Herpes zoster results from reactivation of the varicella zoster virus (VZV). Zoster sine herpete (ZSH) is an uncommon manifestation of VZV infection and presents with similar symptoms but without the vesicular rash. We describe an unusual case of lateral sinus thrombosis (LST) that developed during the clinical course of ZSH in the C2 distribution. A 55-year-old woman presented with a 3-day history of left temporal and postauricular pain, nausea, vomiting, and mild photophobia. She denied otalgia, otorrhea, and hearing loss. Examination revealed hyperesthesia in the left C2 nerve root distribution without evidence of herpetic rash. A computed tomography scan showed minimal fluid in the left mastoid cavity (not mastoiditis) and thrombus within the left lateral and sigmoid dural sinus. Magnetic resonance imaging and magnetic resonance angiogram confirmed these findings. Laboratory studies revealed elevated neurotrophic immunoglobulin G levels to VZV. Hypercoagulable studies were normal. She was subsequently treated with Neurontin, acyclovir, and anticoagulation. Her symptoms improved, and she was discharged 3 days later. LST is generally a complication of middle ear infection. Nonseptic LST, however, may result from dehydration, oral contraceptive use, coagulopathy, or thyroid disease. This unusual case raises the suspicion that thrombosis resulted from VZV associated thrombophlebitis in the ipsilateral cerebral venous sinuses along the second cervical nerve root distribution. A high index of suspicion is necessary in such cases so that a different treatment course can be identified and antiviral medication initiated promptly.