RESUMO
Lomentospora prolificans is an opportunistic pathogen that can cause invasive lomentosporiosis in immunocompromised patients. Patients with hematological malignancies and those who have undergone stem cell or solid organ transplantations are in the highest risk group. In addition to the limitations and delays in diagnostic possibilities, L. prolificans has a high mortality due to its resistance to all available antifungal drugs. In a patient diagnosed with aplastic anemia, we described the first case of L. prolificans in Türkiye. L. prolificans was identified in the blood culture, and despite the initiation of antifungal treatments, the fungemia resulted in mortality on the 7th day of intensive care hospitalization. This case highlights the importance of early recognition and prompt initiation of appropriate antifungal therapy to improve the outcome of patients with rare mold infections.