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Heterotopic brain tisue is defined as displaced neuroglial tissue that has no connection with the central nervous system. Mostly non-midline structures such as middle ear, mastoid, and orbit. Comonly acepted theory is that heterotopic neural tissue is a variant of encephalocele in which the central nervous system conection has been lost or became vestigial. We experienced a case of heterotopic neuroglial tisue found in a patient who had conductive hearing loss in unilateral ear, and report it with a review of the related literature. (Korean J Otolaryngol 2003 ;46 :696-8)