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Acquired Ondine's curse: case report.

Pedro Schestatsky1, Luis Nelson Teixeira Fernandes

  • 1Neurology Department, Hospital de Clínícas de Porto Alegre, Porto Alegre RS, Brasil. lonfra@hotmail.com

Arquivos De Neuro-Psiquiatria
|July 27, 2004
PubMed
Summary
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This case study highlights Ondine's curse, a rare adult presentation of posterior stroke causing loss of automatic breathing. Despite initial recovery, the patient tragically died from complications, underscoring the condition's unpredictable prognosis.

Area of Science:

  • Neurology
  • Pulmonology
  • Critical Care Medicine

Background:

  • Ondine's curse, or congenital central hypoventilation syndrome, is a rare disorder affecting autonomic control of breathing.
  • While typically congenital, this case presents a rare adult-onset manifestation secondary to a posterior stroke.

Observation:

  • A 55-year-old male with a history of stroke and COPD presented with vertebro-basilar syndrome.
  • Brain MRI revealed a brainstem lesion affecting autonomic breathing pathways, leading to prolonged apnea episodes, particularly during sleep.
  • The patient required tracheostomy and mechanical ventilation, with subsequent treatment including medroxyprogesterone, fluoxetine, and acetazolamide.

Findings:

  • The patient was discharged after 64 days breathing independently but tragically returned the next day with aspiration bronchopneumonia, leading to septic shock and death.

Related Experiment Videos

  • This case illustrates the unpredictable clinical course and poor prognosis associated with adult-onset Ondine's curse.

    • Implications:

    • The diagnosis of Ondine's curse in adults requires flexible criteria due to rare reporting and lack of consensus.
    • Therapeutic options are symptomatic and varied, including pharmacotherapy, non-invasive ventilation, and potentially diaphragmatic pacing.
    • This case emphasizes the critical need for vigilant monitoring and prompt management of complications in patients with brainstem stroke affecting respiratory control.