(Kiosk 6) Catatonia After Intrathecal Baclofen Pump Refill Following Acute Baclofen Withdrawal in a Young Adult with Cerebral Palsy: A Case Report

Case Diagnosis: A 20-year-old female with spastic quadriplegic cerebral palsy who developed baclofen-induced catatonia.

Case Description or Program Description: The patient presented to the emergency department in baclofen withdrawal secondary to an empty intrathecal baclofen (ITB) pump. Her spasticity was managed well with an ITB pump for several years prior to presentation. Pump was refilled uneventfully, and her symptoms improved after 1 week. She represented 10 days later with staring spells, dysphagia, and psychomotor retardation. Comprehensive imaging and laboratory workup was unremarkable, and she was diagnosed with catatonia. Her symptoms worsened despite decreasing ITB dosage to minimum rate and initiation of enteral baclofen. She was started on lorazepam and temazepam and discharged after symptom improvement. 3 weeks later, she returned with worsening dysphagia. A nasogastric tube was placed, enteral baclofen was discontinued, and ITB pump was turned off. Temazepam was increased, and she was started on sertraline for agitation and anxiety which improved symptoms. At 1 month post-discharge, she had improvement in staring, sleep, and secretion management but still had delayed response to verbal questioning. She was continued on lorazepam, temazepam, and sertraline. Tone in all extremities was increased from baseline.

Setting: Tertiary care pediatric hospital.

Assessment/Results: Approximately 5 months following initial presentation, she had near complete resolution of catatonic symptoms with only mild delay in speech response noted on exam. She remains on a benzodiazepine wean for her catatonia which may also be partially treating her tone.

Discussion (relevance): This is the fourth reported case, to our knowledge, of baclofen-induced catatonia and second reported case of ITB-induced catatonia. Though catatonia is poorly understood, the theorized pathophysiology involves abnormalities within the GABA neurotransmitter system, which overlaps with baclofen’s GABA-B agonist activity. It is unclear why she developed a catatonic reaction to baclofen despite receiving the medication previously.

Conclusions: Catatonia is a rare adverse effect following reinstitution of ITB after experiencing baclofen withdrawal.