We describe a man aged 26 years who presented with a neurological syndrome, which was found on lumbar radioculopathy to be due to a ganglion cyst originating from the posterior longitudinal ligament. Based on MRI findings, cystic lesion was suspected, a round lesion at L4 level with no connection to the adjacent facet or to the dura matter. During surgery, a liquid-containing cystic lesion was found to originate from the posterior longitudinal ligament at L4 level. The resected cyst was diagnosed histologically as a ganglion cyst. A complete cure was established after surgery and no recurrence was noted at a follow-up 1.7 years postoperatively. A ganglion cyst of the posterior longitudinal ligament should be considered in the differential diagnosis of a cyst in the lumbar region causing neurological complications.