RESUMO
BACKGROUND: Cryptococcus spp. is a rare cause of ventriculoperitoneal shunt (VPS) infection, with a variable clinical presentation. Diagnosis and treatment of this entity are challenging. CASE DESCRIPTION: A cryptococcal VPS infection occurred in a human immunodeficiency virus-infected patient with an excellent immunovirologic status, with an abdominal mass as the only clinical sign at presentation. Microbiologic diagnosis was confirmed when Cryptococcus neoformans was isolated in 4 cerebrospinal fluid samples on different days. The patient was treated with dual antifungal therapy (liposomal amphotericin B plus flucytosine). The VPS was initially externalized and then removed. At 12-month follow-up, the patient remained asymptomatic, and no replacement VPS was required. CONCLUSIONS: This is the first reported case of cryptococcal VPS infection in a patient with human immunodeficiency virus infection. Clinical outcome was excellent after dual antifungal therapy plus device withdrawal. Diagnosis and treatment of this entity remain a challenge for clinicians.