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Pulmonary alveolar proteinosis (PAP) is a rare lung disease characterized by the accumulation of large amounts of phospholipid protein-like surfactant material in the alveolar spaces, leading to severe hypoxemia and respiratory failure. The disease has an insidious onset and is difficult to diagnose in the early stages. Its clinical manifestations primarily include progressive dyspnea and coughing. Currently, whole lung lavage (WLL) is the preferred and effective treatment for PAP, as it improves alveolar ventilation function. Granulocyte colony-stimulating factor is used clinically in the treatment of a variety of disease. Nebulized granulocyte-macrophage colony-stimulating factor (GM-CSF) has a certain therapeutic effect on PAP. Here, we report a case of PAP treated with WLL combined with nebulized GM-CSF, which achieved good therapeutic effects during a 4-year follow-up period, to enhance understanding of the disease. [ABSTRACT FROM AUTHOR]