Acute onset rhythmic hiccup-like respirations secondary to oral baclofen toxicity

Baclofen toxicity has been associated with seizures, coma, apnea, autonomic disturbances, and cardiac conduction abnormalities. It has not been associated with rhythmic hiccup-like respirations.

We report a patient with suspected baclofen toxicity.

Our patient is a 19-year-old girl with cerebral palsy secondary to prematurity and repaired tetralogy of Fallot who had started oral baclofen 8 months before to diminish spasticity. Her main concern was the acute onset of rhythmic, deep, continual, hiccup-like breaths every few seconds, increasing in frequency with exhaustion, and disappearing in sleep. The night after her evaluation, her symptoms significantly worsened. She presented at the Johns Hopkins pediatric emergency room where her symptoms were only somewhat responsive to a benzodiazepine; she was discharged without a clear etiology. After discussion the next day, her baclofen dose was reduced. Within 12 hours, her abnormal respirations disappeared without recurrence.

Respiration involves glutamatergic excitatory synaptic input to medullary inspiratory γ-aminobutyric acid-mediated pacemaker neurons. Baclofen acts on presynaptic γ-aminobutyric acid B receptors on glutamate axons; derangement of this system may explain the irregular respirations in our patient in a dose-dependent fashion.