Varicella Zoster virus (VZV) is known to cause a benign self-limiting exanthem in children rarely with any neurological manifestations. However, with increasing incidence of VZV in adult population, severe neurological manifestations have been reported with cerebellar ataxia and encephalitis being the most common. VZV is also associated with cerebral vasculopathy including cerebral artery stenosis, vasculitis, large artery disease, aneurysm, subarachnoid hemorrhage and cerebral venous sinus thrombosis (CVST). Venous thrombosis secondary to VZV is rare and may occur both during primary infection as well as reactivation of virus.

This is a case of CVST as a rare complication of Varicella Zoster infection. A young male with no significant medical history presented with generalized tonic clonic seizures preceded by vesicular rash on the face diagnosed as Varicella zoster infection. The MRI showed bilateral superficial hemorrhagic infarcts involving frontal and temporal lobes and MR Venogram (MRV) was consistent with extensive CVST involving superior sagittal, right transverse and superficial veins. CSF showed presence of IgG varicella antibodies. Protein C and S levels were normal but serum homocysteine levels were elevated. After initial treatment with intravenous Acyclovir, adequate hydration, antiepileptics and anticoagulants, he showed marked improvement in symptomatology. He went on to make a complete recovery subsequently with recanalization of sinuses seen on MRV done at fourth month follow up. He was continued on anticoagulation for 3 months with an unremarkable course of recovery.

Our case emphasizes on having a high suspicion of Varicella Zoster infection in patient who presents with CVST with an underlying ongoing Rash or a prior history of recent body rash. CVST secondary to VZV is mostly seen in patients with underlying hypercoagulable state like in our patient.