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Leprosy is a chronic infectious disease caused by the Mycobacterium leprae that leads to leprotic neuropathy involving the peripheral nerve and several characteristic skin lesions. Skeletal involvement can occur in peripheral joints, such as the wrist and the ankle. However, there is no report of an axial leprotic lesion involving the spine or paraspinal soft tissue. The authors report the first case of a leprotic cervical lesion involving the axial skeletal system. A 48-year-old male presented with neck pain and severe pain in the right suprascapular area and left arm. Preoperative MRI of the cervical spine revealed signal changes in the prevertebral soft tissue at the level of the C3, 4, 5 vertebral bodies. There were a lower signal intensity on T1-weighted image and high signal intensity on T2WI of the bone marrow at the level of the C5 and C6 vertebral bodies, and a C5/6 segmental ossification of the posterior longitudinal ligament. There were herniated cervical disc on the left C5/6 with C6 root and the right side of C6/7 with a C7 root compression. He was previously diagnosed with leprosy when he was 14 years old and received treatment intermittently over the course of 7 years. But patient did not disclose his past history. Surgical intervention was conducted using an anterior cervical approach. An incision was made in the anterior longitudinal ligament at C5/6, and a pinkish gray friable gelatinous material was observed on the C5/6 disc and on the anterior lower one-third surface of the C5 vertebral body. Specimens were obtained and subjected to pathological evaluation and microbiological culture. After C5/6 and C6/7 discectomies, nerve root decompression and autologous iliac bone grafting were performed at the C5/6 and C6/7 levels. The C5-6-7 vertebrae were fixed with an Atlantis cervical locking plate and a screw system. The pathological report indicated chronic inflammation with heavy plasma cell infiltration on the specimen. We sent the specimens to the Institute of Hansen's Disease, and polymerase chain reaction for leprosy tested positive. After surgery, his pain disappeared and he was given a prescription for antileprotic drugs. The authors describe the first case of leprotic cervical spondylodiscitis that was operatively treated in a 48-year-old patient with known leprosy history since his 14 years old. [ABSTRACT FROM AUTHOR]