Isolated cortical vein thrombosis (ICVT) is a rare entity of cerebral venous thrombosis (CVT) that represents a third leading cause of death during pregnancy and puerperium. IVCT manifests with wide spectrum of symptoms including headache, seizure, focal neurological deficits and changed mental status, makes the diagnosis challenging. Pregnancy, puerperium, lumbar puncture, trauma and infection, genetic predisposing and acquired prothrombotic causes, all are well recognized risk factors for ICVT. Although no established universal consensus for management, IVCT generally treated by anticoagulant. In this paper, a case of IVCT reported in 25 years old postpartum mother, delivered by cesarean using epidural anesthesia. The case presents by persisting non-orthostatic headache in first few days post-delivery that treated as post dural puncture headache but not improved. On day 7 post-delivery developed multiple episodes of focal convulsion progressed to generalized tonic-clonic convulsion associated by left hemi-paresis. MRI gradient echo sequences showed liner hypo-signal lesion with configuration of cortical vein over right high parietal area. The MRV showed multiple small filling defects along the right cortical vein in favor IVCT. The patient treated successfully by enoxaparin (according her body weight) followed by warfarin for 3 months with complete resolution of the neurological deficit.