An 8-year-old female patient presented with progressive bilateral vision loss, headache, and psychiatric symptoms. Imaging studies showed multiple hyperintense lesions in the brain and optic nerve. Cerebrospinal fluid (CSF) analysis confirmed the presence of Paracoccidioides. For confirmation, a culture suspension was inoculated into the testicles of guinea pigs, which developed orchitis and meningoencephalitis. Pieces of guinea pig organs (brain, liver, and prostate) were seeded in enriched media, and the same strain isolated from the patient’s CSF was recovered. Western blotting of the patient's CSF demonstrated a 43-KD protein band characteristic of Paracoccidioides.

The patient was extensively investigated for immunosuppression, but had a normal hemogram, lymphocyte subsets, and immunoglobulins. She was treated with fluconazole 50 mg/12 h p.o. and itraconazole 100 mg/12 h p.o. for 6 years.

 Her vision gradually improved, and imaging revealed lesion reduction and resolution. The patient remained asymptomatic even after treatment discontinuation

Neuroparacoccidioidomycosis should be considered as a differential diagnosis in patients presenting with blindness and neurological symptoms. Prolonged oral antifungal therapy is the mainstay of treatment. This case underscores raises awareness about paracoccidioidomycosis and its neurological complications, even in non-endemic regions.