Air embolism is a preventable, often undiagnosed but potentially treatable cause of ischemic stroke with a high morbidity and mortality. It is usually iatrogenic ocurring especially in patients in ICU setting. We describe the case and neuroimaging of a patient with ischaemic stroke due to air embolism during manipulation of central venous line. We also review the literature with respect to aetiology, incidence pathophysiology, diagnosis, and treatment options for venous and air embolism. Cerebral air embolism should be considered in patients with sudden neurological deterioration after central venous or arterial manipulations or certain neurological procedures. Prevention, as well as early diagnosis and management, may reduce morbidity and mortality.