The oromandibular and limb hypogenesis syndrome is characterized by aglossia or hypoglossia presenting with limb anomalies. In this case report, we describe congenital hypoglossia associated with glossopalatine ankylosis and middle finger hypomelia, a type III-D malformation in the Hall Classification. The orthodontic and surgical treatment consisted of 3 maxillary expansions, mandibular surgery, and dental alignment. This extensive treatment resulted in acceptable function and esthetic appearance; however, because of lingual hypoglossia, it did not effect perfect occlusion.
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