Abstract Paracoccidioidomycosis is an endemic disease in Latin America that is rarely associated with immunosuppression and biological therapy. Herein, we report for the first time a case of pulmonary paracoccidioidomycosis reactivation after infliximab treatment. A 47-year-old man from Brazil received infliximab to treat psoriatic spondyloarthropathy and presented with cough, dyspnea, weight loss, and fever. Chest computed tomography revealed a pulmonary nodule and biopsy confirmed paracoccidioidomycosis. Treatment with sulfamethoxazole and trimethoprim was initiated for fungal infection and infliximab was reintroduced two months later. Considering his clinical improvement and favorable radiologic evolution, antifungal therapy was discontinued after 29 months.