Autoimmune pulmonary alveolar proteinosis (PAP) is a rare lung disease. Although recombinant human granulocyte macrophage colony-stimulating factor (GM-CSF) therapy has emerged as a new therapeutic modality, whole-lung lavage (WLL) with manual chest percussion has been a standard therapy in advanced cases. The application of biphasic cuirass ventilation (BCV) instead of chest percussion has rarely been reported. We describe the case of a patient with advanced PAP who recovered well in both lungs, without complication, after we performed WLL with BCV under anesthetic mechanical ventilation. Both radiographical chest findings and clinical symptoms were improved, and oxygen therapy was finally withdrawn. This case illustrates that BCV can enhance the effective removal of lavage fluid and is an alternative to manual percussion.