Aim: Unilateral vertebral artery hypoplasia is considered a risk factor for posterior circulation infarction. Despite the increasing attention on unilateral vertebral artery hypoplasia, few studies have discussed bilateral vertebral artery hypoplasia, its influence on stroke, or its collateral supply from the circle of Willis. We aimed to identify its characteristics, stroke pattern, and unique ultrasonographic and brain imaging findings.Materials and Methods: Of the 1,301 consecutive in-patients diagnosed with acute ischemic stroke from January 2013 to December 2015, medical and laboratory data and stroke or transient ischemic attack history were recorded. We enrolled patients who underwent both brain magnetic resonance imaging and sonography examinations. Vertebral artery and posterior cerebral artery analyses were conducted in accordance with clinical criteria.Results: Adequate imaging data were available for 467 patients. Of these, eight patients met the criteria for bilateral vertebral artery hypoplasia. The mean age was 62.9 ± 12.1 years. There were six male (75.0%) and two female patients (25.0%). A high prevalence of hypertension (7/8, 87.5%) was noted.Sonograms displayed a very low net flow volume in the vertebral arteries, with the average net flow volume being 28.9 ± 9.7 mL/min. A high frequency (6/8; 75.0%) of the fetal variant posterior cerebral artery from the carotids was found. The infarction patterns in these patients were all bilateral, scattered, and in multiple vascular territories.Conclusion: Patients with bilateral vertebral hypoplasia displayed a unique collateral supply, special stroke pattern, and younger stroke onset. Early recognition and stroke prevention should be considered critical in clinical practice.