We reported a 48-year-old man with Behcet disease, who presented with right hemiparesis. His first brain MRI showed multiple enhanced lesions. During the recovery, he had an episode of left 6th nerve palsy without new lesions in a follow-up MRI. Third episode was cervical myelitis, resulting in respiratory difficulty and quadriplegia without any reflexes. The myelitis was not responsive to immunotherapy. He died of respiratory failure complicated with pneumonia. This is a rare case of full-blown neuro-Behcet disease.