Study design: Case report and review of the literature. Objective: To report a patient presenting with Brown-Sequard syndrome due to cervical intradural disc herniation after spinal manipulation therapy. Summary of background data: Spinal manipulation therapy (SMT) is often used by people with neck pain or discomfort as an alternative therapy due to its claimed less invasiveness and comparable efficacy. However, excessive manipulations are reported to cause rare but serious complications such as tetraplegia, vertebral artery dissection, epidural hematoma, and phrenic nerve injury. Methods: Clinical history, physical examination, and radiographic findings of the patient were described. Anterior cervical discectomy at the C3/C4 level and interbody fusion with a Caspar plate-screw system for fixation, were performed. Results: A favorable surgical outcome was obtained. The Brown-Sequard syndrome improved and the patient regained full muscle power at a 3-months follow-up. Conclusion: Cervical intradural disc herniation after SMT is rare and most often cause Brown-Sequard syndrome. Definite diagnosis and prompt surgery usually achieves a satisfactory outcome. Anterior discectomy with interbody fusion is recommended. The OPLL associated with degenerative disc reminds us of the increased risk of intradural disc herniation. Those high-risk groups should be more cautious with spinal manipulation therapy due to its serious sequelae.