Gastrointestinal cyst of the tongue: A possible duplication cyst of foregut origin

Abstract

Gorlin and Sedano,’ in their 1993 extensive review of hamartomatous and choristomatous lesions of the tongue, pointed out that aberrant gastric mucosa may be seen in an inordinate number of gastrointestinal locations: epiglottis, esophagus, deep within the stomach wall, the small intestine, Meckel’s diverticulum, the biliary tract and gallbladder, pancreas, umbilicus, appendix. rectum, vitelline duct, thoracic duct, and even the spinal canal (neuroenteric cyst). Incorporation of gastric mucosa in the esophageal wall is found at autopsy in almost 8% of infants and children. Approximately 50% of these patients have the heterotopic gastric mucosa in the upper third of the esophagus. Barrett’s esophagus is an acquired condition, characterized by replacement of the squamous epithelium with columnar-lined mucosa and associated with chronic gastroesophageal reflux. Involvement ranges from the lower one third to the entire esophagus, and the condition has a malignant potential. In addition, there are at least 65 descriptions of cysts lined by gastric, esophageal, or intestinal mucosa in the tongue or oral area. These were first reviewed by Gorlin and Jirasek’ in 1970. The reader may find the earlier cases summarized in that article, because they are not cited in the following review. There is at least a 3M:lF sex predilection for such cysts. The choristomatic cyst may be entirely enclosed within the body of the tongue,2-31 oral floor.3,29.32-39 hypopharynx,‘4*40-43 anterior neck.44 larynx,3” or submandibular gland,‘4,45-48 or it may communicate with the surface. A similar lesion has been reported in the lip.49 The cystic wall may be composed partly of stratified squamous and partly (or rarely totally) of gastric mucosa of the type seen in the body and fundus of the stomach or the intestinal mucosa. In one patient, the cyst was stated to contain pigmented ciliated epithelium, although this was not illustrated.50 However, respiratory epithelium has been shown.z1.51 52 Glands usually are present. Schultz” described these as Brunner’s glands, although marked resemblance to gastric glands can be noted in the photomicrographs in his article. Both parietal and chief cells may be found. Associated pancreatic tissue was noted by Douniau et a1.45 A few examples of both oral dermoid cyst and choristomatous cyst occurring together have been noted.53.54 A muscularis mucosa was noted around the cyst in several cases. In a case reported by Gorlin and Jirasek,’ a tube surrounded by muscle connected the cyst to the oral mucosa. Sebaceous glands emptied into the tube. Several authors13,55-57 have reported a lingual tube surrounded by sebaceous glands without gastric or intestinal epithelium being present. We suspect that the sebaceous glands associated with papillary atrophy on the dorsum of the tongue of a 28-year-old-man was a minimal expression of this disorder.58 The origin of the heterotopic gastric or intestinal epithelium is not known. The tongue arises from the floor of the pharynx in the region of the first three branchial arches. In the 3to 4-mm embryo, the undifferentiated primitive stomach lies adjacent to the anlage of the tongue. The cyst is probably derived from misplacement of embryonal rests and represents their pleuripotentiality. We do not believe that the cyst differs, in essence, from the few examples of lingual cyst lined entirely by respiratory epithelium described by Gorlin and Sedano.’