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BACKGROUND Primary melanomas affecting the central nervous system are very rare, and melanomas originating in the spinal canal or origin of the spinal nerve root are even rarer. As a consequence, not much is known about this. CASE SUMMARY Here we report a case of primary malignant melanoma originating in the cervical spinal cord nerve root. A 64-year-old woman presented with symptoms of numbness in the right side of the neck, pain, and hypoesthesia in the right upper limb which persisted for 1 year. Neurological examination showed that the superficial sensation in the right upper limb had decreased with muscle strength of grade 4. Magnetic resonance imaging examination revealed a mass (approximately 2.5 cm × 1.4 cm × 1 cm) in the right side of the spinal canal in the C-2 plane. Based on findings obtained during operation, perioperative examination, pathological diagnosis, and the diagnostic criteria of primary central melanoma proposed by Hayward, the mass was confirmed to be a melanoma of intraspinal nerve root origin. CONCLUSION This is the first case of primary malignant melanoma originating from cervical spinal cord nerve roots and spread along the inside and outside of the spinal canal. The clinical relevance of this case is discussed to provide new insights into the differential diagnosis of intraspinal tumours. Further studies are needed to better understand the mechanisms driving the growth pattern and development of this type of tumour.