Varicella zoster virus (VZV) has been known to cause cerebral arterial vasculopathy as well as an acquired antibody-mediated coagulopathy associated with purpurafulminans and generalized thrombo-embolism. There is no published report on cerebral venous sinus thrombosis (CVST) associated with primary VZV infection. We hereby report two cases to highlight an unusual presentation of VZV infection i.e. CVST with primary varicella infection. One patient had extensive CVST with co-existent middle cerebral artery involvement as well. .Primary VZV infection can be associated with thrombosis of cerebral arteries and venous sinuses