Objective: Pleural effusion secondary to blastomycosis infection is an uncommon clinical manifestation of the disease. We undertook a retrospective study to assess the incidence and involvement of pleural effusion in patients with blastomycosis infection. Study Design: Institutional cytology and surgical pathology records were searched from December 1995 to October 2017 for cases of blastomycosis. The cytologic, surgical pathology, and clinical pertinent information was reviewed in detail. Results: A total of 77 cases of blastomycosis infection were recorded, with a male-to-female ratio of 1.7: 1.0. Forty-eight cases of blastomycosis were pulmonary (62.3%), while 29 cases of blastomycosis were found in extrapulmonary sites (37.7%). The diagnosis of pulmonary blastomycosis was established by 24 lung biopsies/ wedge resections, 22 bronchial alveolar lavages, and 2 lung fine needle aspirations. The 29 cases of extrapulmonary blastomycosis included 13 cases of bone (44.8%), 8 cases of skin