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Introduction: An estimated 150,000 cerebrovascular events are misdiagnosed in US emergency departments each year. Cerebral vein thrombosis (CVT) is a rare cerebrovascular disease caused by a thrombus formed in the cerebral sinuses or veins that can result in intracerebral hemorrhage and ischemia. Early treatment of CVT may improve patient outcomes, but diagnostic errors leading to delayed or missed CVT diagnosis have received limited attention.Hypothesis: CVT misdiagnosis rate in emergency department (ED) and outpatient settings would be high and associated with adverse outcome.Methods: We performed a retrospective cohort study at Montefiore Medical Center using electronic medical records. We identified all CVT patients hospitalized between 9/1/2005 to 9/1/2015 using validated International Classification of Diseases, Ninth Revision codes and detailed chart review. Misdiagnosis of CVT was defined as any instance when a patient sought medical attention for headache or seizure within 30 days of initial CVT diagnosis in any care setting that resulted in discharge to home. We tested whether patients’ demographic and clinical features were associated with CVT misdiagnosis. Outcomes at hospital discharge were reported. Categorical variables were compared using chi-squared and continuous variables using student t-test. P-value of <0.05 was considered statistically significance.Results: We identified 74 CVT patients. Mean age was 46 years (SD: 18) and 44 were female (67%). A total of 15 patients (20%) had a CVT misdiagnosis; 12 initially presented to the ED and 3 to outpatient clinics. Patients with CVT misdiagnosis were younger (35 vs. 48 years of age, P=0.02), and more often Spanish speaking (n=3 vs. n=0, P = 0.02). There was no difference in clinical characteristics, radiographic features, or outcomes between misdiagnosed and accurately diagnosed CVT patients.Conclusion: Misdiagnosis of CVT occurred in a fifth of CVT patients and was more common in younger and non-English speaking patients. Interventions to improve diagnostic accuracy among patients with CVT should be explored, particularly in high-risk patient subgroups.