Abstract
BACKGROUND: Lomentospora prolificans is an opportunistic fungal pathogen associated with significant morbidity and mortality in immunocompromised individuals. Its intrinsic resistance to most antifungal agents poses challenges in clinical management, particularly in patients with hematological malignancies. CASE SUMMARY: Herein, we report the case of a 60-year-old male with recently diagnosed acute monocytic leukemia who developed a disseminated L. prolificans infection during induction chemotherapy. Despite standard prophylactic antimicrobials, the patient experienced febrile neutropenia, progressive pulmonary findings, and neurological complications, culminating in disseminated fungal infection. Blood cultures confirmed the diagnosis, and antifungal therapy was promptly expanded. However, the infection progressed rapidly, and the patient died despite aggressive therapeutic interventions. CONCLUSION: This case highlights the complexities of diagnosing and treating L. prolificans infections in profoundly immunosuppressed patients. It underscores the importance of heightened clinical suspicion, early recognition, aggressive management, and the need for continued development of novel antifungal agents and diagnostic techniques.