OBJECTIVES: This SLR assessed the economic burden of SjD using published literature on direct/indirect costs, health care resource utilization (HCRU), and economic models.

METHODS: PRISMA guidelines were followed using key databases to identify cost/healthcare resource utilization (HCRU) studies in SjD from last 10 years. Conference proceedings from the last 3 years, health technology assessment (HTA) databases, and bibliography searches were used to supplement the results.

RESULTS: Of the 50 HCRU/cost studies identified, 31 fulfilled PICOS criteria (12 North America, 12 Europe, 4 Asia, 2 Middle East, 1 South America). Twenty-six studies reported HCRU, 13 studies direct costs, and 2 studies indirect costs.

Patients with SjD were compared to non-SjD matched controls across 9 studies: SjD had greater direct costs, dental costs, indirect costs, and HCRU. Cost drivers included hospitalizations (Sweden) and healthcare visits (UK); HCRU drivers included outpatient visits (Sweden) and office visits (US). In patients with SjD in the US, rheumatologist visits were greater cost drivers than ophthalmologist or neurologist visits.

Seven studies compared HCRU/costs among patients with secondary vs primary SjD, glandular vs extra-glandular manifestations, interstitial lung disease (ILD) vs no ILD, renal disease vs no renal disease, ESSDAI <5 vs ≥5, and low vs high ESSPRI. Patients with secondary SjD, extra-glandular manifestations, ILD, and renal disease had greater direct costs/HCRU. Patients with greater ESSDAI or ESSPRI scores had greater annual mean total costs (USD) and higher hospitalization rates, respectively. Recently diagnosed patients had lower ESSPRI scores, lower hospitalization rates, and lower total costs compared to patients with severe disease manifestations and comorbidities.

CONCLUSIONS: This is the first SLR investigating economic burden of SjD. The available literature suggests patients with SjD have a substantial economic burden which increases with disease and symptom severity; however, more research is required to identify which SjD characteristics are associated with higher costs/HCRU.