Successful granulocyte-macrophage colony-stimulating factor inhalation therapy for recurrent autoimmune pulmonary alveolar proteinosis after lung transplantation: A case report

Respir Investig. 2025 Jan;63(1):180-182. doi: 10.1016/j.resinv.2024.12.001. Epub 2025 Jan 4.

Abstract

Sargramostim, a recombinant human granulocyte-macrophage colony-stimulating factor (GM-CSF) inhalation therapy, was recently approved for pharmaceutical use in Japan and shows promise as a treatment for autoimmune pulmonary alveolar proteinosis (APAP). For APAP patients with severe respiratory failure due to advanced lung fibrosis, lung transplantation is also a treatment option; however, APAP may recur after the procedure. Here, we report a case of successful sargramostim inhalation therapy for post-transplant APAP relapse in a patient who underwent living lung transplantation owing to severe fibrosis. Inhaled GM-CSF may be a useful treatment option for APAP recurrence in patients who have undergone lung transplantation. CLINICAL TRIAL REGISTRATION: Pulmonary Alveolar Proteinosis GM-CSF Inhalation Efficacy Trial II registered to Japan Registry of Clinical Trials (jRCTs031220127).

Keywords: Autoimmune pulmonary alveolar proteinosis; Granulocyte-macrophage colony-stimulating factor; Lung transplant; Sargramostim.

Publication types

  • Case Reports

MeSH terms

  • Administration, Inhalation
  • Autoimmune Diseases* / drug therapy
  • Autoimmune Diseases* / therapy
  • Female
  • Granulocyte-Macrophage Colony-Stimulating Factor* / administration & dosage
  • Humans
  • Lung Transplantation*
  • Male
  • Middle Aged
  • Pulmonary Alveolar Proteinosis* / drug therapy
  • Pulmonary Alveolar Proteinosis* / etiology
  • Pulmonary Alveolar Proteinosis* / therapy
  • Recombinant Proteins / administration & dosage
  • Recurrence*
  • Treatment Outcome

Substances

  • Granulocyte-Macrophage Colony-Stimulating Factor
  • Recombinant Proteins
  • sargramostim

Supplementary concepts

  • Pulmonary Alveolar Proteinosis, Acquired