OBJECTIVES: Compare healthcare resource utilization and costs of Cushing disease (CD) cases to CD-free controls. METHODS: A retrospective matched-cohort study design was used to analyze the administrative claims of commercial health plan enrollees with evidence of CD from 2007-2011. CD cases were matched 1:3 to CD-free controls by age, sex, region, and index year. CD cases were identified using a Cushing syndrome diagnosis code (ICD-9: 255.0) and codes for a CD-related diagnosis or procedure (e.g., pituitary neoplasm or hypophysectomy). CD cases were observed for ≥6 months after their first CD-related claim. Controls were observed starting in the same index year. Per-patient-per-month (PPPM) counts and costs of all-cause healthcare resource utilization were compared descriptively. Costs were CPI-adjusted to 2011 dollars and included health plan- and patient-paid amounts. RESULTS: Among the 885 selected CD cases and 2,655 matched controls, the mean (SD) age was 42 (14) years and 75% were female. Median follow-up was 2.4 years for cases and 1.5 years for controls. Compared to controls, cases had a higher proportion of inpatient admissions (50% vs. 11%; p<0.001), emergency department visits (61% vs. 20%; p<0.001), and outpatient visits (95% vs. 63%; p<0.001). Average monthly counts of utilization for cases were 2-4 times higher than controls: ambulatory visits (2.5 vs. 0.9; p<0.001), ED visits (0.1 vs. 0.04; p<0.001), and inpatient admissions (0.03 vs. 0.01; p<0.001). Average PPPM total all-cause costs were also higher for cases than controls ($3,224 vs. $486; p<0.001), and were largely driven by medical costs ($2,790 vs. $382; p<0.001). Average PPPM pharmacy costs were 4 times higher for cases than controls ($434 vs. $104; p<0.001). CONCLUSIONS: In this study, high healthcare resource utilization and costs were identified for CD cases compared to CD-free controls.  In addition to CD treatment costs, differences included the costs of diagnosing and treating the multiple comorbidities often observed in CD patients.