STUDY DESIGN: Case-control study. OBJECTIVES: Investigating the association between neurodegeneration within rostral spinal cord and brain gray matter volume (GMV) and assessing the relationship between remote neurodegenerative changes and clinical outcomes at the early phase of Cervical Spondylotic Myelopathy (CSM). SETTING: University/hospital. METHODS: Using Spinal Cord Toolbox, spinal cord morphometrics (cross-sectional area [CSA], gray matter area [GMA], white matter area [WMA]) of 40 patients with CSM and 28 healthy controls (HCs) were computed and compared using two-sample t test. Brain GMV of the two groups was analyzed using voxel-based morphometry approach. Pearson's correlation between spinal cord morphometrics and altered brain GMV and Spearman's relationship between remote neurodegenerations and clinical outcomes were conducted in CSM group. RESULTS: Compared to HCs, CSA and WMA at C2/3 and GMV in right postcentral gyrus (PoCG.R) and left supplementary motor area (SMA.L) were significantly decreased in patients with CSM. CSA and WMA at C2/3 were associated with GMV in SMA.L and MCG.R in patients with CSM. CSA at C2/3 and GMV in PoCG.R were related to modified Japanese Orthopedic Association score in patients with CSM. CONCLUSIONS: The associations between CSA and WMA at C2/3 and GMV in SMA.L and MCG.R suggest a concordant change pattern and adaptive mechanisms for neuronal plasticity underlying remote neurodegeneration in early CSM. The atrophy of CSA at C2/3 and GMV loss in PoCG.R can serve as potential neuroimaging biomarkers of early structural changes within spinal cord and brain preceding marked clinical disabilities in patients with CSM.