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

Siddharth Srivastava, Alexander Hoon, Jean Ogborn, Michael Johnston

Research output: Contribution to journalArticlepeer-review

2 Scopus citations


Objective 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. Method We report a patient with suspected baclofen toxicity. Results 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. Conclusions 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.

Original languageEnglish (US)
Pages (from-to)252-254
Number of pages3
JournalPediatric Neurology
Issue number2
StatePublished - Aug 2014


  • abnormal breathing
  • baclofen
  • toxicity

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Neurology
  • Developmental Neuroscience
  • Clinical Neurology


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