Congenital maxillomandibular fusion is a rare disorder of the craniofacial region. Maintenance of the airway and difficulty in feeding are the main concerns for such patients. We performed a PubMed search to identify reported cases of syngnathia. With reference to the site of fusion and corresponding anomalies, the available studies were reviewed. The maxillomandibular fusion can be anterior fusion, unilateral or bilateral fusion, and complete fusion. The condition can be an isolated problem or associated with a syndrome. The condition requires immediate attention because it can be fatal. A case of isolated unilateral syngnathia, with the detailed review of the published data, is reported.
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