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A Case Report of a Vallecular Cyst and Literature Review Tuan M. Hoang, DDS, MD,* and Beomjune B. Kim, DMD, MDy
Cysts in the vallecula are rare entities and seldom have been reported in the literature. However, these masses can have very important clinical implications. When they are small, they are usually asymptomatic. When they reach a large enough size, they can cause dysphagia, odynophagia, and acute airway complications. With the expanded scope of oral and maxillofacial surgery, the proper diagnosis and management of vallecular lesions could become a routine part of oral and maxillofacial surgical practice and aid in further development of the specialty. This report describes the case of a benign lymphoepithelial cyst of the vallecula. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg -:1.e1-1.e3, 2015 Cysts in the vallecula are rare entities and seldom have been reported in the literature. However, these masses can have very important clinical implications. When they are small, they are usually asymptomatic. When they reach a large enough size, they can cause dysphagia, odynophagia, and acute airway complications.1,2 With the expanded scope of oral and maxillofacial surgery, the proper diagnosis and management of vallecular lesions could become a routine part of oral and maxillofacial surgical practice and aid in further development of the specialty. This report describes the case of a benign lymphoepithelial cyst of the vallecula.
cedure proceeded without complications. Computed tomogram of the neck visualized a mass in the right vallecula with uniform radio-opacity that occupied much of the right side of the vallecula. There also were 4 benign coarse punctate calcifications. The mass showed no narrowing of the airway in this region (Figs 2, 3). The patient did not report any symptoms of dysphagia, odynophagia, or changes to her voice. She subsequently was scheduled for a formal panendoscopy. During the procedure, an excisional biopsy of the mass was performed. When the mass was excised, the capsule was violated and yellowishbrown substance exuded from the mass. The content of the mass was very similar to that found in a sebaceous cyst. The final pathology result was a benign lymphoepithelial cyst with a squamous epithelial lining (Figs 4, 5). At follow-up visits, the patient did not report any difficulty swallowing or breathing.
Report of Case
A 55-year-old woman with a medical history of hypertension, type 1 diabetes mellitus, and anxiety presented with the chief complaint of difficulty breathing through her nose. She previously underwent a septorhinoplasty. After her evaluation, she was scheduled for bilateral inferior turbinectomy. Because of her history of difficulties with intubation, the anesthesia team proceeded with a GlideScope. During this procedure, an exophytic, pedunculated mass with a pinkyellowish hue approximately 2 cm in diameter was found in the right vallecula (Fig 1). The intubation pro-
Discussion The term lymphoepithelial cyst was first introduced by Bernier and Bhaskar in 1958.3 There have been Q3 numerous case reports of this entity in the head and neck region occurring predominantly on the lateral aspect of the neck (branchial cleft cyst) and the parotid gland.
Received from the Department of Oral and Maxillofacial Surgery,
Received April 21 2015
Louisiana State University Health Sciences Center, New Orleans, LA. *Chief Resident.
Ó 2015 American Association of Oral and Maxillofacial Surgeons
Accepted May 1 2015
Address correspondence and reprint requests to Dr Kim: Depart-
ment of Oral and Maxillofacial Surgery, Louisiana State University Health Sciences Center, 1100 Florida Avenue, Box 220, New Orleans, LA 70119; e-mail: [email protected]
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web 4C=FPO FIGURE 1. The vallecular mass viewed through the GlideScope. Hoang and Kim. Vallecular Cyst. J Oral Maxillofac Surg 2015.
There is controversy regarding the pathogenesis of these lesions; however, enclavement of epithelium within lymph nodes is a well-known phenomenon, especially in the parotid glands and the lateral cervical region.4 Reports of these lesions occurring in the oral cavity are sparse, and reports of those occurring in the vallecular region are even sparser. When these lesions occurred intraorally, the floor of the mouth and the tongue were the most common locations.5-7 These lesions were mostly asymptomatic and were discovered mostly during routine dental examinations. Local excision is the treatment of choice and the prognosis is excellent, without recurrences.
FIGURE 3. Axial view shows the lesion on the right side of the vallecula. Note the coarse calcifications within the lesion. Hoang and Kim. Vallecular Cyst. J Oral Maxillofac Surg 2015.
Benign lymphoepithelial cysts in the vallecular and pharyngeal region are very rare.8 However, there is always a concern for acute airway compromise when they occur in this region. One theory as to why cysts can develop in the vallecular region states that ducts of the mucous gland or lingual tonsillar crypt can become obstructed from inflammation, irritation, or trauma, thus causing dilatation and cystic formations.2 The DeSanto classification subdivided laryngeal cysts Q4 into ductal, saccular, and thyroid cartilage foraminal cysts. More recent classifications have divided these lesions into congenital, retention, inclusion, and
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FIGURE 2. Sagittal view of radio-opaque lesion. The lesion occupies much of the right side of the vallecula but does not cause major narrowing of the airway.
FIGURE 4. Section showing a cystic lesion lined by stratified squamous epithelium; underneath this layer, lymphoid tissue can be visuQ8 alized (hematoxylin and eosin stain).
Hoang and Kim. Vallecular Cyst. J Oral Maxillofac Surg 2015.
Hoang and Kim. Vallecular Cyst. J Oral Maxillofac Surg 2015.
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HOANG AND KIM
FIGURE 5. Section showing the overlying mucosa, the epithelial lining, and subepithelial lymphoid tissue, some of which is arranged in germinal centers (hematoxylin and eosin stain). Hoang and Kim. Vallecular Cyst. J Oral Maxillofac Surg 2015.
lymphoepithelial cysts. Asymptomatic cysts in the vallecula can cause difficulty with intubation as was the case with the present patient and a GlideScope had to be used.2 When these lesions are small, they are usually asymptomatic; however, when they reach a large enough size, they can cause symptoms, such as dysphagia, odynophagia, and a change in the quality of the voice. Another important complication that can arise is when these cystic lesions become secondarily infected.1 A relatively large retrospective chart review performed by Berger et al1 showed that of 38 cases of complications related to vallecular cysts for which patients were admitted, 24 were associated with an infection leading to epiglottitis and abscess formation. When infections and abscess formation occurred, the airway had to be urgently secured with transnasal endotracheal intubation or tracheotomy or cricothyrotomy when the former was not successful. Although the contents of benign lymphoepithelial cysts resemble those of sebaceous cysts, sebaceous differentiation within the linings of these lesions is rare. In 2000, Fujiwara et al reported findings of sebaceous differentiation in the histologic findings of the lesions that were examined. The present case did not show sebaceous differentiation, but the content appeared to resemble the content of sebaceous cysts when the cyst was ruptured. Another question of concern is whether malignant transformations can occur with these lesions. Wu et al3 in their series of 64 cases reported that 2 of the cysts had dysplastic changes in the lining epithelium, which suggests that carcinomatous changes can occur. The clinical implications are important should this transformation take place in those lesions that occur in the pharyngeal region. The differential diagnoses for a mass in the vallecula in neonates and young children are rather narrow and can include lingual thyroid tissue, thyroglossal duct cyst, hemangioma, dermoid cyst, and cystic tumors, such as teratomas.9-11 When occurring in neonates
and infants, there is an increased risk for stridor, feeding difficulty, failure to thrive, and, rarely, sudden supraglottic airway obstruction and death.10 In adults, vallecular cysts are more common but less dangerous. Another possible diagnosis for a vallecular mass in the adult is lymphangioma, the involvement of which is usually caused by direct extension of a congenital lesion within the neck.12 Lymphoepithelial cysts are uncommon lesions. In the head and neck region, the parotid gland and the lateral aspect of the neck are the most common locations. There have been very few case reports of these cysts in the vallecula. When these lesions occur in the laryngeal region, they are usually asymptomatic. However, when they reach a large enough size, they can cause complications, including dysphagia, odynophagia, and voice changes. More serious complications include difficulty with intubation and secondary infection leading to abscess formations and epiglottitis, which can become manifest in acute airway complications. The primary mode of treatment is surgical excision, for which the recurrence rate is very low. Lymphoepithelial cysts rarely occur in the vallecular region and they should be reported when incidences are found. This will lead to an increased awareness so that more specific management strategies can be developed. Such strategies can mitigate airway complications when they do occur with these entities. Q7
References 1. Berger G, Averbuch E, Zilka K, et al: Adult vallecular cyst: Thirteen-year experience. Otolaryngol Head Neck Surg 138: 321, 2008 2. Kothandan H, Ho VK, Chan YM, et al: Difficult intubation in a patient with vallecular cyst. Singapore Med J 54:e62, 2013 3. Wu L, Chen J, Maruyama S, et al: Lymphoepithelial cyst of the parotid gland: Its possible histopathogenesis based on clinicopathologic analysis of 64 cases. Hum Pathol 40:683, 2009 4. Vickers RA, Gorlin RJ, Smart EA, et al: Lymphoepithelial lesions of the oral cavity. Report of four cases. Oral Surg Oral Med Oral Pathol 16:1214, 1963 5. Acevedo A, Nelson F: Lymphoepithelial cysts of the oral cavity. Report of nine cases. Oral Surg Oral Med Oral Pathol 31:632, 1971 6. Giunta J, Cataldo E: Lymphoepithelial cysts of the oral mucosa. Oral Surg Oral Med Oral Pathol 35:77, 1973 7. Yang X, Ow A, Zhang CP, et al: Clinical analysis of 120 cases of intraoral lymphoepithelial cyst. Oral Surg Oral Med Oral Pathol Oral Radiol 113:448, 2012 8. Noh HJ, Koo JS, Oh HY, et al: Lymphoepithelial cyst of the upper esophagus. Endoscopy 43(suppl 2 UCTN):E254, 2011 9. Leibowitz JM, Smith LP, Cohen MA, et al: Diagnosis and treatment of pediatric vallecular cysts and pseudocysts. Int J Pediatr Otorhinolaryngol 75:899, 2011 10. Raftopulos M, Soma M, Lowinger D, et al: Vallecular cysts: A differential diagnosis to consider for neonatal stridor and failure to thrive. JRSM Short Rep 4:29, 2013 11. Gutierrez JP, Berkowitz RG, Robertson CF: Vallecular cysts in newborns and young infants. Pediatr Pulmonol 27:282, 1999 12. Castle JT: Lymphangioma of the vallecula. Head Neck Pathol 5: 20, 2011
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