We aimed to investigate the density and morphology of corneal dendritic cells (DCs) in dry eye (DE) patients with or without Sjogren’s syndrome (SS). This study included 28 patients with Sjogren’s syndrome dry eye (SSDE), 33 patients with non-Sjogren’s syndrome dry eye (NSSDE), and 30 age and sex matched healthy volunteers. In vivo confocal microscopy (IVCM) was used to investigate density and morphology (size, dendrites, and field) of DC. Compared with NSSDE and healthy group, SSDE showed significantly higher DC density, larger DC size, more DC dendrites with larger DC field (all P < 0.001). Comparison between NSSDE and healthy group demonstrated that DC density, dendrites and field were significantly higher in NSSDE. However, there was no significant difference in DC size (P = 0.076). DC density and morphological parameters showed significant associations with the systemic severity (salivary gland biopsy and serum antibodies) and ocular surface damage. The corneal epithelium DC density and morphological alterations were obvious in SSDE, which reflected higher level of immune activation and inflammatory response in SS. Marked correlations were found between DC density/morphology and systemic/ocular severity. Dynamic assessment of corneal DC may facilitate to clarify pathogenesis, stratify patient, and tailor treatment in SS patients.