INTRODUCTION: Direct intramedullary infections are considered very rare. Only few reports of Staphylococcus aureus myelitis have been published. CASE PRESENTATION: Our patient, a 79-year-old male, presented with a 2-day history of high-grade fever and high inflammatory markers and progressively developed tetraplegia during hospitalization. Lumbar puncture revealed cerebrospinal fluid pleocytosis and a spinal cord MRI revealed transverse myelitis at the level of C3-C5 and possible osteomyelitis of C5-T1. Two blood cultures were positive for methicillin-sensitive S. aureus. Despite control of the infection, there was no neurologic improvement. DISCUSSION: The morbidity of infectious myelitis can be severe. Considering the rarity of S. aureus myelitis, experience gained from case reports is important. A brief review of the available literature is provided.