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

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).