Disease burden of patients with pemphigus from a societal perspective.

Introduction: Cost-of-illness studies are widely used for healthcare decision-making; however, no such study is available in pemphigus from the societal perspective. The purpose of this analysis was to estimate annual cost-of-illness per patient with pemphigus from a societal perspective. Areas covered: Between 2014 and 2017, a multicenter, cross-sectional study was carried out. Consecutive pemphigus patients aged ≥18 years were recruited at all four university dermatology departments in Hungary. Direct and indirect costs were calculated, including costs for treatments, outpatient visits, hospital admissions, informal care, travel costs and productivity loss. Generalized linear model was used to analyze predictors of costs. Atotal of 109 patients with pemphigus enrolled with amean age of 57.1 (SD 14.8) years. Total cost per pemphigus patient was €3,995 (SD €7,526) peryear, with productivity loss (58%) and informal care (19%) accounting for the majority. Annual means of 189 and 41 working hours were lost due to absence from work and reduced productivity, respectively. Younger age and pemphigus vulgaris were associated with higher costs (p < 0.05). Expert opinion: This is the first cost-of-illness study applying the societal perspective in pemphigus. Our results indicate a substantial economic burden on society, mainly driven by productivity loss and informal care.

National trends in incidence, mortality, hospitalizations, and expenditures for pemphigus in Taiwan.

BACKGROUND: Data on trends in epidemiological characteristics and economic burden of pemphigus are scarce. OBJECTIVE: To describe national trends in pemphigus' incidence, mortality, hospitalizations, and expenditures between 2003 and 2015 in Taiwan. METHODS: This nationwide study used the Taiwan National Health Insurance Research Database to identify pemphigus patients from 2003 to 2015. Annual incidence, prevalence, healthcare utilization, and expenditure trends were calculated and analyzed. RESULTS: Pemphigus' incidence increased significantly from 3.19 to 4.70 per million person-years in 2003-2010 but fluctuated in 2011-2015. Pemphigus patients had higher mortality and care costs. Medical utilization and expenditure declined for pemphigus inpatients and outpatients. Systemic corticosteroid use decreased, but mortality remained stable. CONCLUSION: The health expense reduction for pemphigus was mainly attributed to decreased utilization, length of stay, and inpatient costs. The persistently elevated mortality rate highlights an unmet need in pemphigus therapy.

Associations of pemphigus or pemphigoid with autoimmune disorders in US adult inpatients.

BACKGROUND: The associations and predictors of the gamut of autoimmune conditions in pemphigus and pemphigoid have been examined in few large-scale controlled studies. OBJECTIVE: To examine associations of pemphigus or pemphigoid with autoimmune disorders and related outcomes in adults. METHODS: Data from the 2002-2012 National Inpatient Sample were analyzed, including an ∼20% sample of all US hospitalizations (n = 72,108,077 adults). RESULTS: In multivariable logistic regression models, pemphigus (adjusted odds ratio 1.46, 95% confidence interval 1.30-1.63) and pemphigoid (adjusted odds ratio 1.35, 95% confidence interval 1.24-1.48) were associated with ≥1 autoimmune disorder. Pemphigus was associated with 9 of 29 and pemphigoid with 13 of 32 autoimmune disorders examined in bivariable models. Among pemphigus inpatients, unspecified autoimmune disease, vitiligo, eosinophilic esophagitis, and myasthenia gravis had the strongest effect sizes. Whereas, among pemphigoid inpatients, unspecified autoimmune disease, vitiligo, and chronic urticaria had the strongest effect sizes. There were significant differences of autoimmune comorbidities by age, sex, and race/ethnicity. The estimated excess annual costs of hospital care attributed to autoimmune disorders among inpatients with pemphigus was $2,286,588 and pemphigoid $4,301,681. LIMITATION: Lack of treatment history data. CONCLUSION: Inpatients with pemphigus or pemphigoid had increased odds of multiple cutaneous, extracutaneous, and systemic autoimmune disorders, which were associated with a considerable cost burden.

The Inpatient Burden of Autoimmune Blistering Disease in US Children: Analysis of Nationwide Inpatient Sample Data.

BACKGROUND: Little is known about the epidemiology of pediatric autoimmune blistering disorders (PAIBD). OBJECTIVE: We sought to determine the inpatient burden and comorbidities of PAIBD. METHODS: We analyzed data from the Nationwide Inpatient Sample from 2002 to 2012, which contained a representative 20% sample of all US hospitalizations. RESULTS: The most common PAIBD with a primary admission was pemphigus (8.0 per million), whereas the most common secondary diagnosis of PAIBD was dermatitis herpetiformis (DH; 9.6 per million). Bullous pemphigoid (BP) was inversely associated with being female and having government or no insurance but positively associated with Black and Hispanic race/ethnicity and more chronic conditions. Pemphigus was associated with being female, Hispanic, having government or no insurance, and having a higher number of chronic conditions. DH was inversely associated with non-White race but positively associated with having government insurance and more chronic conditions. BP was associated with dialysis, hypertension, and diabetes. Pemphigus was associated with osteoarthritis, renal failure, hypothyroidism, and weight loss. DH was associated with herpes simplex virus infection, rheumatoid arthritis, and fungal, viral, and other skin infections. CONCLUSION: PAIBD are associated with a considerable inpatient burden and comorbid health conditions.

Costs of Care for Hospitalization for Pemphigus in the United States.

IMPORTANCE: Pemphigus is an autoimmune blistering disorder associated with significant morbidity and mortality. However, little is known about the inpatient burden of pemphigus. OBJECTIVE: To determine the incidence of and risk factors for hospitalization with pemphigus and cost of care. DESIGN, SETTING, AND PARTICIPANTS: The 2002-2012 Nationwide Inpatient Sample provided by the Healthcare Cost and Utilization Project from the Agency for Healthcare Research and Quality was analyzed. A total of 87 039 711 children and adults (mean [SD] age, 57.7 [0.98] years for those with a primary diagnosis of pemphigus; 70.6 [0.32] years for those with a secondary diagnosis of pemphigus; and 47.9 [0.19] years for those without a diagnosis of pemphigus) were studied. Data analysis was performed from June 1 to August 30, 2015. MAIN OUTCOMES AND MEASURES: Hospitalization rates, length of stay, and cost of care. RESULTS: There were 1185 and 5221 patients admitted with a primary or secondary diagnosis of pemphigus, respectively; when factoring in weights that generalize the sample to the entire hospitalized US cohort, these admissions represented weighted frequencies of 5647 and 24 880, respectively. In multivariable logistic regression models with stepwise selection, increasing age (adjusted odds ratios [95% CIs]: 18-39 years: 5.53 [4.28-7.14], P < .001; 40-59 years: 10.98 [8.46-14.24], P < .001; 60-79 years: 7.54 [5.75-9.89], P < .001; ≥80 years: 7.57 [5.71-10.04], P < .001), female sex (1.10 [1.01-1.20], P = .047), nonwhite race/ethnicity (black: 1.94 [1.75-2.14], P < .001; Hispanic: 4.10 [3.74-4.48], P < .001; Asian: 3.16 [2.68-3.73], P < .001; Native American: 2.11 [1.45-3.08], P < .001), lower household income (quartile 2: 1.19 [1.07-1.32], P < .001), being insured with Medicare (1.56 [1.41-1.74], P < .001) or Medicaid (1.55 [1.39-1.73], P < .001), number of chronic conditions (2-5: 2.36 [2.10-2.65], P < .001; ≥6: 1.47 [1.29-1.69], P < .001), hospital location in a metropolitan area (not metropolitan or micropolitan: 0.60 [0.49-0.72], P < .001), and summer season (1.12 [1.02-1.23], P = .02) were all associated with hospitalization for pemphigus. The total inflation-adjusted cost of care for patients with a primary inpatient diagnosis of pemphigus was $74 466 305, with a mean (SD) annual cost of $14 520.93 ($913.22). The inflation-adjusted cost of care for patients with a primary diagnosis of pemphigus increased significantly from 2002 to 2012 (analysis of variance, P < .001). In particular, length of stay was higher in racial/ethnic minorities compared with whites (survey linear regression, log ß [95% CI]: black: 0.076 [0.075-0.076]; Hispanic: 0.021 [0.021-0.022]; Asian: 0.037 [0.036-0.039]; Native American: 0.010 [0.0076-0.013]), lower quartile household income (quartile 1: 0.024 [0.023-0.024]; quartile 2: 0.0029 [0.0022-0.0035]), and those without private insurance (Medicare: 0.12 [0.12-0.12]; Medicaid: 0.082 [0.081-0.083]; no charge: 0.051 [0.047-0.055]). CONCLUSIONS AND RELEVANCE: There is a significant inpatient burden for pemphigus in the United States. Moreover, there appear to be racial/ethnic and health care disparities with respect to pemphigus, such that poor, nonwhite, and/or uninsured or underinsured patients have higher odds of hospitalization.

Cost and Resource Use of Pemphigus and Pemphigoid Disorders Pre- and Post-Rituximab.

BACKGROUND: Rituximab (RTX) is increasingly used for the treatment of pemphigus and pemphigoid disorders. The high cost of RTX frequently limits its use and access. OBJECTIVE: To determine the health system resources and costs associated with RTX treatment of pemphigus and pemphigoid. METHODS: Health system resources and costs attributed to a convenience sample of 89 patients with either pemphigus or pemphigoid were identified, quantified, and valued 6 months prior to and following RTX initiation between May 2006 and August 2012. Overall cohort costs and costs per patient were calculated (2013 Can$). RESULTS: The overall cohort cost for 6 months pre-RTX was $3.8 million and for 6 months post-RTX was $2.6 million. The average cost per patient decreased from $42,231 to $29,423 (30.3% decrease). The main cost driver was intravenous immunoglobulin. CONCLUSIONS: Our findings suggest that RTX is effective in reducing health system resources and the costs associated with the treatment of pemphigus and pemphigoid.