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Patients with leukemia may occasionally suffer from rare opportunistic fungal infections with poor prognosis. Fungal infection caused by Geotrichum captitatum has not yet been described in Hungary. With this case report, we would like to draw attention to the fungal infection caused by G. capitatum. The 1.5-year-old girl with acute myeloid leukemia was treated for relapse diagnosed +120 days after a sibling donor bone marrow transplantation. High-grade, fluctuating fever began 11 days after the start of chemotherapy which did not decrease despite combined treatment with broad-spectrum antibiotics and antifungals (posaconasole). Due to worsening respiratory symptoms, a chest CT-scan was performed, raising suspicion of an invasive fungal infection. Blood culture confirmed G. capitatum infection. Initial empiric treatment with liposomal amphotericin B was combined with voriconazole based on international experience. However, we did not observe any improvement, and a few days later the patient passed away due to progression of the underlying disease. G. capitatum (presently known as Saprochaete capitata) is an ubiquitous yeast that can cause an infection with a poor prognosis, mainly in patients with leukemia. Its symptoms primarily appear in the skin and respiratory tract. The accurate identification of this pathogen is essential because the standard diagnostic tests do not give a specific reaction. Based on the limited international experience, the combination of amphotericin B and voriconazole can play a fundamental role in the treatment, however, even with adequate therapy 50% of the cases are fatal. By describing the first Hungarian case caused by G. capitatum, we draw attention to the importance of this rare, opportunistic fungal species with a poor prognosis that develops in immunosuppressed patients. Orv Hetil. 2023; 164(26): 1034-1038.