We present a case of childhood arterial ischemic stroke associated with proven adenoviral upper respiratory tract infection in a previously healthy 4-year-old boy. Adenoviral meningitis and encephalitis have been reported repeatedly, thus confirming the neuroinvasive capability of these viruses. However, an association between adenoviral infection and arterial ischemic stroke has not been described thus far. HIV and varicella zoster virus are the only microorganisms that have been consistently associated with arterial ischemic stroke in the absence of acute central nervous system infection. In HIV-infected individuals ischemic stroke can be caused by vasculitis and hypercoagulability. Granulomatous arteritis of the vessel wall causes post-varicella cerebral infarction and ischemic stroke after herpes zoster ophthalmicus. We suggest that in our patient a post-varicella cerebral infarction-like mechanism of adenoviral spread to the affected artery wall occurred through the ophthalmic branch of the trigeminal nerve. Adenoviruses are neuroinvasive and inflamed conjunctiva might have permitted introduction of the virus into ophthalmic nerve tissue. In consequence, the stenotic lesion of the artery might have been induced by the presence of adenovirus and the subsequent inflammatory reaction. We recommend a prompt quest for adenoviral infection in all previously healthy children with fever and clinical presentation compatible with ischemic stroke, because timely diagnosis and treatment could improve the outcome and hasten neurological recovery.