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Craniofacial microsomia is a unique clinical presentation of "1 st and 2 nd arch syndrome" with asymmetrical craniofacial development along with conductive hearing loss. It involves the underdevelopment of the structures of the first and second pharyngeal arches: Maxilla, mandible, external and middle ear, facial and trigeminal nerves, muscles of mastication and overlying soft tissue. In this article, we report a case of craniofacial microsomia diagnosed based on clinical and cone beam computed tomography (CBCT) findings. CBCT scan revealed incompletely formed inner ear structures in association with craniofacial microsomia, rarely reported in the literature.