CASE REPORT
Foregut duplication cyst arising in the floor of mouth

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Abstract

Enteric duplications are uncommon. A rare foregut duplication cyst arising in the floor of mouth is reported. Diagnosis of cystic lesion within the oral cavity was made in utero. Enteric duplications have not yet been attributed to a single embryogenesis but are likely to represent an error in migration of normal cells that rest in an abnormal position. Immediately following delivery of the neonate via an ex-utero intrapartum treatment (EXIT) procedure, the cyst was marsupialised but required definitive surgical excision at 5 weeks of age.

Introduction

Enteric duplication cysts are an uncommon congenital anomaly. They are described arising anywhere along the alimentary tract, from oral cavity to rectum. Midgut duplications are most common [1]. Foregut duplication cysts account for about one third of duplications and arise from remnants of the stomach and first and second parts of the duodenum. They are classified as choristomas-containing tissues ectopic to their site of origin. Up to 2003 only 21 oral cavity enteric duplications have been reported in the literature [2], [3], [4], which constitutes 0.3% of all enteric duplications [5]. Oral cavity cysts, lined with gastric or intestinal mucosa were first reported in 1927 by Toyama [6] and Schultz [7].

This case report describes the management of a foregut duplication cyst, highlighting important clinical issues including pre-natal diagnosis, perinatal management of an infant with a potentially obstructing airway lesion, accuracy of imaging modalities and definitive surgical cure.

Section snippets

Case report

A 31-year-old Caucasian woman with four previous live births, and her Australian Aboriginal husband, presented to the John Hunter Children’s Hospital, Newcastle Australia after routine ultrasonography showed an anomaly of the fetal mouth. High-resolution ultrasonography confirmed a cystic lesion in the foetal mouth. In utero magnetic resonance imaging (MRI) of the fetal head and neck at 20 weeks gestation showed a cystic mass filling the oral cavity and displacing the tongue to the roof of the

Discussion

During the first 4 weeks of gestation the process of cephalo-caudal folding of developing embryo results in part of the endoderm-lined secondary yolk sac being incorporated to form the primitive gut, ending cephalically as a blind sac dorsal to the pericardial sac, and separated from the ectoderm-lined primitive mouth cavity or stomodeum by the oro/bucco-pharyngeal membrane. This membrane ruptures by 4 weeks so the primitive oral cavity becomes continuous with the primitive foregut. By the end

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