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Syringobulbiais very rare condition defined as slit-like fluid cavity in the brain stem. Several conditions have been reported to be associated with syringobulbia including neoplasms, spinal cord traumas or lesions such as tethered cord, hind-brain herniation, infections such as meningitis and in isolation.Although post-traumatic syringomyelia has been wieldy described previously, traumatic brain injury has not been reported as the mechanism and etiology of isolated syringobulbia. We herein report a 24-year old man with previous history of severe traumatic brain injury who presented with recent onset inability to walk or coordinate movements, ataxia, dysphonia, dysarthria, bilateral third nerve palsy with fixed dilated pupils and eyes deviated outward and downward.He was further diagnosed to have isolated syringobulbia extending to upper pons and lower midbrain.Isolated post-traumatic syringobulbia is extremely rare condition presenting insidiously with cranial nerve palsies, ataxia and dysarthria.