A Systematic Literature Review on the Economic Burden of Sjögren’s Disease

BACKGROUND AND OBJECTIVE: Sjögren's disease (SjD) is a chronic autoimmune disorder characterized by exocrine glandular dysfunction, chronic fatigue, and pain, with considerable morbidity, mortality, and significant quality-of-life burden. The economic impact of SjD thus far has been underappreciated and understudied, limiting understanding of its implications for patients. Therefore, we conducted a systematic literature review to identify, evaluate, and summarize the economic burden on patients with SjD. METHODS: The systematic literature review 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 July 14, 2025. Additional searches were performed in the bibliographies of relevant studies, Google Scholar, disease-specific websites, and conference proceedings. Data regarding healthcare resource utilization (HCRU), direct medical costs, as well as indirect and societal costs were extracted. 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 49 unique studies reported outcomes related to economic burden of SjD; HCRU was reported in 27 studies, while 21 studies reported direct or indirect costs, and 16 focused on productivity loss. The studies were performed across the globe, but predominately in North America (n = 22) and Europe (n = 16). Direct healthcare costs for patients with SjD were noted to be 1.6-3.1 times higher than for patients without SjD owing to more frequent hospitalizations, outpatient visits, emergency department care, and dental visits compared with patients without SjD. In addition, total indirect costs were around 2 times higher for patients with SjD than those without SjD, a consequence of substantial productivity loss, including activity impairment (27%-61%), overall work impairment (26%-38%), absenteeism (2.4%-27%), and presenteeism (21%-35%). Patients with SjD from the focus group reported frequent emergency care for SjD-related symptoms, high out-of-pocket medical costs (which includes over-the-counter medication cost and specialized diets), significant work and daily productivity loss due to fatigue and cognitive symptoms, and ongoing financial strain requiring community or family support. CONCLUSION: This systematic review demonstrates that SjD results in measurable healthcare costs, loss of productivity, and a notable financial burden on both individuals and healthcare systems, highlighting an increased need for urgent care, hospital admissions, and extended stays. Indirect costs, primarily arising from lost productivity, further amplify the economic burden of SjD. In total, these costs highlight the economic and societal burden of SjD, reflected in both direct healthcare costs and indirect costs.