title: Management of patients with Cushing's disease: a Canadian cost of illness analysis.
abstract: BACKGROUND
  Cushing's disease (CD) is a rare disorder caused by increased pituitary secretion of adrenocorticotropic hormone (ACTH) resulting in elevated production of cortisol. It is associated with multiple adverse cardiovascular, metabolic, musculoskeletal and mental consequences. Patients with CD require substantial health care resources both in terms of treatments with a curative intent and control of disease related co-morbidities. In this study, a cost of illness analysis was conducted to estimate the direct cost of CD care in Canada.
  
  
  METHODS
  This was a retrospective cohort study of 86 CD patients. Data collection included patient demographic and disease related information, existing comorbidities, treatments received and all clinical outcomes. In addition, healthcare resource utilization to manage CD was also collected. Once the mean cost per patient was determined, the overall disease prevalence was used to estimate the total direct cost of illness in Canada.
  
  
  RESULTS
  The sample included 86 CD patients, with a mean age of 43 years at diagnosis, 72% were female. All received a first line intervention consisting of transsphenoidal pituitary surgery (78%), bilateral adrenalectomy (5%), radiation therapy (5%) or medical therapy ± radiation (13%). In addition, 18 and 14 patients subsequently received a second and third line intervention, respectively. The mean cost was $85,946 per patient over the first three lines of therapy. Combining this estimate with the reported disease prevalence (5.5 patients per 100,000 [95%CI: 4.2 to 6.8]), the total direct cost of CD in Canada was estimated to be approximately $80.6 million (95%CI: $61.5 to $99.6 million) over the first 3 lines of therapy.
  
  
  CONCLUSIONS
  CD is a debilitating condition that is associated with substantial health care costs. Strategies that provide clinical cure or long term disease control need to be identified to reduce patient morbidity and to save health care costs in patients who remain uncontrolled.
authors.forename: S Van
authors.initials: SV
authors.lastname: Uum
authors.forename: M
authors.initials: M
authors.lastname: Hurry
authors.forename: R
authors.initials: R
authors.lastname: Petrella
authors.forename: C
authors.initials: C
authors.lastname: Koch
authors.forename: G
authors.initials: G
authors.lastname: Dranitsaris
authors.forename: A
authors.initials: A
authors.lastname: Lacroix
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D015897:Comorbidity; D003362:Cost-Benefit Analysis; D016208:Databases, Factual; D016527:Drug Costs; D004636:Emergency Service, Hospital / Q000191:economics; D005260:Female; D017048:Health Care Costs; D006295:Health Resources / Q000191:economics* / Q000706:statistics & numerical data; D017721:Hospital Costs; D006760:Hospitalization / Q000191:economics; D006801:Humans; D008297:Male; D008875:Middle Aged; D018803:Models, Economic; D009819:Office Visits / Q000191:economics; D009864:Ontario / Q000453:epidemiology; D047748:Pituitary ACTH Hypersecretion / Q000175:diagnosis / Q000191:economics* / Q000453:epidemiology / Q000628:therapy*; D015995:Prevalence; D012189:Retrospective Studies; D016896:Treatment Outcome; D055815:Young Adult