Limited data are available on healthcare resource use and costs in patients with sarcoidosis.The primary aim of this study was to describe cost-drivers of the top 1% and top ≥1-5% high-cost patients with sarcoidosis. The secondary aim was to compare costs of patients with and without fatigue complaints and to compare comorbidities.We conducted a retrospective observational cross-sectional study in 200 patients diagnosed with sarcoidosis. Hospital administrative databases were used to extract healthcare utilization on the individual patient level. Healthcare costs were categorized into nine groups.Average total health care costs for the top 1% (n=22), top ≥1%-5% (n=88) and bottom 95% beneficiaries (n=90) were € 108.296, €53.237 and €4.817, respectively. Mean treatment time in days for the top 1%, top ≥1-5% and the random sample of the bottom 95% was 1688 days (±225), 1412 days (±367) and 775 days (±659), respectively. Mean annual costs for the top 1%, top ≥1-5% and the random sample of the bottom 95% are €51.082, €27.840 and €8.692, respectively. We identified three cost-drivers in the top 5% high-cost patients: 1) expensive medication, 2) intensive care and 3) costs made at the respiratory unit. Patients with and without fatigue showed to have comparable mean costs. High-cost patients were more likely to have multiple organs involved due to sarcoidosis.We identified expensive medication as the main cost-driver in the top 5% high-cost patients with sarcoidosis. The study findings can help to tailor interventions for improving the quality of care and reducing overall costs. (Sarcoidosis Vasc Diffuse Lung Dis 2020; 37 (3): e2020002).