sympathetic hyperactivity (PSH) is characterized by increased activity of the sympathetic nervous system. Although several treatments have been proposed for PSH, their efficacies are uncertain. We report a case of a patient with intractable PSH after pontine hemorrhage who was treated with intrathecal baclofen (ITB). A 61-year-old man was diagnosed with pontine hemorrhage expanding to the bilateral midbrain. Beginning 2 weeks after onset, he developed paroxysmal hyperthermia, tachycardia, tachypnea, hypertension, and generalized dystonic movements. Several medications were administered to the patient, including fentanyl patch, dantrolene, gabapentin, and β blockers. Nevertheless, PSH episodes continued to occur more than twice per day. Seven months after onset, we performed a trial of ITB injection, which led to reduced severity and frequency of PSH. Therefore, the ITB therapy was implanted. Continuous ITB was initiated at a rate of 100 μg/d, which was gradually increased up to 200 μg/d. Oral medications were tapered off. No PSH episode occurred for 4 weeks. Our findings suggest that ITB therapy may be used to manage intractable PSH. Intrathecal baclofen may inhibit postsynaptic activity and suppress sympathetic activity via the stimulation of γ-aminobutyric acid B receptors the brain.