Enteric Duplication Cyst Located at the Posterior Tongue: A Rare Case Report and Review of the Literature

Abstract

The lingual localization of an enteric duplication is extremely rare but may present with respiratory and feeding problems that require emergency intervention. A 7-month-old boy was brought to our clinic with feeding difficulties and tongue swelling. Physical examination showed a cystic lesion located near the left side of the tongue base that caused tongue protrusion to the contralateral side. During surgery, a 3-cm diameter opaque thick-walled cyst was found to be very closely adherent to the base of tongue, which was excised in its entirety. Following surgery, the patient fed during the early postoperative period and no complications were observed other than hypersalivation. On histological examination, a cystic lesion lined with intestinal mucosa and goblet cells was found. We present the rare case of a duplication cyst of the posterior tongue, with a literature review.

An adult case of a retroperitoneal isolated enteric duplication cyst with the imaging changes over time

“…Such non-communicating, isolated EDCs have a wall with gastrointestinal epithelium and a well-developed coating of smooth muscle, similar to that seen in regular EDC, but without an anatomical association with the alimentary tract. This type of tumor has been reported in various locations, including the tongue [ 7 ], pleural space [ 8 ], liver [ 9 ], pancreas [ 10 ], biliary tree [ 11 ] and retroperitoneum (as is described in the presented case). IEDC in adults is very rare and the consensus on the diagnosis, including the characteristic feature of malignancy, has not been established.…”

A case report of oral heterotopic gastrointestinal cysts (HGIC) and review of the literature

“…9 Savran, describes these HGIC (enterocysts, enteric duplication cysts) as a choristoma which has arisen from islands of endodermal cells that prevent the fusion of the lateral lingual protuberances and the tuberculum impar during the third week of embryonic development. 10 Kinoshita 11 reports that analysis from the mucin derived from these cysts is different in composition from normal gastrointestinal mucosa. This suggests that the lesion originates from undifferentiated endoderm subjected to inductive influences, which results in varying amounts of differentiation.…”

“…9 The development of vestigial structures such as the thyroglossal duct which has endodermal potential have also been proposed. 10 The last three theories are not widely accepted.…”

A Rare Case of Ectopic Colonic Mucosa Presenting With Airway Compromise in a Neonate

“…Enteric duplication cysts can be found anywhere along the alimentary canal and are benign congenital heterotopic tissue with two layers of smooth muscle, gastrointestinal, or respiratory epithelium [1,2]. These cysts are rare entities and have a reported incidence of one per 4,500 births, with a slight male predominance [3].…”

“…These cysts are rare entities and have a reported incidence of one per 4,500 births, with a slight male predominance [3]. Gastrointestinal tract duplication cysts usually present with gastric type epithelium and can occur anywhere from the oral cavity to the rectum, but they are most commonly found in the ileum, followed by the esophagus, colon, jejunum, stomach, and duodenum [1,2,[4][5][6]. Their presence within the oral cavity is rare -only 0.3% of cases are seen in the tongue [4,7].…”