Background:Spinal epidural abscess resulting from piriformis pyomyositis is extremely rare. Such condition can result in serious morbidity and mortality if not addressed in a timely manner.Case Description:The authors describe the case of a 19-year-old male presenting with a 2-week history of fever, low back pain, and nuchal rigidity. When found to have radiographic evidence of a right piriformis pyomyositis, he was transferred to our institution for further evaluation. Because he demonstrated rapid deterioration, cervical, thoracic, and lumbar magnetic resonance imaging scans were emergently performed. They revealed an extensive posterior spinal epidural abscess causing symptomatic spinal cord compression extending from C2 to the sacrum. He underwent emergent decompression and abscess evacuation through a dorsal midline approach. Postoperatively, he markedly improved. Upon discharge, the patient regained 5/5 strength in both upper and lower extremities. Cultures from the epidural abscess grew methicillin-sensitive Staphylococcus aureus warranting a 6-week course of intravenous nafcillin.Conclusion:A 19-year-old male presented with a holospinal epidural abscess (C2 to sacrum) originating from piriformis pyomyositis. The multilevel cord abscess was emergently decompressed, leading to a marked restoration of neurological function.