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Dyke-Davidoff Masson Syndrome (DDMS) is a rare syndrome characterized by seizures, facial asymmetry, and contralateral hemiplegia or hemiparesis. The typical radiological features of DDMS include cerebral hemiatrophy with ipsilateral hypertrophy of the skull and sinuses. It is usually diagnosed in childhood. Presently described are 2 rare cases of DDMS that were diagnosed in adults based on magnetic resonance imaging of the brain.