Congenital maxillomandibular fusion (syngnathia) is rare. The first case was reported in 1936. By 2004, only 25 cases of syngnathia had been reported including 6 cases with fusion of the ascending rami of the mandible with the maxilla and zygomatic complex. This report concerns a 6-month-old girl with a type of congenital fusion of the ascending rami of the mandible with the upper alveolar arch and zygomatic complex that leads to restriction of mouth opening. The clinical features of this anomaly and a brief review of literature are presented.
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