Neuroparacoccidioidomycosis is the central nervous system involvement by Paracoccidioides brasiliensis, a condition that may be underdiagnosed and has been scarcely reported. We describe a case of neuroparacoccidioidomycosis in a diabetic male with antecedent of heavy cigarette smoking and alcohol abuse. Thirty years before, he lived in the Amazon area and exerted rural activities in more recent years. The patient's main complaints were headache, visual deficit, hemiparesis, and weight loss. Imaging studies detected changes in the lungs and right adrenal gland, in addition to brain lesions. Paracoccidioides brasiliensis was found in tissue samples collected by the lung and brain biopsies. The patient is under ambulatory surveillance and in use of trimethoprim-sulfamethoxazole.