A 40-year old conscious man developed central neurogenic hyperventilation (CNH). He had tumor dissemination to the brainstem 10 months after undergoing partial removal of a pineal glioblastoma. To the best of our knowledge, this is the first report of CNH caused by the cerebrospinal fluid dissemination of a tumor. The authors suggest that multiple lesions from an infiltrative tumor in the brainstem may give rise to CNH and further our understanding of the pathogenesis of CNH.