Rationale: Cerebrovascular accidents (CVAs) after percutaneous coronary intervention (PCI), although rare, are associated with high in-hospital morbidity and mortality rates. Cerebral venous thrombosis (CVT) is an uncommon cause of CVAs compared with arterial disease but is associated with favorable outcomes in most cases. We present a rare case of CVT following a simple PCI procedure with stent implantation, which has not been previously reported in the literature. Patient concerns: A 78-year-old woman with hypertension, hyperlipidemia, and coronary artery disease received simple PCI with stent implantation. After PCI, she developed a throbbing headache with nausea and vomiting, with her blood pressure increasing to 190/100 mmHg. Drowsiness, disorientation, and neck stiffness were noted. Neurological complication due to the PCI procedure was highly suspected. Diagnosis: Noncontrast brain computed tomography was performed along with emergency neurological consultation, and the patient was diagnosed as having acute CVT. Interventions: The patient was treated with anti-intracranial pressure therapy and anticoagulation therapy through low-molecular-weight heparin and was subsequently treated with warfarin. Outcomes: After treatment, the patient's symptoms and signs gradually subsided, and her clinical condition improved. She was discharged with full recovery thereafter. Lessons: A case of acute CVT, a rare, and atypical manifestation of venous thromboembolism and CVA, complicated simple PCI with stent implantation. During PCI, identifying patients with a high risk of a CVA is critical, and special care should be taken to prevent this devastating complication.