A case of neurohistoplasmosis successfully treated with single high-dose liposomal amphotericin B

Abstract Disseminated histoplasmosis is a leading AIDS-related opportunistic mycosis and carries high mortality risk. A simplified induction strategy using single high-dose of liposomal amphotericin B (L-AmB) has shown promise for disseminated disease, but persons with central nervous system (CNS) involvement were excluded from the clinical trial. We describe a 35-year-old man with advanced HIV disease (CD4 24 cells/µL) and a high fungal burden who was diagnosed with disseminated histoplasmosis and, subsequently, culture-confirmed CNS involvement. He was administratively withdrawn from the trial when CNS involvement was noted. He was treated within a randomized trial with a single 10 mg/kg dose of L-AmB, followed by antiretroviral therapy (ART) and itraconazole consolidation therapy, with full clinical recovery, normalization of cerebrospinal fluid biochemistry findings, and microbiological clearance. This suggests that a single high-dose L-AmB induction strategy, combined with effective ART and azole consolidation therapy, may be viable in selected patients with CNS involvement.