Marked improvement in autoimmune pulmonary alveolar proteinosis with severe hypoxemia in a patient treated with ambroxol: a case report

J Med Case Rep. 2015 May 6:9:100. doi: 10.1186/s13256-015-0588-6.

Abstract

Introduction: Pulmonary alveolar proteinosis is characterized by accumulation of surfactant and phospholipids in the pulmonary alveoli. Whole lung lavage is considered the first-line therapy, which requires special techniques. To the best of our knowledge, there have only been limited reports that have demonstrated the effectiveness of ambroxol on a mild case of pulmonary alveolar proteinosis.

Case presentation: A 72-year-old Japanese woman presented to our hospital with a one-year history of productive cough and progressive dyspnea. Her chest computed tomography scan showed a bilateral crazy-paving pattern in both of her lungs. She was diagnosed with autoimmune pulmonary alveolar proteinosis based on bronchoalveolar lavage findings and the presence of serum anti-granulocyte macrophage colony-stimulating factor antibodies. She was severely hypoxemic, so we recommended whole lung lavage or inhaled granulocyte macrophage colony-stimulating factor treatment, which she refused. We initiated treatment with ambroxol and her symptoms markedly improved.

Conclusions: Although whole lung lavage is the first-line therapy for pulmonary alveolar proteinosis, oral ambroxol could be an alternative treatment option, even in patients with severe respiratory compromise.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Ambroxol / therapeutic use*
  • Autoimmune Diseases / drug therapy*
  • Expectorants / therapeutic use*
  • Female
  • Granulocyte-Macrophage Colony-Stimulating Factor / administration & dosage
  • Humans
  • Hypoxia / drug therapy
  • Lung Diseases / diagnostic imaging
  • Pulmonary Alveolar Proteinosis / drug therapy*
  • Tomography, X-Ray Computed

Substances

  • Expectorants
  • Ambroxol
  • Granulocyte-Macrophage Colony-Stimulating Factor

Supplementary concepts

  • Pulmonary Alveolar Proteinosis, Acquired