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Case reports of patients with cervical myelopathy to hypoplasia of the atlas.To report cases of cervical myelopathy due to congenital hypoplasia of the atlas and to review the literature.Six previously documented cases of congenital hypoplasia of the atlas as a cause of cervical myelopathy are reported in the literature.Three patient’s clinical record and radiologic imaging studies as well as a thorough literature search are reported. Plain radiographs, computed tomography scans, magnetic resonance images, as well as somatosensory-evoked potential changes are displayed.Cervical myelopathy developed in three patients who were found to have congenital hypoplasia of the atlas. Laminectomy of C1 provided neurologic improvement in all three patients presented.Congenital hypoplasia of the atlas is a rare cause of cervical myelopathy. This report should broaden the radiographic differential diagnosis when seeking an explanation for the signs and symptoms of cervical myelopathy.