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Patients with acquired immunodeficiency syndrome (AIDS) are potentially at increased risk for developing secondary pulmonary alveolar proteinosis because of underlying immunosuppression and frequent opportunistic lung infections. This condition. however, has been diagnosed uncommonly in these patients and, with the exception of one previously reported case. only in the presence of concurrent pulmonary infection. The case of a 35-year-old male with AIDS who was found on open lung biopsy to have pulmonary alveolar proteinosis without evidence of associated lung infection is presented.