We herein report a young patient with cerebral venous thrombosis (CVT) with clinical and neuroradiological findings of the left maxillary and anterior ethmoid sinusitis. Serial brain MRIs showed cerebral venous infarct and thrombosis in the superior sagittal sinus (SSS). MR angiography demonstrated nonvisualization of SSS and bilateral transverse sinus. According to our knowledge, CVT associated with maxillary and ethmoid sinusitis has been reported very rarely. High index of suspicion and neuroimaging studies, especially brain MRI, and conventional or MR angiography are very important for the early diagnosis of CVT.