Pudendal nerve entrapment syndrome is an unusual cause of chronic pelvic pain. We experienced a case of pudendal neuralgia associated with a ganglion cyst. A 60-year-old male patient with a tingling sensation and burning pain in the right buttock and perineal area visited our outpatient rehabilitation center. Pelvis magnetic resonance imaging showed the presence of multiple ganglion cysts around the right ischial spine and sacrospinous ligament, and the pudendal nerve and vessel bundle were located between the ischial spine and ganglion cyst at the entrance of Alcock's canal. We aspirated the lesions under ultrasound guidance, and consequently his symptoms subsided during a 6-month follow-up. This is the first report of pudendal neuralgia caused by compression from a ganglion cyst around the sacrospinous ligament.