BACKGROUND Lemierre’s syndrome is a potential life-threatening disease commonly occurring in young, healthy individuals. It is often preceded by an oropharyngeal infection causing bacteremia. This may rapidly progress into thrombophlebitis of the internal jugular venous system, its branches, and septic embolization and often fulminant organ failure. CASE REPORT A previously healthy 31-year-old male with recent history of facial herpes zoster infection, presented with 1-week history of increasingly painful nasal, and periorbital swelling. Imaging confirmed superior ophthalmic vein thrombosis. Staphylococcus aureus was isolated in blood cultures and had an uncomplicated hospital course with full recovery. CONCLUSIONS Early recognition of Lemierre’s syndrome contributes significantly in reducing morbidity and mortality associated with it. Staphylococcus aureus skin infection is a very rare cause of Lemierre’s syndrome, and its association with superior ophthalmic vein thrombosis has not yet been reported in literature.