OBJECTIVES: To estimate the healthcare resource utilization and costs associated with Cushing’s disease (CD) in Québec. METHODS: A retrospective cohort study was conducted using data from the Quebec public health plan (RAMQ) from January 2001 to June 2011. Patients with at least two CD diagnoses (ICD9=2550) were initially selected. CD was further confirmed with at least 2 diagnoses of CD reported by an endocrinologist, or a diagnosis of pituitary adenoma, or a transsphenoidal surgery (TSS) or a bilateral adrenalectomy (BLA). Healthcare resources comprise medical/surgical services, hospitalization, emergency visits and medications. Costs of these resources were estimated annually over a three-year period from the time of diagnosis of CD and from the time of specific intervention (TSS, BLA or medical treatment). RESULTS: Of the 810 patients with two diagnoses of CD, 322 were considered confirmed cases of endogenous CD. The average age was 48.0yrs (SD=16.8) and 72.0% were females.  During the study period, TSS and BLA were performed for 23% and 21.1% of patients respectively while 11.8% had a medical treatment to control hypercortisolism. Among these patients 5.9% had two interventions and 0.9% had the three interventions. Annual costs for all patients were $14,451, $5,737 and $5,679 respectively for each of the three year following initial CD diagnosis. For patients who had a TSS, or a BLA, or a medical treatment, or a combination of these, total cost in the first year following the initial intervention varied from $12,258 to $28,888. The most frequent comorbidities were diabetes (58.4%), hypertension (57.8%), and osteoporosis (51.2%). Patients had numerous comorbidities; 85.1% had 2 or more and 69.0% 3 or more. Average annual costs of medications for the treatment of comorbidities were  $2,252 (SD=5,713). CONCLUSIONS: Results of this analysis of the RAMQ database illustrate the significant economic burden of CD and of its comorbidities.