Many authors have reported on iatrogenic vertebral artery (VA) injury, but, to our knowledge, this is the first report of a dominant VA injury with compensatory blood flow from the hypoplastic VA. A 23-year-old woman with juvenile rheumatoid arthritis and atlantoaxial subluxation sustained injury to her dominant VA after occipitocervical fusion using transarticular screws. This did not result in lethal consequences due to compensation from her hypoplastic contralateral VA. Postoperative angiography, however, illustrated occlusion of the dominant left side, while the hypoplastic VA of the right side was enlarged. The patient experienced vertigo and loss of consciousness several times during rehabilitation. At the 4-year follow-up exam, bony fusion was observed, with no neurological deficits or correction loss. She had had no episodes of unconsciousness and no recurrence of any symptoms over the previous 3 years.