Neurologic manifestations of paracoccidioidomycosis are uncommon. Generally, these are characterized by headache, seizures, or focal neurologic deficits due to a focal brain lesion. We describe a patient with subacute meningoencephalitis, CSF evidence of paracoccidioidomycosis, and striking abnormalities on brain CT scan. Combined fluconazole and sulfamethoxazole/trimethoprim was started with good response. The patient is asymptomatic after 10 years. Cerebral paracoccidioidomycosis should be included in the spectrum of possible causes of meningoencephalitis in a patient from an endemic region. Paracoccidioidomycosis is endemic to Latin America. In Brazil, it is the most common deep mycosis and is prevalent mainly in rural areas, with an estimated annual incidence of 1 to 3 cases in every 100,000 inhabitants.1 In a few patients the infection results in overt disease that evolves into one of the two clinical forms: juvenile or adult type paracoccidioidomycosis. The CNS is affected in very few patients, mainly in those with the adult form.2–5 Generally, cerebral paracoccidioidomycosis is characterized by hypodense focal brain lesions with mass effect and contrast enhancement, resulting in focal neurologic deficits, headache, and seizures. Meningoencephalitis with psychiatric symptoms in the absence of meningeal signs or focal brain lesions has not been reported in cerebral paracoccidioidomycosis. We describe a case of meningoencephalitis and psychiatric manifestations secondary to Paracoccidioides brasiliensis infection in a patient with juvenile paracoccidioidomycosis without focal brain lesions on CT scan.