Aim: To present clinico-pathological findings in a case of pulmonary alveolar proteinosis complicated by fungal infection. Case Report: A 48-year-old female presented with progressively increasing exertional breathlessness for the past 6 months. On imaging she showed extensive air space filling with crazy paving pattern in the left lung with multiple nodular lesions in the right lung and mild bilateral pleural effusions. Core needle biopsies of the lung showed features consistent with pulmonary alveolar proteinosis in the left lung and fungal infection in the right lung. Discussion: Infections with unusual organisms can complicate pulmonary alveolar proteinosis (PAP) due to inherent alveolar macrophage dysfunction and intra alveolar accumulation of surfactant offering a good culture medium for the microbes. The risk is further increased if such patients are treated with steroids. Conclusion: Opportunistic infections can complicate the clinical course of PAP which is associated with high mortality. High index of suspicion, early diagnosis and aggressive treatment can prevent the adverse outcomes