Context: Osteochondroma is the most common benign tumor of the bone, but spinal osteochondroma is rare. We report a case of cervical osteochondroma in multiple exostoses arising from the lamina of the C2 vertebra, presenting with features of compressive myelopathy in a 22-year-old male. Total resection of the tumor and atlantoaxial fixation and fusion after reconstruction of the C1 posterior arch were performed. Findings: The patient recovered significantly. He was asymptomatic and no sign of recurrence was observed after one-year follow-up. Conclusions: Osteochondroma should be considered as a rare cause of spinal cord compression. Entire removal of the tumor will result in complete decompression and can reduce the risk of recurrence. We provide a new approach to reconstruct after resection. © 2018, © The Academy of Spinal Cord Injury Professionals, Inc. 2018.