Pediatric Rehabilitation Fellow Physician Children's Hospital of Philadelphia Philadelphia, Pennsylvania, United States
Case Diagnosis: Paroxysmal Sympathetic Hyperactivity
Case Description: Two adolescent males who presented with headaches that progressed to altered mental status were found to have intraventricular hemorrhage (IVH) in the setting of ruptured arteriovenous malformation (AVM) with increased intracranial pressure.
One patient underwent craniotomy for AVM resection. On presentation, he demonstrated hyperthermia, tachycardia, and tachypnea concerning for paroxysmal sympathetic hyperactivity (PSH). He was started on bromocriptine 1.25 mg, later increased to 2.5 mg daily, and finally twice daily with the goal of addressing hyperthermia. He also started propranolol for PSH treatment. Fevers resolved after 8 days.
Another patient underwent angiography and embolization of AVM as well as therapeutic hypothermia. When this was discontinued, he developed hyperthermia as well as hypertension and tachycardia concerning for PSH. He started bromocriptine 2.5 daily in addition to multiple agents for sedation and hypertension management. Fevers resolved 2 days after starting bromocriptine.
Discussions: PSH can be characterized by autonomic dysregulation and motor symptoms after acute brain injury, including non-traumatic etiology. Limited evidence exists regarding abortive and prophylactic management for pediatric patients. Pharmacologic management can include bromocriptine, propranolol, clonidine or dexmedetomidine, benzodiazepines, opioids, and gabapentin. Bromocriptine is a synthetic dopamine agonist with an unclear mechanism for the treatment of autonomic dysfunction, but may act on the hypothalamus and corpus striatum and may be used to target hyperthermia. The presented cases demonstrate PSH including hyperthermia, hypertension, and tachycardia can be present in the setting of non-traumatic brain injury in adolescent males. They also demonstrate use of bromocriptine in conjunction with other pharmacologic interventions to manage PSH including hyperthermia in adolescent males with non-traumatic brain injury.
Conclusions: Bromocriptine can be utilized in conjunction with other pharmacological therapies for management of PSH including hyperthermia in adolescent patients with AVM rupture and IVH.