Congenital syngnathia: review of clinical profile and proposal of a new classification-based management protocol

Abstract

Syngnathia is a rare congenital anomaly that presents as fusion of the jaws. The aims of this study were to review the clinical profile of reported cases of syngnathia available in the literature with a view to suggesting a classification that may simplify the understanding of syngnathia and to propose an appropriate management protocol. A PubMed database search of articles published in English was conducted. Selected articles were analyzed according to year of publication, type of article, number of cases per article, patient biodata, description of syngnathia, and associated anomalies. Data were analyzed using IBM SPSS version 19; the level of statistical significance was set at P≤ 0.05. Cases were categorized according to a proposed classification, and a review of the management of congenital syngnathia from 153 previously reported cases (in 110 articles) was done to propose a treatment protocol based on the proposed classification model. The findings are presented under three headings: pre-surgical considerations, surgical (intraoperative) considerations, and post-surgical considerations. The proposed classification categorizes syngnathia into soft tissue union (type 1) and hard tissue union (type 2). These two types were found to have similar frequencies in the literature, while their management differed in terms of mode of anaesthesia and surgical approach.

Keywords: classification; management protocol; maxillomandibular union; synechiae; syngnathia.