Disseminated fungal infection is an important cause of morbidity and mortality among patients with hematological malignancies. Ochroconis gallopavum is a dematiaceous and thermotolerant fungus that causes opportunistic infections in immunocompromised hosts. About only 30 cases of this organism infection have been reported worldwide. We report a disseminated Ochroconis gallopavum infection in a B-cell chronic lymphocytic leukemia patient. In spite of intensive anti-fungal treatment, no improvement in the clinical condition was observed and the patient died 4 months after diagnosis of the infection. Ochroconis gallopavum infection is a potentially fatal disease in hematological malignancies.