OBJECTIVES: Cushing’s disease (CD) is a rare disorder resulting from adrenocorticotropic hormone secreting pituitary tumor; limited US data are available on health care utilization for this rare condition. We evaluated health care utilization and costs associated with CD. METHODS: We identified CD patients in 2010 in claims databases (Thomson Reuters MarketScan Commercial; IMS Health PharMetrics). Since no CD diagnosis code exists, patients were identified with a claim for Cushing’s syndrome and either benign pituitary adenomas or hypophysectomy (adenectomy) and continuous-enrollment in the study year. We estimated total and CD-related utilization and costs using pharmacy and medical claims. CD-related outcomes were determined using claims with primary diagnosis of Cushing’s syndrome, benign pituitary adenoma, or claims with CD treatment (pharmacologic/surgery/radiotherapy).  RESULTS: We identified 685 CD patients across US (mean age: 41.7years; 81% female; mean Charlson-comorbidity: 1.6; mean no. chronic conditions: 4.2); 30.5% (209) patients had diabetes, 22.5% had psychiatric disturbances, 8.6% had osteoporosis, 8% had cardiovascular disease/stroke, 5.5% had kidney stones, and 0.7% had compression fracture of vertebra. 38.4% patients had inpatient hospitalizations and 34.2% had emergency department visits. Patients had a mean of 19.8 physician office visits. CD-related hospitalization was observed in 26.9%, emergency department visits in 0.9%, CD-treatment in 36.8%. Patients had mean of 3.2 CD-related visits. Mean overall costs were $34,992 (SD:$45,811; median:$18,031): $3,597 (SD:$6,323) were pharmacy and $31,395 (SD:$44,082) were medical costs. Of this total, CD-related costs accounted for $14,310 (SD:$25,161; median:$2,079): $9,353 (SD:$19,259) from treatment and $4,957 (SD:$11,805) from non-treatment costs. CONCLUSIONS: Economic burden of CD is substantial, with hospitalizations or emergency department visits observed in >34% patients, 19.8 office visits/patient, and up to $35,000 in annual total cost.  Direct CD-related costs are likely underestimated because of under-coding of CD in claims and use of primary diagnosis claims only.  Future research should further examine CD-comorbidity costs and impact of long-term comorbidities of CD.