In this case report we describe the presence of a dural sinus cavernosus fistula presenting with unilateral oculomotor palsy. Dural arterio-venous fistula is a rare abnormal connection between dural arteries and the venous system. Venous stasis, infarctions and intracranial haemorrhage might arise. Hence, timely diagnosis and intervention is crucial. Symptoms involve focal neurological deficits, cognitive impairment, movement disorders, seizures, symptoms of elevated intracranial or intraocular pressure and bruits or pulsatile tinnitus, which may be heard by auscultation of the skull. Invasive angiography is diagnostic.