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Vertebrobasilar insufficiency (VBI) provoked by physiological head rotation is known as rotational vertebral artery syndrome (RVAS) or Bow Hunter syndrome. RVAS most often occurs at C1-2 level with head rotation and presents with symptoms of VBI. Several previously published studies have reported RVAS at subaxial sites (V2 segment), however, tumor-induced RVAS has never been reported. The authors report the first case of RVAS at V2 segment due to compression from a spinal tumor. A 71-year-old man presented with symptoms of dizziness provoked by head rotation or neck extension. computed tomography (CT) angiography and dynamic cerebral angiography revealed circumferential stenosis with neutral neck position and complete occlusion of the left dominant vertebral artery (VA) at C5 level with his neck extended or rotated to the left. Complete neurological recovery was achieved after removal of a spinal osteochondroma and surgical decompression of the left VA via an anterior approach. Spinal tumors should be included in the differential diagnosis in cases of RVAS. Spinal degenerations or sarcomatous transformation of the tumor could lead to clinical manifestations of RVAS in cases with spinal osteochondroma. Complete removal of the tumor with or without spinal fusion would be the treatment of choice, in addition to medical treatment in the cases of acute stroke., Competing Interests: Conflicts of Interest Disclosure The authors have no conflicts of interests.