Background: Scedosporium species are increasingly recognized as a cause of invasive mould disease in haematology patients, but little is known about the hospitalization costs and outcomes attributable to invasive scedosporiosis (SCEDO). Methods: A retrospective case-control study was undertaken during 2002-10 to determine the attributable inpatient costs, length of stay (LOS) and mortality associated with SCEDO in haematology patients. Case patients with SCEDO (n = 30) were matched 1 2 to controls (n = 60) according to haematological diagnosis, admission year and age. Diagnostics, antifungal drugs, ward and other SCEDO-related costs were estimated using actual cost data. Median regression modelling was used to adjust for variables that were not accounted for in the matched-pairs analysis. Results: The crude total median cost of treating SCEDO was AU 32 182 per patient versus AU 17 424 per control. In multivariable analysis, SCEDO was associated with median excess costs of AU 23 611 (95 CI = AU 17 992-AU 29 231; P <0.001), approximating US 15 509 at purchasing power parity, with prolonged LOS of 13 days (95 CI = 8.2-17.8 days; P <0.001). Exclusion of cases and matched pairs with early death further increased the median excess cost and LOS. The cost differential was driven by ward costs (64 , P = 0.005) and antifungal treatment costs (29 , P <0.001). The all-cause inpatient mortality was 38 times higher for the SCEDO cases versus the control group (63.3 versus 1.7 ; P <0.001). Conclusions: SCEDO has substantial impact on hospital resource consumption, LOS and mortality in haematology patients. Risk factors and preventative measures for SCEDO should be further studied.