OBJECTIVES: Treatment of Cystic Fibrosis (CF) as rare disease is generally perceived as highly expensive, but we did not have more specific national or local costs data. Since patients are on lifelong treatment we tried to analyzed direct costs associated with CF in single center.   METHODS: We analyze direct costs associated with treatment management of patients with CF in selected center in Slovakia from 2009 till 2013. We reviewed 182 adult patients regularly visiting CF centrum. We distributed patients according to age that is closely connected with CF prognosis. We included: treatment costs, healthcare procedures, medical devices and hospitalization costs. We used perspective of healthcare provider. We chose cost-of-illness, prevalence-based approach and compared costs based on time/year, gender, age and type of procedure.         RESULTS: We have found that adult patients with CF between age 30 and 35 (21 patients) consumed most resources from 2009 till 2012, but most significantly in 2009: 73% of all costs, cumulatively 300.805,52 EUR (2009-2013). Total average costs for period of 5 years (2009-2013) per 1 patient was 48.884,01 EUR. Year-by-year average costs for 1 patient were as follows: in 2009 24.795 EUR, in 2010 4.682 EUR, in 2011 3.273 EUR, in 2012 3.630 EUR and in 2013 12.456 EUR. Most influential cost factor was average hospitalization/1 patient that dramatically decreased between 2009 and 2011 (16.651 EUR vs 2.009 EUR) and than increase between 2011 and 2013 (2.009 EUR vs 10.531 EUR). We also found negative significant statistical correlation between age of patients and costs. CONCLUSIONS: We performed first direct costs analysis in patients with CF in single center. More data are needed to be able to evaluate exact costs associated with CF treatment that should serve as solid background for decision making process at national level.