A case of invasive Geotrichum capitatum infection is reported; a young patient had an acute leukemia for which he received a chemotherapy, and presented sepsis with blood cultures for Geotrichum capitatum, namely Dipodascus spicifer; this pathogen only described in cactus rot, is responsible for the first case of a human disseminated infection reported in literature. Then he developed a splenic and epididymic infection, with positive cultures for Geotrichum capitatum after splenectomy and castration. Treatment with amphotericin B and itraconazole was started with low minimal inhibitory concentration (0.1 microgram/ml). The patient died of massive hemoptisis. Autopsy findings demonstrated a lung, brain and kidneys seeding.