Context: Sjögrens syndrome (SS) is an autoimmune disease. Patients with SS experience an impaired quality of life and impose a significant economic burden on healthcare systems. Aims: This systematic literature review aimed to analyze the direct and indirect costs (ICs), as well as healthcare resource utilization (HRU) among patients with SS. Methods: A literature search was conducted in Medline and EMBASE from January 2020 to August 2024. All costs were standardized to USD and adjusted for inflation to December 2025. Results: A total of 973 citations were screened, with 14 studies meeting the inclusion criteria. These studies were conducted across ten different countries with sample sizes ranging from 12 to 32,184 individuals. Included studies report costs which varied across countries and patient subtypes. Among all patients with SS, the annual mean cost ranged from USD 2556 to USD 17,024, while the costs for patients with pSS, ranged from USD 710 to USD 14,363. sSS was associated with higher absolute costs, while pSS showed a steeper increase over time. Disease severity and comorbidities were associated with higher healthcare expenditures. One study reported annual ICs, reporting losses at approximately USD 39,061. HRU was assessed across seven studies, with outpatient visits ranging between 3.37 and 10.1 per patient per year (PPPY) and inpatient stays that varied from 0.17 to 0.59 PPPY, depending on the disease type and severity. The mean length of stay (LOS) among pSS patients ranged from 2.6 to 13.6 days, with higher emergency department (ED) visits among patients with sSS. Conclusion: This systematic review highlights the substantial and variable economic burden of SS. Costs are higher in sSS and in patients with greater disease severity or comorbidities. Evidence of rising healthcare expenditures in some regions, particularly pSS, underscores the need for improved data collection strategies and policies to address such needs and resource planning.
Umapathi et al. (Thu,) studied this question.