Algorithm development and the clinical and economic burden of Cushing's disease in a large US health plan database.

PURPOSE: This study aimed to develop an algorithm to identify patients with CD, and quantify the clinical and economic burden that patients with CD face compared to CD-free controls. METHODS: A retrospective cohort study of CD patients was conducted in a large US commercial health plan database between 1/1/2007 and 12/31/2011. A control group with no evidence of CD during the same time was matched 1:3 based on demographics. Comorbidity rates were compared using Poisson and health care costs were compared using robust variance estimation. RESULTS: A case-finding algorithm identified 877 CD patients, who were matched to 2631 CD-free controls. The age and sex distribution of the selected population matched the known epidemiology of CD. CD patients were found to have comorbidity rates that were two to five times higher and health care costs that were four to seven times higher than CD-free controls. CONCLUSION: An algorithm based on eight pituitary conditions and procedures appeared to identify CD patients in a claims database without a unique diagnosis code. Young CD patients had high rates of comorbidities that are more commonly observed in an older population (e.g., diabetes, hypertension, and cardiovascular disease). Observed health care costs were also high for CD patients compared to CD-free controls, but may have been even higher if the sample had included healthier controls with no health care use as well. Earlier diagnosis, improved surgery success rates, and better treatments may all help to reduce the chronic comorbidity and high health care costs associated with CD.

Incremental healthcare resource utilization and costs in US patients with Cushing's disease compared with diabetes mellitus and population controls.

PURPOSE: Resource utilization and costs in Cushing's disease (CD) patients have not been studied extensively. We compared CD patients with diabetes mellitus (DM) patients and population-based controls to characterize differences in utilization and costs. METHODS: Using 2008-2012 MarketScan® database, we identified three patient groups: (1) CD patients; (2) DM patients; and (3) population-based control patients without CD. DM and control patients were matched to CD patients by age, gender, region, and review year in a 2:1 ratio. Outcomes included annual healthcare resource utilization and costs. RESULTS: There were 1852 CD patients, 3704 DM patients and 3704 controls. Mean age was 42.9 years; 78.2 % were female. CD patients were hospitalized more frequently (19.3 %) than DM patients (11.0 %, p < .001) or controls (5.6 %, p < .001). CD patients visited the ED more frequently (25.4 %) than DM patients (21.1 %, p < .001) or controls (14.3 %, p < .001). CD patients had more office visits than DM patients (19.1 vs. 10.7, p < .001) or controls (7.1, p < .001). CD patients on average filled more prescriptions than DM patients (51.7 vs. 42.7, p < .001) or controls (20.5, p < .001). Mean total healthcare costs for CD patients were $26,269 versus $12,282 for DM patients (p < .001) and $5869 for controls (p < .001). CONCLUSIONS: CD patients had significantly higher annual rates of healthcare resource utilization compared to matched DM patients and population controls without CD. CD patient costs were double DM costs and quadruple control costs. This study puts into context the additional burdens of CD over DM, a common, chronic endocrine condition affecting multiple organ systems, and population controls.

Burden of illness, annual healthcare utilization, and costs associated with commercially insured patients with Cushing disease in the United States.

OBJECTIVE: To describe the burden of illness, healthcare utilization, and costs associated with Cushing disease (CD), a rare disorder resulting from adrenocorticotropic hormone-secreting pituitary tumors, in commercially insured patients in the U. S. METHODS: Patients with CD were identified in 2010 in the IMS Health PharMetrics and Truven Health Analytics MarketScan claims databases. Because there is no diagnosis code for CD, patients were identified with a claim for Cushing syndrome and either benign pituitary adenoma or hypophysectomy. We estimated total and CD-related utilization and costs using pharmacy and medical claims. RESULTS: We identified 685 CD patients (81% female; mean age, 41.7 years; mean Charlson comorbidity index, 1.6; mean number of chronic conditions, 4.2); 30.5% of the patients had diabetes, 22.5% had psychiatric disturbances, 21% had infections, 8.6% had osteoporosis, 8% had cardiovascular disease/stroke, 5.5% had kidney stones, and 0.7% had compression fracture of a vertebra. Patients had a mean of 19.8 office visits per year; 38.4% had inpatient hospitalizations and 34.2% visited the emergency department (ED). Patients had a mean of 3.2 CD-related office visits per year; 26.9% had CD-related hospitalizations, 0.9% had CD-related ED visits, and 36.8% had CD treatments. Mean annual total costs were $34,992 (pharmacy, $3,597; medical costs, $31,395). CD-related costs accounted for $14,310 of total costs (CD treatment costs, $9,353; other CD-related costs, $4,957). CONCLUSION: CD patients have a high burden of illness. Among CD patients in this study, 30.5% had diabetes, 22.5% had psychiatric disturbances, 21% had infections, 8.6% had osteoporosis, 8% had cardiovascular disease/stroke, and 5.5% had kidney stones. Patients had 19.8 office visits per year, and >34% of patients were hospitalized. Mean total cost of care was approximately $35,000 per year.

Management of patients with Cushing's disease: a Canadian cost of illness analysis.

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.

Budget impact of pasireotide for the treatment of Cushing's disease, a rare endocrine disorder associated with considerable comorbidities.

OBJECTIVES: Cushing's disease (CD) is a rare condition with a prevalence of roughly 39 cases per million in the general population. Healthcare costs are substantial for CD patients with either untreated or inadequately controlled disease. This study assesses the 3-year budget impact of pasireotide on a US managed care health plan following pasireotide (Signifor) availability. METHODS: Two scenarios were evaluated to understand the differences in costs associated with the introduction of pasireotide. The first scenario evaluates the budget impact of pasireotide from the perspective of an entire health plan (total budget impact) and the second from the perspective of the pharmacy budget (pharmacy budget impact). Both scenarios evaluate the annual incremental budget impact with and without pasireotide. Scenario 1 includes costs for medical procedures, drug therapies, monitoring, surgical complications, comorbidities for patients with controlled or uncontrolled CD, and adverse events. Procedures include transsphenoidal surgery, bilateral adrenalectomy, radiotherapy and radiosurgery. Drugs include pasireotide (indicated for CD), mifepristone (indicated to control hyperglycemia secondary to hypercortisolism in patients with Cushing's syndrome) as well as several off-label treatments (ketoconazole, cabergoline, mitotane). Scenario 2 considers costs solely from the perspective of a health plan pharmacy. Costs are in $2013. RESULTS: The estimated total budget impact is $0.0115 per-member per-month (PMPM) in the first year following FDA approval, $0.0184 in the second year, and $0.0194 in the third year. Introduction of pasireotide is expected to increase the pharmacy budget by $0.0257 PMPM in the first year, $0.0363 in the second year, and $0.0360 in the third year. LIMITATIONS: Model inputs rely on the small body of literature available for Cushing's disease. CONCLUSIONS: Cushing's disease is severe disease with debilitating comorbidities and substantial healthcare costs when untreated or inadequately controlled. The inclusion of pasireotide in a health plan formulary appears to have only a small impact on the total health plan or pharmacy budget.

Predictors of resource utilization in transsphenoidal surgery for Cushing disease.

OBJECT: The short-term cost associated with subspecialized surgical care is an increasingly important metric and economic concern. This study sought to determine factors associated with hospital charges in patients undergoing transsphenoidal surgery for Cushing disease in an effort to identify the drivers of resource utilization. METHODS: The authors analyzed the Nationwide Inpatient Sample (NIS) hospital discharge database from 2007 to 2009 to determine factors that influenced hospital charges in patients who had undergone transsphenoidal surgery for Cushing disease. The NIS discharge database approximates a 20% sample of all inpatient admissions to nonfederal US hospitals. A multistep regression model was developed that adjusted for patient demographics, acuity measures, comorbidities, hospital characteristics, and complications. RESULTS: In 116 hospitals, 454 transsphenoidal operations were performed. The mean hospital charge was $48,272 ± $32,060. A multivariate regression model suggested that the primary driver of resource utilization was length of stay (LOS), followed by surgeon volume, hospital characteristics, and postoperative complications. A 1% increase in LOS increased hospital charges by 0.60%. Patient charges were 13% lower when performed by high-volume surgeons compared with low-volume surgeons and 22% lower in large hospitals compared with small hospitals. Hospital charges were 12% lower in cases with no postoperative neurological complications. The proposed model accounted for 46% of hospital charge variance. CONCLUSIONS: This analysis of hospital charges in transsphenoidal surgery for Cushing disease suggested that LOS, hospital characteristics, surgeon volume, and postoperative complications are important predictors of resource utilization. These findings may suggest opportunities for improvement.

The burden of Cushing's disease: clinical and health-related quality of life aspects.

OBJECTIVE: Cushing's disease (CD) is a rare endocrine disorder characterized by excess secretion of ACTH due to a pituitary adenoma. Current treatment options are limited and may pose additional risks. A literature review was conducted to assess the holistic burden of CD. DESIGN: Studies published in English were evaluated to address questions regarding the epidemiology of CD, time to diagnosis, health-related quality of life (HRQoL), treatment outcomes, mortality, prevalence of comorbidities at diagnosis, and reversibility of comorbidities following the treatment. METHODS: a two-stage literature search was performed in Medline, EMBASE, and Science Citation Index, using keywords related to the epidemiology, treatment, and outcomes of CD: i) articles published from 2000 to 2012 were identified and ii) an additional hand search (all years) was conducted on the basis of bibliography of identified articles. RESULTS: At the time of diagnosis, 58-85% of patients have hypertension, 32-41% are obese, 20-47% have diabetes mellitus, 50-81% have major depression, 31-50% have osteoporosis, and 38-71% have dyslipidemia. Remission rates following transsphenoidal surgery (TSS) are high when performed by expert pituitary surgeons (rates of 65-90%), but the potential for relapse remains (rates of 5-36%). Although some complications can be partially reversed, time to reversal can take years. The HRQoL of patients with CD also remains severely compromised after remission. CONCLUSIONS: These findings highlight the significant burden associated with CD. As current treatment options may not fully reverse the burden of chronic hypercortisolism, there is a need for both improved diagnostic tools to reduce the time to diagnosis and effective therapy, particularly a targeted medical therapy.

Health care resource use and costs among patients with cushing disease.

OBJECTIVE: To assess health care costs associated with Cushing disease and to determine changes in overall and comorbidity-related costs after surgical treatment. METHODS: In this retrospective cohort study, patients with Cushing disease were identified from insurance claims databases by International Classification of Diseases, 9th Revision, Clinical Modification (ICD-9-CM) codes for Cushing syndrome (255.0) and either benign pituitary adenomas (227.3) or hypophysectomy (07.6×) between 2004 and 2008. Each patient with Cushing disease was age- and sex-matched with 4 patients with nonfunctioning pituitary adenomas and 10 population control subjects. Comorbid conditions and annual direct health care costs were assessed within each calendar year. Postoperative changes in health care costs and comorbidity-related costs were compared between patients presumed to be in remission and those with presumed persistent disease. RESULTS: Of 877 identified patients with Cushing disease, 79% were female and the average age was 43.4 years. Hypertension, diabetes mellitus, and hyperlipidemia were more common among patients with Cushing disease than in patients with nonfunctioning pituitary adenomas or in control patients (P<.01). For every calendar year studied, patients with Cushing disease had significantly higher total health care costs (2008: $26 440 [Cushing disease] vs $13 708 [nonfunctioning pituitary adenomas] vs $5954 [population control], P<.01). Annual outpatient costs decreased significantly for patients in remission after surgery, and there was a trend towards improvement in overall disease-related costs with remission. A significant increase in postoperative health care costs was observed in those patients not in remission. CONCLUSIONS: Patients with Cushing disease had more comorbidities than patients with nonfunctioning pituitary adenomas or control patients and incurred significantly higher annual health care costs; these costs decreased after successful surgery and increased after unsuccessful surgery.