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Behçet's disease (BD) is a systemic vasculitis characterized by recurrent oral and genital ulcers, with a frequent ocular and skin involvement. Neurological involvement in BD has been reported in 4–49% of cases and determines functional and vital prognosis of the disease. Based on clinical and imaging evidence, two major forms can be identified: a parenchymal or an extra parenchymal involvement essentially represented by cerebral vein thrombosis. Awareness disorders are rarely reported as tell-tale signs of neurological involvement in BD. We hereby report the peculiar case of a young patient, 39 years old, admitted to the emergency department for an acute onset of altered mental status in a non-traumatic setting where investigations led to the diagnosis of neurobehçet disease. Patient was placed on high doses corticosteroids with unfavorable outcome; patient passed away after 01 week.