Carotid cavernous fistula (CCF) is an abnormal communication between cavernous sinus and carotid arterial system. Diagnosis depends on clinical manifestations and MRI findings and angiography. Clinical presentation of CCFs is characterized with chemosis, orbital bruit and pulsatile proptosis. Cranial nerve dysfunction and orbital pain might accompany these symptoms. Although spontaneous remission might occur, sometimes CCF might develop life threatening complications. Here in this case we presented a patient with CCF who afterwards developed bilateral thalamic infarct. Cerebral ischemia in CCF is a rare complication and CCF together with bilateral thalamic infarct has not been reported before.