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The Chiari I malformation has not been previously linked to sustained hypertension. Other forms of medullary compression have, however, been shown to be associated with elevated arterial pressure. This association has been demonstrated through numerous studies that have implicated compression of the rostral ventrolateral medulla, usually by vascular structures, as a factor responsible for hypertension. We present a case of a young man with a 2-year history of hypertension who was found to have a Chiari I malformation. No other cause for hypertension was found. After subsequent surgical decompression, his hypertension resolved immediately. No other therapeutic options were activated. This is the first reported case linking Chiari I malformation and sustained hypertension. This case presents further evidence linking medullary compression and elevation of hemodynamic tone.