Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 5 S1 2021 03 07 19 22 Athena Sharif Razavi Clinical Research Development Unit of Bou-Ali Sina Hospital, Mazandaran University of Medical Science, Sari, Iran. Narges Karimi Associate Professor of Neurology, School of Medicine, Immunogenetics Research Center, Toxoplasmosis Research Center, Clinical Research Development Unit of Bou Ali Sina Hospital, Mazandaran University of Medical Sciences, Sari, Iran. 2020 09 01 2020 10 18 Background: The most common symptom of the novel Coronavirus Disease 2019 (COVID-19) infection is fever and dyspnea that leads to hypoxia in severe cases. Some COVID-19 patients experience neurological symptoms, including ischemic stroke and intracerebral hemorrhage. Sickle Cell Disease (SCD) is a hypercoagulable state, however, it has not been approved as a significant cause of Cerebral Venous Thrombosis (CVT).  Case presentation: In this case report, we described CVT in an SCD patient who had COVID-19, as well. We reported a 32-year-old man with a history of sickle cell anemia presented with left hemiparesis, headache, and seizure. After evaluation of the patient, CVT accompanied by COVID-19 infection was diagnosed. He was treated with intravenous unsaturated heparin, antiepileptic drugs, and antiviral agents with a favorable outcome. Based on our knowledge, this is the first case study to describe an association between CVT and COVID-19 infection in a patient with SCD. Conclusion: During the recent pandemic, vaso-occlusive attacks in SCD patients can be evaluated for COVID-19 as an etiological factor. https://crcp.tums.ac.ir/index.php/crcp/article/view/404 https://crcp.tums.ac.ir/index.php/crcp/article/download/404/385