In a review of the vertebral angiograms of 300 patients free from disease at the craniovertebral junction, we found atlantoaxial arterial anomalies in 2,3%. These were: 2 cases in which the vertebral artery ran in the spinal canal below C1, 3 cases of duplication of the vertebral artery above and below C1, and 2 cases of origin of the posterior inferior cerebellar artery at C2. Although these arteries ran in the spinal canal between C1 and C2, they never encroached upon the posterior third of the canal. From the survey of another 21 patients having bony abnormalities at the craniovertebral junction, the first type of arterial anomaly described above was seen in 4 patients and associated with failure of segmentation of the embryonic sclerotome such as occipitalization of the atlas or Klippel-Feil syndrome. It is possible to relate the development of these anomalous vessels to malarrangement of the embryonic segmental arteries. Our results indicate that one must be cautious with lateral C1/2 puncture or surgical exposure of the region.