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Introduction: Moesziomyces spp. are connected with poaceous plants (Grass family), and Moesziomyces bullatus is commonly associated with smut in pearl millet. Currently, the scientific knowledge of this pathogen is limited to only a few taxonomic studies and there are no clinical reports that describe the isolation of M. bullatus from humans. Case presentation: A female neonate born prematurely at 32 weeks of gestation was referred to the University Teaching Hospital in Jos, Nigeria, with a provisional diagnosis of preterm low birth weight at risk for sepsis. The birth weight of the newborn was 2000 g and her body temperature on admission was 34.3 °C. Blood cultures revealed the presence of a fungal isolate that was identified as M. bullatus by molecular methods. This fungus showed high MIC values for anidulafungin, caspofungin and micafungin, as well as fluconazole and 5‐flucytosine, and exhibited varying degrees of susceptibility to itraconazole, amphotericin B, posaconazole and voriconazole. To the best of our knowledge, this is the first case of a human M. bullatus bloodstream infection. Conclusion: Here, we report the first case of an unusual human infection caused by the fungal plant pathogen M. bullatus and highlight a high level of resistance to classical and modern antifungal drugs

First report of neonatal sepsis due to Moesziomyces bullatus in a preterm low‐birth‐weight infant