Congenital cholesteatoma within the tympanic membrane is an uncommon entity, with only few cases being documented. The aetiopathogenesis of this lesion is still unknown; however, when cholesteatoma develops in subjects without any history of previous ear inflammation, as in the case we report here, an embryologic origin is deeply suspected. An acquired origin is hypothesized in patients with a previous history of an inflammatory process of the external or middle ear because of the proliferation

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... of the basal cell layer of the tympanic membrane epithelium. We report a rare case of congenital cholesteatoma of the tympanic membrane in an adult patient and review the literature KEYWORDS: Congenital intratympanic cholesteatoma, intratympanic cholesteatoma, congenital cholesteatoma of the tympanic membrane, cholesteatoma of the tympanic membrane Design -B.P., M.M.; Supervision -F.C.; Resources -M.M.; Materials B.P.; Data Collection and/or Processing -B.P., M.M.; Analysis and/or Interpretation -M.M., B.P., F.C.; Literature Search -B.P., M.M.; Writing Manuscript -M.M.; Critical Review -F.C.