Economic burden of incident interstitial lung disease (ILD) and the impact of comorbidity on costs of care.

INTRODUCTION: Evidence about the economic burden related to interstitial lung diseases (ILDs) and the cost-driving factors is sparse. In the knowledge that distinct comorbidities affect the clinical course of ILDs, our study investigates their impact on costs of care within first year after diagnosis. METHODS: Using claims data of individuals diagnosed with Idiopathic Interstitial Pneumonia (IIP) (n = 14 453) or sarcoidosis (n = 9106) between 2010 and 2013, we calculated total and ILD-associated mean annual per capita costs adjusted by age, sex and comorbidity burden via Generalized Linear Gamma models. Then, we assessed the cost impact of chronic obstructive pulmonary disease (COPD), diabetes, coronary artery disease, depression, gastro-esophageal reflux disease, pulmonary hypertension (PH), obstructive sleep apnoea syndrome (OSAS) and lung cancer using the model-based parameter estimates. RESULTS: Total mean annual per capita costs were €11 131 in the pooled cohort, €12 111 in IIP and €8793 in sarcoidosis, each with a 1/3 share of ILD-associated cost. Most comorbidities had a significant cost-driving effect, which was most pronounced for lung cancer in total (1.989 pooled, 2.491 sarcoidosis, 1.696 IIP) and for PH in ILD-associated costs (2.606 pooled, 2.347 IIP, 3.648 sarcoidosis). The lung-associated comorbidities COPD, PH, OSAS more strongly affected ILD-associated than total costs. CONCLUSION: Comorbidities increase the already substantial costs of care in ILDs. To support patient-centred ILD care, not only highly cost-driving conditions that are inherent with high mortality themselves require systematic management. Moreover, conditions that are more rather restricting the patient's activities of daily living should be addressed - despite a low-cost impact.

Direct and indirect economic and health consequences related to sarcoidosis in Denmark: A national register-based study.

BACKGROUND: Sarcoidosis is a systemic disease that primarily affects the younger population. Longitudinal studies of the economic burden of sarcoidosis are scarce. This study evaluates overall the economic burden of sarcoidosis in Denmark before and after initial diagnosis. METHODS: We identified patients with sarcoidosis in the Danish National Patient Registry (1998-2010). All controls were randomly selected and matched concerning age, gender and residence. Data on direct and indirect costs, including frequency of primary and secondary sector contacts and procedures, medication, unemployment benefits and social transfer payments were extracted from national databases for patients and controls. RESULTS: We identified 9119 patients with sarcoidosis and 36,432 matching controls. Patients with sarcoidosis had significantly higher rates of health-related contacts, higher usage of medication and more received unemployment and social payments compared with controls. The total health expenses were significantly higher in the sarcoidosis group 11 years before and 11 years after diagnosis than among controls. Income from public transfer payments was significantly higher in the sarcoidosis group five years prior of diagnosis until nine years post diagnosis. Especially women between 40 and 59 years have a lower income than their controls. CONCLUSION: Sarcoidosis has a socioeconomic impact on adults especially during their prime working and providing years and this is evident several years prior to and post diagnosis. The authors advocate that further research should focus on identifying the proportion of patients with sarcoidosis that imposes the largest economic burden. Increased focus on early diagnosis and disease management may reduce the socioeconomic burden of sarcoidosis.

Inpatient Prevalence, Expenditures, and Comorbidities of Sarcoidosis: Nationwide Inpatient Sample 2013-2014.

PURPOSE: To investigate inpatient prevalence, expenditures, and comorbidities of hospitalized patients with sarcoidosis in the USA. METHODS: Patients with sarcoidosis were identified within the Nationwide Inpatient Sample (NIS) database for the years 2013 and 2014 using the respective ICD-9 diagnostic code. Data on patient and hospital characteristics, comorbidities, total hospital costs, and total hospitalization charges were collected. A propensity-matched cohort of patients without sarcoidosis from the same database was created and used as comparators for the analysis of comorbidities. RESULTS: A cohort of 78,055 patients with sarcoidosis was identified within the database, corresponding to an inpatient prevalence of 2.21 cases per 1000 admissions. Analysis of comorbidities found that patients with sarcoidosis had significantly higher odds of atrial fibrillation [adjusted odds ratio (aOR): 1.41, 95% CI 1.13-1.76, p < 0.01], conduction abnormalities [aOR: 2.04, 95% CI 1.45-2.89, p < 0.01], aortic valvulopathy [aOR: 1.78, 95% CI 1.30-2.44, p < 0.01], congestive heart failure [aOR: 1.23, 95% CI 1.04-1.45, p = 0.02], cardiomyopathy [aOR: 1.25, 95% CI 1.08-1.44, p < 0.01], deep venous thrombosis (aOR: 1.58, p < 0.01), pulmonary embolism (aOR: 1.70, p < 0.01), and osteoporosis (aOR: 1.81, p < 0.01), compared with propensity-matched patients without sarcoidosis. After adjusting for confounders, patients with sarcoidosis displayed a mean additional $1,250 (p = 0.24) in total hospital costs and a mean additional $27,205 (p < 0.01) in total hospitalization charges when compared to hospitalized patients without sarcoidosis. CONCLUSIONS: The inpatient prevalence of sarcoidosis was relatively high compared with its overall incidence. Hospitalization of patients with sarcoidosis was associated with a significantly higher total hospitalization charges compared to hospitalized patients without sarcoidosis. Patients with sarcoidosis have a higher risk of several cardiac comorbidities.

A new side of sarcoidosis: medication and hospitalization use in a privately insured patient population.

OBJECTIVE: This study describes patterns of medication prescriptions for sarcoidosis patients in a large commercially insured U.S. population, with specific focus on prescribing practices across medical specialties and their associated hospitalization risk. METHODS: Using the Marketscan Database we selected adult patients with a diagnosis of sarcoidosis by ICD-9 code during the 2012 calendar year. Differences in prescribing practices were evaluated between provider types. A multivariate model controlling for age, sex, and region assessed hospitalization risk associated with provider type, prednisone dose, and use of non-steroid sarcoidosis medications. RESULTS: Using the described criteria, 11,042 total patients were identified. A majority were female, mean age 49.3 years. Of these, 1,792 (16.2%) had one or more hospital admissions (mean 1.6, SD 1.3) with a mean length of stay of 8.1 days (SD 14.5). 25.5% of patients were prescribed prednisone with a 1 year mean cumulative dose of 250mg. Pulmonary/Rheumatology providers prescribed the highest cumulative prednisone dose (961 mg) and were more likely to prescribe methotrexate and monoclonal antibody medications. Sarcoidosis patients receiving a cumulative prednisone dose >500 mg had an increased risk for hospitalization (OR 2.512, 2.210-2.855), while those prescribed methotrexate and azathioprine had decreased risk (OR 0.633, 0.481-0.833 and 0.460, 0.315-0.671). Monoclonal antibody use was associated with increased OR for hospitalization at 1.359. CONCLUSION: Sarcoidosis patients treated by subspecialists were more likely to receive higher doses of prednisone and non-steroid sarcoidosis medications. Higher doses of prednisone and monoclonal antibody use were associated with higher hospitalization risk while methotrexate and azathioprine were associated with lower hospitalization risk.

Work ability before and after sarcoidosis diagnosis in Sweden.

BACKGROUND: Information on how sarcoidosis affects one's ability to work is needed to clarify the burden of disease on the individual and society. Our aim was to describe the work ability of individuals with sarcoidosis before and after diagnosis compared to people without sarcoidosis. METHODS: Swedish national registers were used to identify adults diagnosed with sarcoidosis aged 25-59 years old and matched general population comparators without sarcoidosis 2006-2012. Information on work loss (days of sick leave and disability pension) and earnings (gross salary) was collected from the Longitudinal Integration Database for Health Insurance and Labor Market Studies. Mean earnings and work loss days per year from 5 years before to 5 years after diagnosis were calculated. RESULTS: During diagnosis year, sarcoidosis patients (n = 3347) earned 8% less and registered 26 more work loss days than comparators (n = 33,407). The number of work loss days was higher than the general population in the years leading up to sarcoidosis diagnosis. Five years after diagnosis, the number of total work loss days decreased among sarcoidosis patients, but remained higher than that of the general population. Individuals who were older, female, less educated and received treatment at diagnosis registered the highest number of work loss days and had the largest difference in earnings at all time points. CONCLUSIONS: Individuals with sarcoidosis experience a reduction in work ability which persists for up to five years after diagnosis. Interventions targeted at getting sarcoidosis patients back to work should be evaluated.

High-Cost Sarcoidosis Patients in the United States: Patient Characteristics and Patterns of Health Care Resource Utilization.

BACKGROUND: Sarcoidosis is a multisystem inflammatory disorder characterized by the presence of noncaseating granulomas in involved organs. Prior research has found that sarcoidosis imposes a significant economic burden to U.S. payers. However, the drivers of high health care costs among sarcoidosis patients are unknown. OBJECTIVE: To characterize sarcoidosis patients who were among the top 20% of total health care costs. METHODS: Patients with a first diagnosis of sarcoidosis between January 1, 1998, and March 31, 2015 (index date) were selected from a deidentified privately insured administrative claims database. Study patients must have at least 12 months of continuous health plan enrollment prior to the index date. High-cost patients were those in the top 20% of total health care costs during the 12 months following the index date (follow-up period), and the remaining patients were classified as lower-cost patients. Patient characteristics, comorbidities, health care resource use, and health care costs in the study period were compared between the high-cost and lower-cost patients. Multiple logistic regression was used to assess the relationship between patient characteristics and being a high-cost sarcoidosis patient. RESULTS: A total of 7,173 sarcoidosis patients met the selection criteria. The 20% of patients classified as high-cost patients accounted for approximately 72% of the total health care costs in the 12-month follow-up period. Compared with lower-cost patients, high-cost patients were slightly older (50.6 vs. 49.1 years) and had a higher comorbidity burden at baseline (Charlson Comorbidity Index = 1.8 vs. 0.7). Mean annual total health care cost for high-cost sarcoidosis patients was 10 times that of their lower-cost counterparts ($73,345 vs. $7,073). Mean annual health care cost was $119,878 for patients in the 95th-99th percentile and $375,436 for patients in the top 1% of spend. High-cost patients had greater medical resource use and costs across all places of service (i.e., inpatient, emergency department, outpatient, and other) compared with lower-cost patients. Findings showed that higher total health care cost resulted in a larger proportion of inpatient spend and a smaller proportion of outpatient and pharmacy spend. Adjusting for baseline characteristics, high-cost patients were associated with a number of factors with high ORs: the presence of comorbidities such as deficiency anemia (OR = 1.606; P < 0.001), depression (OR = 1.504; P < 0.001), or cardiac arrhythmia (OR = 1.493; P < 0.001); having an inpatient admission (OR = 9.771; P < 0.001); and use of biologic therapies adalimumab and/or infliximab (OR = 31.821; P < 0.001). CONCLUSIONS: This study described the characteristics of high-cost sarcoidosis patients and identified several high-cost indicators using contemporary administrative data. The health care cost distribution for sarcoidosis patients is highly skewed, making it a worthwhile endeavor to focus improvement efforts on patients in the top quintile. The study findings can help population health decision makers identify a subset of patients for targeted interventions aimed at improving quality of care and reducing overall costs. DISCLOSURES: This study was funded by Mallinckrodt Pharmaceuticals. Rice, White, and Lopez are employees of Analysis Group, which received funding from Mallinckrodt Pharmaceuticals to conduct this research. Nelson is an employee of Mallinckrodt Pharmaceuticals. Study concept and design were contributed by Rice, White, and Nelson, along with Lopez. Lopez took the lead in data collection, with assistance from Rice and White. Data interpretation was performed by all of the authors. The manuscript was written by Rice, Lopez, White, and Nelson and revised by Rice, along with White and Nelson.

Economic burden of sarcoidosis in a commercially-insured population in the United States.

BACKGROUND: Sarcoidosis is a multi-system inflammatory disorder characterized by the presence of non-caseating granulomas in involved organs. Patients with sarcoidosis have a reduced quality-of-life and are at an increased risk for several comorbidities. Little is known about the direct and indirect cost of sarcoidosis following the initial diagnosis. AIMS: To provide an estimate of the healthcare resource utilization (HCRU) and costs borne by commercial payers for sarcoidosis patients in the US. METHODS: Patients with a first diagnosis of sarcoidosis between January 1, 1998 and March 31, 2015 ("index date") were selected from a de-identified privately-insured administrative claims database. Sarcoidosis patients were required to have continuous health plan enrollment 12 months prior to and following their index dates. Propensity-score (1:1) matching of sarcoidosis patients with non-sarcoidosis controls was carried out based on a logistic regression of baseline characteristics. Burden of HCRU and work loss (disability days and medically-related absenteeism) were compared between the matched groups over the 12-month period following the index date ("outcome period"). RESULTS: A total of 7,119 sarcoidosis patients who met the selection criteria were matched with a control. Overall, commercial payers incurred $19,714 in mean total annual healthcare costs per sarcoidosis patient. The principle cost drivers were outpatient visits ($9,050 2015 USD, 46%) and inpatient admissions ($6,398, 32%). Relative to controls, sarcoidosis patients had $5,190 (36%) higher total healthcare costs ($19,714 vs $14,524; p < 0.001). Sarcoidosis patients also had significantly more work loss days (15.9 vs 11.3; p < 0.001) and work loss costs ($3,288 vs $2,527; p < 0.001) than matched controls. Sarcoidosis imposes an estimated total direct medical cost of $1.3-$8.7 billion to commercial payers, and an indirect cost of $0.2-$1.5 billion to commercial payers in work loss. CONCLUSIONS: Sarcoidosis imposes a significant economic burden to payers in the first year following diagnosis.

Conjunctival Biopsy as a First Choice to Confirm a Diagnosis of Sarcoidosis.

BACKGROUND: Sarcoidosis is a granulomatous systemic disease of unknown aetiology. The diagnosis needs histological confirmation of the presence of non-caseating granulomata. One option is a conjunctival biopsy. The aims of this study were to evaluate conjunctival biopsy for the diagnosis of sarcoidosis with respect to its sensitivity and to assess its cost effectiveness by comparison with other histopathological diagnostic procedures. METHODS: Patients were identified from the database of the Interstitial Lung Disease Clinic (ILDC) of the Chest Department of Ege University Hospital from May 2008 to June 2014.  The patients who had biopsy procedures performed for the definitive diagnosis of sarcoidosis were assessed. Their diagnostic procedures and the cost of procedures were recorded. The cost per positive result for each procedure was calculated.  Results: In total, 280 patients were followed up with a diagnosis of sarcoidosis, of whom 174 had histological confirmation; these constitute the study population. There were 127 females and 47 males with a median age of 46 years (range 14-78 years). Forty three patients had conjunctival biopsy and we could establish a diagnosis in 54% of these by means of conjunctival biopsy. Moreover, we showed that this biopsy can provide positive result for sarcoidosis patients who lack abnormal eye findings. Additionally, it is cost effective approach and without complications. CONCLUSION: This study re-asserts the value of  conjunctival biopsy, which was described in the past but is not commonly used nowadays. In the presence of suggestive clinic and radiologic findings, we recommend conjunctival biopsy as the first choice for the histopathological confirmation of sarcoidosis.

The indirect costs of systemic autoimmune diseases, systemic lupus erythematosus, systemic sclerosis and sarcoidosis: a summary of 2012 real-life data from the Social Insurance Institution in Poland.

Systemic lupus erythematosus, systemic sclerosis and sarcoidosis are three different autoimmune systemic diseases that generate a significant burden to society due to treatment costs and also those caused by a work disability or absenteeism among patients. Relevant 2012 data referring to the three components of absenteeism produced by autoimmune systemic diseases, sick leave, short-term and long-term work disability, were obtained from the Social Insurance Institution in Poland (PSII). By applying the Human Capital Approach using gross domestic product per capita, gross value added per worker and gross income per worker in 2012, total indirect costs for the diseases were calculated. All costs were presented in euros and were valid for 2012. The PSII recorded 1600 patients with systemic lupus erythematosus, 500 patients with systemic sclerosis and 2700 patients with sarcoidosis in the 2012 - total indirect costs were as high as 7,260,595, 2,268,571 and 4,027,575 EUR, respectively. Costs were estimated using gross domestic product per capita; 17,485,412, 5,463,312 and 9,699,455 EUR, accordingly, calculated using gross value added per worker and 5,346,933, 1,670,648 and 2,966,034 EUR estimated using gross income per worker, respectively. Considering only data on absenteeism gathered by the PSII we can conclude that the three autoimmune systemic diseases bore great indirect costs. Their social burden for Poland could be even greater when considering presenteeism as well as other components of absenteeism such as loss of unpaid work, a gray economy or loss of leisure time.

Clinical value of mediastinoscopy in the diagnosis of sarcoidosis: an analysis of 68 cases.

Mediastinoscopy was performed for confirmation of the diagnosis in 68 patients who were suspected clinically and radiologically of having sarcoidosis. In 66 of 68 cases in which mediastioscopy was performed a diagnosis was attained. In 35 cases, endobronchial biopsy was performed by bronchoscopy. In only 5 of these (14.2 %) was the diagnosis of sarcoidosis confirmed. The sensitivity of mediastinoscopy was remarkably superior compared with that of endobronchial biopsy. No complication developed with either mediastinoscopy or endobronchial biopsy. In Turkey, mediastinoscopy without any complication costs about 650 USD while bronchoscopy and endobronchial biopsy cost about 150 USD. In our study in which we looked for a histological confirmation -- in the cases suspected of sarcoidosis -- mainly through mediastinoscopy and rarely through other methods (i.e., endobronchial biopsy in one case, skin biopsy in another), we did not come up with a different diagnosis. Therefore, patients suspected of having sarcoidosis should undergo a careful clinical, laboratory, and radiologic examination; they should be under continuous close observation; when necessary (e.g., skin and lip biopsy), the tissue diagnosis should be made by other methods, but if there is the possibility of a disease such as tuberculosis and lymphoma, mediastinoscopy should be performed. The diagnosis of stage 3 sarcoidosis is difficult. For diagnosis, sometimes videothoracoscopy or explorative thoracotomy may be necessary. However, in all our 3 cases with stage 3, we reached the diagnosis of sarcoidosis by the less invasive and less expensive method of mediastinoscopy. Despite our small number of cases, we believe that mediastinoscopy is a very important instrument for diagnosis of stage 3 sarcoidosis.

The clinical application and cost analysis of fine-needle aspiration biopsy in the diagnosis of mass lesions in sarcoidosis.

BACKGROUND: Sarcoidosis is a prevalent disease of unknown cause characterized by granulomatous inflammation that often creates deep and/or superficial mass lesions. Tissue samples are considered the "gold standard" in diagnosis; however, it is a medically treated disease. We analyzed the utility and relative cost-effectiveness of fine-needle aspiration biopsy (FNAB) in the clinical investigation of patients with both suspected and unsuspected sarcoidosis. METHODS: All FNAB cases with sarcoidosis either as the cytologic diagnosis or mentioned as part of the differential diagnosis were retrospectively reviewed for clinical history, follow-up, cytologic features, and surgical pathology findings. Comparative analysis of cost of FNAB and excisional biopsy were also made. RESULTS: Thirty-two FNABs in 28 patients included 17 women and 11 men. Anatomic sites included lymph node (n = 17), lung (n = 5), salivary gland (n = 8), and liver (n = 2). Sarcoidosis had already been diagnosed or was a clinical consideration prior to FNAB in 14 cases. Chest radiograph showed abnormal findings in 19 cases. Angiotensin-converting enzyme (ACE) was measured in seven patients and was elevated in four. All aspirates showed granulomatous inflammation; in 22 patients, special stains or cultures for microorganisms were negative. Simultaneous or subsequent excisional biopsies confirmed the FNAB findings in 17 patients. Institutional ratios of excisional biopsy to FNAB in the diagnosis of sarcoidosis ranged from 4 to 19:1. The cost of FNAB was only 12.5 to 50% that of tissue biopsy. CONCLUSIONS: FNAB appears to be underutilized in the diagnosis of sarcoidosis. When used in conjunction with radiologic and laboratory data, FNAB may be a reliable and cost-effective method of diagnosis, especially in patients with an established diagnosis of sarcoidosis.