This study aimed to compare morphologic changes of deep paraspinal muscles at C4-7 in patients with C5-6 single-level cervical spondylotic myelopathy (CSM), and to evaluate the relationship between morphologic changes and the level of spinal cord compression. The study included 15 patients with C5-6 single-level CSM and 15 age- and sex-matched healthy subjects. Cross-sectional area (CSA) and functional CSA of the bilateral longus capitis (LCap), longus colli (LC), multifidus (MF), semispinalis cervicis (SSC), semispinalis capitis, splenius capitis, and splenius cervicis were measured on preoperative magnetic resonance images at C4-7, and calculated as ratios with respect to the corresponding vertebral body CSA. The mean maximum spinal cord compression was 22.30% in the CSM group. At the cranial level (C4-5), the CSM group had more fat infiltration in the MF and SSC (P < 0.05). At the spinal cord compression segment and caudal adjacent segment (C5-6 and C6-7), the degree of fat infiltration of all paravertebral muscles was aggravated, accompanied by atrophy of LCap, LC, and MF (P < 0.05). Compared between different levels, fat infiltration in the MF at C5-6 was greater than adjacent levels. In patients with C5-6 single-level CSM, fat infiltration and atrophy of deep paraspinal muscles, especially the Lcap, LC, and MF, mainly occurred in the level of spinal cord compression and caudal adjacent level. In the cranial adjacent segment, the degree of MF and SSC fat infiltration in patients with CSM was also aggravated. This may suggest that multiple mechanisms are involved in paraspinal muscles degeneration in CSM. Copyright © 2019 Elsevier Inc. All rights reserved.