In this report we analyse the risk factors, clinical characteristics and outcome of patients with myelodysplastic syndrome (MDS) who developed a invasive fungal infection (IFI). This was a multicentric study involving 14 Italian Haematological Divisions during a 10-year-period whose object was to identify the characteristics of patients with this infection. The study recorded 391 consecutive documented IF, 12 of which (3%) occurred in MDS patients from five of the participating centres. The primary localisation of infection was the lung in 10 cases and skin and paranasal sinus in one case each. Ten patients died at the end of the follow up. The death was mainly attributable to IFI progression in nine of them. The factors which appeared related to an unfavourable outcome were intensive chemotherapy within 30 days before IFI diagnosis, presence of multiple localisation at chest X-ray in patients with isolated pulmonary IFI and multiple sites of infection.