Dantrolene and cyproheptadine as salvage therapy for severe intrathecal baclofen withdrawal: A case report

Withdrawal from intrathecal baclofen can be a life-threatening situation. Despite this well-described complication, guidance for prevention and treatment when intrathecal baclofen cannot be reestablished is not well described. We report a case involving use of dantrolene and cyproheptadine for treatment of baclofen withdrawal not responsive to other therapies.

A 51-year-old man presented for intrathecal baclofen pump explantation due to a nonhealing surgical wound post catheter revision. At the time of pump removal, the patient was clinically stable and oral baclofen was scheduled to prevent withdrawal. Approximately 24 hours after pump removal, the patient developed worsening spasticity, tachycardia, and labile hemodynamics despite oral and intravenous (IV) diazepam. The patient was transferred to the intensive care unit, where midazolam and propofol continuous infusions were initiated. The patient continued to have persistent tachycardia, developed supraventricular tachycardia with hypotension, and developed hyperthermia with a maximum temperature of 39.9 °C. At that time, dantrolene 1 mg/kg IV every 4 hours for 24 hours and enteral cyproheptadine 8 mg every 6 hours were initiated as recommended by the consultant toxicologist. Over the next 4 days, both propofol and midazolam infusions were weaned as the patient's clinical status improved, and cyproheptadine was discontinued. By postoperative day 5 the patient's withdrawal symptoms had resolved, and the patient was continued on enteral baclofen and diazepam therapy.

Therapy options are limited for treatment of severe intrathecal baclofen withdrawal. High-dose dantrolene and cyproheptadine appeared to be well tolerated and improved our patient's symptoms when used as adjunctive therapy along with enteral baclofen and high-dose GABA agents.