Thrombosis-thrombocytopenia syndrome and cerebral venous sinus thrombosis have been rarely reported in patients who have received severe acute respiratory syndrome coronavirus 2 adenoviral vector vaccines. Awareness of this potential adverse effect, recognizing early clinical symptoms and subtle signs of cerebral venous sinus thrombosis on head computed tomography and brain magnetic resonance imaging, appropriate vascular imaging, and unique treatment for this condition is critical. This is a report of a case of vaccine-induced thrombotic thrombocytopenia and associated cerebral venous sinus thrombosis with emphasis on imaging and clinical course.