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Sachs S, Oral Surg Oral Med Oral Pathol Oral Radiol Endod. (1999)
Heterotopic oral gastrointestinal cyst: report of 2 cases and review of the literature., 88
Lobe TE, Am J Perinatol. (1997)
Perinatal management of enteric duplication cysts of the tongue., 14
Reychler H, Int J Oral Maxillofac Surg. (1991)
Enteric duplication in the oral cavity., 20
Gunhan O, J Oral Maxillofac Surg. (2001)
Gastric mucosal choristoma of the tongue and floor of the mouth., 59
Gloster ES, Arch Pathol Lab Med. (1989)
Congenital oral cyst with heterotopic gastrointestinal and respiratory mucosa., 113
Yonehara Y, Ann Plast Surg. (1990)
Heterotopic gastrointestinal cyst of the oral cavity., 25
Smith AJ, Oral Surg Oral Med Oral Pathol. (1988)
Heterotopic gastrointestinal cyst of oral cavity: a developmental lesion?, 66
Diagnosis: Heterotopic gastrointestinal cyst of the oral cavity The MRI scan revealed a multilobulated 3.5 × 2.7 × 3.0-cm cystic structure in the floor of the mouth. There was no evidence of invasion into adjacent structures. The tongue was displaced superiorly and posteriorly, causing a slight narrowing of the oropharynx. A normal-appearing thyroid gland was seen. The differential diagnosis included ranula (mucocele), dermoid and epidermoid cysts, thyroglossal duct cyst, retention cyst of the salivary gland, lymphoepithelial cyst, cystic hygroma (lymphangioma), cyst with heterotopic gastrointestinal mucosa, and sialolithiasis of the sublingual or submandibular glands. The initial biopsy specimen revealed gastric foveolar, squamous, and ciliated columnar epithelium, findings that were highly suggestive of a congenital heterotopic gastrointestinal cyst. Excision of the lesion revealed a red fibromembranous piece of tissue measuring 5.0 × 2.0 × 0.3 cm in thickness and containing soft, white, amorphous cyst material and fluid. Pathologic evaluation revealed ciliated columnar epithelium with focal keratinization, findings that were consistent with a foregut duplication cyst. Heterotopic gastrointestinal cyst of the oral cavity, also known as a foregut duplication or choristomatic cyst, is an extremely rare lesion. It has been reported in the anterior two thirds of the tongue and especially in the floor of the mouth. Features of the cyst include a coat of alimentary (usually smooth) muscle, a mucosal lining similar to that of some part of the alimentary tract, and attachment to some part of the alimentary tract.1-3 Several theories of pathogenesis have been proposed. The most widely accepted theory is that there is a physical displacement of gastrointestinal cells during the fourth week of embryogenesis, when the primitive stomach lies in the neck region. The misplaced embryonic gastric rests become entrapped at the midline of the tongue by fusion of the lateral lingual swellings over the tuberculum impar. The cysts have also been attributed to invasive migration and local differentiation of existing pluripotential cells. A difference in cellular differentiation between the cyst and normal gastrointestinal mucosa is supported by the results of lectin studies that have demonstrated a difference in mucin composition.4,5 Large intraoral cysts in the newborn may cause airway obstruction. Prenatal ultrasound can be used for early diagnosis, possible prenatal or immediate postnatal aspiration, appropriate counseling, and preparation at delivery. Elective intubation is preferred in patients with large cysts. The tongue may be retracted forward to allow pharyngeal access. Tracheotomy may be necessary if intubation is not possible, and, if anticipated, preparation should be made in the delivery room prior to delivery. The placental cord should be left undivided until the airway is secured. When the airway is secure and the patient stable, imaging can play an important role in the diagnosis and treatment of patients with congenital head and neck masses. There are several choices for imaging of congenital head and neck malformations. Ultrasound is a safe method of prenatal evaluation and a good tool for monitoring progression, but it is technique sensitive and lacks detail because of its low penetration. Magnetic resonance imaging is superior in soft tissue definition, thereby facilitating determination of lesion extent, invasion of surrounding tissues, and surgical planning. It is most useful in distinguishing lymphatic from vascular malformations when contrast enhancement is used. Computed tomography is superior to MRI in delineating bony deformation and phleboliths (pathognomonic for venous malformation). If thyroglossal duct cyst or lingual thyroid is suspected, a thyroid scan should also be performed to verify the location of the thyroid.3 In this particular case, prenatal ultrasound and aspiration had shown a fluid-filled cyst. Suspicion was low for vascular malformation and bony involvement. Thus, MRI was performed for confirmation of the relationship of the cyst to surrounding anatomy and for surgical planning. In the case of gastrointestinal cysts, recurrence is likely with aspiration alone. Therefore, wide excision with removal of mucosal lining is the treatment of choice once a diagnosis is made. The definitive diagnosis of a gastrointestinal cyst relies on tissue diagnosis, as was the case in this clinical episode. Once the mass is excised, the patient should be kept intubated until edema has subsided. Oral feeding may begin after extubation. With a large cyst, tongue coordination may be poor and gavage feeding may be required temporarily. Long-term prognosis is excellent, with complete resolution expected.2,6,7 The present case was managed with wide excision of the sublingual cyst via an intraoral approach. The cyst tract was excised directly down to the mandible. The patient underwent extubation on postoperative day 1, and oral feeding was begun that day. The patient was suckling normally by 1 week after surgery. Follow-up 6 months after surgery revealed that the floor of the patient's mouth was normal and there was no evidence of recurrence. Residents and fellows in otolaryngology are invited to submit quiz cases for this section and to write letters to the ARCHIVES commenting on cases presented. Quiz cases should follow the patterns established. See "Instructions for Authors." Material for CLINICAL PROBLEM SOLVING: RADIOLOGY should be mailed to the Editor. Reprints not available from the authors. References 1. Mirchandani RSciubba JGloster ES Congenital oral cyst with heterotopic gastrointestinal and respiratory mucosa. Arch Pathol Lab Med.1989;113:1301-1302.PubMedGoogle Scholar 2. Said-Al-Naief NFantasia JESciubba JJRuggiero SSachs S Heterotopic oral gastrointestinal cyst: report of 2 cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod.1999;88:80-86.PubMedGoogle Scholar 3. Erdem ETuz HHGunhan O Gastric mucosal choristoma of the tongue and floor of the mouth. J Oral Maxillofac Surg.2001;59:210-212.PubMedGoogle Scholar 4. Takato TItoh MYonehara Y Heterotopic gastrointestinal cyst of the oral cavity. Ann Plast Surg.1990;25:146-149.PubMedGoogle Scholar 5. Woolgar JASmith AJ Heterotopic gastrointestinal cyst of oral cavity: a developmental lesion? Oral Surg Oral Med Oral Pathol.1988;66:223-225.PubMedGoogle Scholar 6. Chen MKGross ELobe TE Perinatal management of enteric duplication cysts of the tongue. Am J Perinatol.1997;14:161-163.PubMedGoogle Scholar 7. Awouters PReychler H Enteric duplication in the oral cavity. Int J Oral Maxillofac Surg.1991;20:12-14.PubMedGoogle Scholar
Archives of Otolaryngology - Head & Neck Surgery – American Medical Association
Published: Mar 1, 2004
Keywords: cysts,radiology specialty,mouth
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