Despite the severity of coronavirus disease 2019 (COVID-19) being more frequently related to acute respiratory distress syndrome and acute cardiac and renal injuries, thromboembolic events have been increasingly reported. We report a unique series of young patients with COVID-19 presenting with cerebral venous system thrombosis. Three patients younger than 41 years of age with confirmed Severe Acute Respiratory Syndrome coronavirus 2 (SARS-Cov-2) infection had neurologic findings related to cerebral venous thrombosis. They were admitted during the short period of 10 days between March and April 2020 and were managed in an academic institution in a large city. One patient had thrombosis in both the superficial and deep systems; another had involvement of the straight sinus, vein of Galen, and internal cerebral veins; and a third patient had thrombosis of the deep medullary veins. Two patients presented with hemorrhagic venous infarcts. The median time from COVID-19 symptoms to a thrombotic event was 7 days (range, 2-7 days). One patient was diagnosed with new-onset diabetic ketoacidosis, and another one used oral contraceptive pills. Two patients were managed with both hydroxychloroquine and azithromycin; one was treated with lopinavirritonavir. All patients had a fatal outcome. Severe and potentially fatal deep cerebral thrombosis may complicate the initial clinical presentation of COVID-19. We urge awareness of this atypical manifestation.ABBREVIATIONS: COVID-19 ¼ coronavirus disease 2019; CVT ¼ cerebral venous thrombosis; RT-PCR ¼ real-time reverse transcriptase polymerase chain reaction; SARS-CoV2 ¼ Severe Acute Respiratory Syndrome-coronavirus 2 A novel Severe Acute Respiratory Syndrome-coronavirus 2 (SARS-CoV2) was identified on January 7, 2020, by the Chinese Center for Disease Control and Prevention from a nasopharyngeal swab in a patient with atypical pneumonia in Wuhan, Hubei province, China. 1 The mortality rate of critically ill patients with COVID-19 is high. As of May 13, 2020, more than 1.4 million individuals had tested positive for the SARS-CoV2 in the United States, leading to .84,000 deaths. Acute cardiac injury, acute kidney injury, and precipitous thromboembolic events have been increasingly reported and may develop irrespective of pulmonary or respiratory symptoms at presentation. [1][2][3][4][5] Recent studies have reported the potential development of a hypercoagulable state in COVID-19. 2,3,6,7 Viral infections may promote dysfunction of endothelial cells, leading to excess thrombin