Patients with Bertolotti’s syndrome have characteristic lumbosacral anomalies and often have severe sciatica. We describe a patient with this syndrome in whom standard decompression of the affected nerve root failed, but endoscopic lumbosacral extraforaminal decompression relieved the symptoms. We suggest that the intractable sciatica in this syndrome could arise from impingement of the nerve root extraforaminally by compression caused by the enlarged transverse process.