Healthcare Utilization and Costs associated with Sickle-cell disease in the United States: A Retrospective Claims Analysis

Abstract

Introduction: Sickle cell disease (SCD) is a group of hereditary hematological disorders that are associated with high rate of medical resource utilization and medical costs. In the past few decades, improvement in medical care has contributed to increased survival in individuals with SCD resulting in a growing aging population which will consequently place a huge economic burden on health care resources in upcoming years. Past studies in SCD are either outdated, or focused on individual U.S. states, or focused on specific utilization categories, or had small sample size, or focused primarily on pediatric populations. An updated estimation of healthcare utilization and costs associated with sickle cell disease remedying the aforementioned shortcomings would be important to guide medical resource allocation and would be useful for various payers to anticipate and better plan for the needs of their patients along with appraising value of upcoming novel treatment options. The objective of this study was to measure the economic and healthcare resource burden associated with SCD in the United States. Methods: A retrospective cross-sectional study using matched cohorts of SCD cases and population controls was conducted using IBM® MarketScan® Commercial Claims and Encounters database. Eligible patients of all ages within both SCD and control cohorts were included if they had at least 24 months of continuous enrollment within the patient identification period from 1 January 2012 to 31 December 2017. Patients were identified as having SCD if they had at least 3 paid SCD-related claims in a given year. Control patients were obtained from the general population and were required to have no SCD diagnosis or claims during the patient identification period and were matched with SCD cases on age, sex, type of benefit plan, geographic location (state) of residence of the enrollee, and calendar year of eligibility. All-cause health care resource utilization and costs were measured during the 24-months continuous enrollment period for SCD cases and controls. Associations of SCD with cost and utilization were evaluated after adjustment of matching covariates and Charlson comorbidity index through generalized estimating equations using gamma and negative binomial distribution, respectively. Statistical analyses were conducted using SAS version 9.3 (SAS Institute, Cary, NC), STATA SE version 14.2 (StataCorp, College Station, TX) and R version 3.4.3. Results: A total of 7,446 patients with SCD and an equal number of matched population controls were identified. Nearly, half (44.7%) of the sample comprised of pediatric and adolescent patients aged 0-19. Majority of the patients were female (57.1%), from the southern region of the United States (52.7%), and covered under a Preferred Provider Organization (PPO) health plan (56.7%). SCD patients had significantly more health care resource utilization and incurred higher medical costs than controls across all service categories. After adjusting for age, sex, type of benefit plan, geographic location (state) of the enrollee, calendar year of eligibility, and Charlson Comorbidity Index, SCD patients on average had 83.20 (95%CI: 79.84–86.55), 1.18 (95%CI: 1.14–1.22), and 9.87 (95%CI: 9.08–10.67) greater outpatient visits, inpatient hospitalizations, and prescription drug dispensed than matched controls, respectively. Similarly, after adjusting for the aforementioned set of covariates, we found that SCD patients, on average had $40,657 (95%CI: $37,994 - $43,321) higher total medical costs than matched controls. Conclusion: We used real-world administrative claims data to measure the burden of SCD in a commercially insured population and found that SCD is associated with high health care resource utilization along with significant direct medical costs. Increased life expectancy among SCD patients in recent decades along with advent of new expensive treatment options like gene therapy will consequently add to the economic burden of SCD.