Multisystem Lomentospora prolificans progression on fosmanogepix despite in vitro activity: a call for expanding clinical research on novel antifungals

January 29, 2026
Case Report
Ryan A Cooper, Sana Arif, Arthur W Baker, Nathan P Wiederhold, Stefanie Sarantopoulos, Mustafa Iftikhar, Kelly Stanly, Brennan Collis, Bin Ni, Beatrice Z Sim, John R Perfect, Barbara D Alexander, Madeleine R Heldman
Pathogen:Lomentospora prolificans
Infection Type:Multisystem fungal infection
Pathogen Type:Fungus

Summary

A severely immunocompromised patient with acute myeloid leukemia developed multisystem Lomentospora prolificans infection, presenting with skin lesions, pulmonary nodules, and eye involvement. Despite initial in vitro susceptibility to fosmanogepix and its administration, the infection progressed, leading to the patient's death. Diagnosis was confirmed through biopsy and culture from multiple sites. This case highlights the critical challenge of treating highly resistant fungal infections like L. prolificans, even with novel antifungals showing in vitro activity. The discrepancy between in vitro susceptibility and clinical outcome underscores the urgent need for expanded clinical research into new antifungal agents and their real-world efficacy.

Key note: In vitro susceptibility to novel antifungals does not always predict successful clinical outcomes in severely immunocompromised patients with highly resistant infections.

DOI: 10.1128/asmcr.00189-25