OBJECTIVES: To examine the demographic and clinical characteristics in patients with Cushing’s disease (CD) and to estimate the health care resource utilization associated in these patients in Ontario.  METHODS: Retrospective analysis of resource use captured in the Southwestern Ontario Database from 2001 to June 2011. A total of 86 patients (72% females) were analyzed based on diagnosis, out of a total population of 523,718 patients. A matched control group (CG) (N=86) was also included from the general population.  RESULTS: Age of patients at the time of diagnosis was 43±25.4 years (mean ± SD). Baseline co-morbidities (CM) included hypertension (67.4%), dyslipidemia (25%), diabetes (23.3%), renal calculi (17.4%), visual disturbance (20.9%), carpal tunnel syndrome (19.8%) and osteoporosis (11.6%). Distribution of co-morbidities was statistically significantly higher than general population (p-value <0.05); 27% had 2 CM and 35% had 3 or more CM. Baseline Urinary Free Cortisol (UFC) level was 207.7± 118.3 nmol/day (UFC ULN= 110 nmol/day). Primary treatment options included transsphenoidal surgery (TSS), bilateral adrenalectomy (BLA), radiosurgery and medical therapy, used in 79%, 6%, 2.3% and 12.7% of patients respectively. Secondary treatment was surgical in 37% of patients: consisting of repeat TSS in 21%, BLA in 10% and RS in 6%, while the majority received medical therapy (63%). Average length of stay for surgery was 6 days (SD=4) and 9 days (SD=7) for TSS and BLA respectively. Medical therapy, prescribed as monotherapy, included ketoconazole (38%), cabergoline (21%), bromocriptine (20%) and mitotane (15%). Health care provider interactions per year for CD post intervention compared to CG were: Emergency Room visit: 1.01 vs. 0.069; clinic visits: 4.86 vs. 1.89; specialist clinic visits: 5.57 vs. 0.92; and hospitalizations: 0.34 vs. 0.15. CONCLUSIONS: This retrospective analysis of patients diagnosed with Cushing’s disease indicates that they require substantially higher resource use and experience a high burden of comorbidities.