The authors report the rare case of a 48-year-old woman with a cervical subpial lipoma unassociated with spinal dysraphism. Her symptoms were progressive weakness and numbness in the four extremities and the neurological symptom of mild cervical myelopathy. Plain radiographs of the cervical spine showed diffuse widening of the spinal canal, which was occupied by a large low-density mass revealed by axial CT scan. MR imaging identified the mass as being dorsolateral to the cord in the intradural region. It was hyperintense on both T1- and T2-weighted images. Axial images with fat suppression sequence clearly showed dorsal nerve roots traveling through the lesions, but the interface between the spinal cord and lesion was not well demarcated. A cervical laminectomy between C3-6 revealed a yellowish subpial mass after the dura was opened. The mass was only partially resected and intraoperative SEP monitoring data remained normal including N20 latency. Histological examination found mature adipose tissue covered by thickened pia mater, which was compatible with a diagnosis of lipoma. After surgery, no complications occurred and the patient's myelopathy subsequently improved. Subpial spinal lipomas unassociated with dysraphism are uncommon and they are reported in only 1% of all spinal cord tumors. They are thought to arise from premature dysjunction of the cutaneous ectoderm during neural tube formation. Without any invasive procedure, MR imaging is indispensable to show their longitudinal dimension as well as their infiltrative extension into the spinal cord. The main purpose of surgery is to decompress the lipoma from the adjacent neural structures, because it is benign in nature and there is no cleavage plane identifiable between the lipoma and the spinal cord.