Pulmonary alveolar proteinosis can be secondary to inhaled dust exposure, malignancy, and chronic pulmonary infections. However, pulmonary alveolar proteinosis secondary to extrapulmonary aspergillosis has never been reported. We report herein a case of pulmonary alveolar proteinosis secondary to invasive rhinocerebral aspergillosis. Neither immune modulators nor whole lung lavage was applied during the treatment course. The severe respiratory distress subsided, hypoxia resolved, and radiological infiltrates improved following the successful treatment of invasive rhinocerebral aspergillosis alone.
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