Background/Purpose: Sjögren’s disease (SjD) is a chronic systemic autoantibody driven disorder characterized by glandular dysfunction that typically presents with oral and ocular dryness, fatigue, and pain. Ninety percent of affected individuals are female, and the condition imposes significant clinical, humanistic, and economic burden. Limited information is known regarding the total economic burden associated with SjD including direct, indirect, or societal costs and the implications for patients living with SjD. We conducted a systematic literature review (SLR) to identify, evaluate, and summarize the economic burden on patients with SjD. The SLR was supplemented by a roundtable discussion of patients living with SjD to add additional insight and context regarding the burden of disease.

Methods: The SLR was developed using the PRISMA reporting guidelines for systematic reviews and conducted using the MEDLINE and Embase databases (via OvidSP), covering the period from January 1, 2014 to April 12, 2024. Additional searches were performed in the bibliographies of relevant studies, Google Scholar, disease-specific websites, and conference proceedings. Data were extracted to report on healthcare resource utilization (HCRU), direct medical costs, as well as indirect and societal costs. A focus group discussion of patients living with SjD generated qualitative insights to identify themes across diagnosis, treatment, and burden of disease.

Results: A total of 53 publications (43 unique studies) reported outcomes related to economic burden of SjD. The studies were performed across many countries, predominately in North America (23) and Europe (13). The publications reflected 23 studies reporting on HCRU, with 18 assessing direct and/or indirect costs. Direct health care costs for patients with SjD were, on average, 2.8 times higher than for patients without SjD (Table 1). Direct patient out-of-pockets costs were not measured in any study. The studies underscore that patients with SjD require more frequent hospitalizations, outpatient visits, emergency room care, and dental visits compared to the patients without SjD. Productivity loss was reported in 15 studies. On average, patients with SjD had two times higher total indirect costs (Table 2). The studies also highlighted significant societal costs associated with SjD, including a higher likelihood of early retirement, reduced work hours, and part-time work due to health. The patients interviewed confirmed they were unable to work due to disease impact and fatigue as well as an inability to afford living alone and experiences of financial insecurity.

Conclusion: Current published studies demonstrate that SjD results in considerable healthcare costs, loss of productivity, and a profound financial burden on both individuals and healthcare systems, highlighting an increased need for inpatient care and extended stays. Indirect costs, primarily arising from lost productivity, further amplify the economic burden of SjD. In total, these costs highlight a significant economic and societal burden of SjD, reflecting both direct healthcare costs and significant indirect costs.

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ACR Meeting Abstracts - https://acrabstracts.org/abstract/a-systematic-literature-review-on-the-economic-burden-of-sjogrens-disease/