Pulmonary alveolar proteinosis is a rare disease characterized by the deposit of lipid-rich proteinaceous material in alveolar spaces. The only effective treatment known to date has been periodic alveolar lavage, a technique that is laborious and requires that the patient be under general anesthesia, with selective endotracheal-bronchial intubation. Complications are not unknown. Progress in our understandint of the pathogenesis of pulmonary alveolar proteinosis has shown it to be related to changes it to be related to changes in alveolar macrophagocytic capability. We describe a patient in whom 8 weeks of subdermal GM-CSF treatment led to significant clinical, radiologic and lung function improvement.