Association between neuroborreliosis and cerebral sinuvenous thrombosis is rare, and it can be made only when other, more common predisposing conditions are excluded. In the case of increased intracranial pressure and confirmed neuroborreliosis, early magnetic resonance venography and combination of antibacterial therapy with anticoagulation provide better long-term outcome. We present a case of a patient with cerebral sinuvenous thrombosis who was first treated for neuroborreliosis.