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Lingual tonsil hypertrophy causing severe dysphagia: Treatment with plasma-mediated radiofrequency-based ablation (Coblation).

Abstract

Lingual tonsil hypertrophy is an uncommon cause of upper aerodigestive tract pathology. We present the case of a 17-year-old boy who developed severe dysphagia and subsequent weight loss as a result of lingual tonsil hypertrophy. He was successfully treated with plasma-mediated radiofrequency-based ablation (Coblation). In the past, traditional surgical procedures for lingual tonsil hypertrophy were difficult to perform and recovery was difficult, but the introduction of Coblation has made lingual tonsillectomy much easier.

Introduction

The lingual tonsils, located at the base of the tongue, are part of the Waldeyer ring, which is made up of lymphoid tissue that surrounds the oropharyngeal inlet and includes the palatine tonsils and the adenoid pad. While symptomatic enlargement of the palatine tonsils and adenoids is common in the pediatric population, symptomatic enlargement of the lingual tonsils is not. Enlargement of the lingual tonsils can cause a variety of symptoms, including obstructive sleep apnea, (1) globus sensation, (2) and persistent cough. (3) The English-language literature contains limited reports of lingual tonsil hypertrophy in an adult that had caused significant dysphagia and that was treated conservatively. (4,5)

A variety of surgical techniques have been used to perform lingual tonsillectomy, including cold-knife and snare procedures, electrocautery, and laser resection. In this report, we describe the case of an adolescent with severe dysphagia secondary to lingual tonsil hypertrophy who was treated with plasma-mediated radiofrequency-based ablation (Coblation; ArthroCare Corp.; Sunnyvale, Calif.).

Case report

A 17-year-old boy was referred to the Division of Otolaryngology-Head and Neck Surgery for evaluation of a 6-month history of increasing dysphagia. By the time he presented, his swallowing difficulty had become so severe that he was able to swallow only liquids, and he had lost more than 60 lbs. He reported that he could not lie flat to sleep because of obstructive sleep apnea, and he required two pillows to sleep comfortably. He had undergone adenotonsillectomy at the age of 8 years.

Prior to his presentation to the ENT service, the patient had been evaluated by several other physicians. He had been initially referred by his primary pediatrician to a pediatric gastroenterologist, who obtained an esophagogram, an upper GI series, and a gastric emptying study; the results of all were normal. A pH probe revealed a Johnson-DeMeester score of 21.7 (normal: <22). Serologic tests for Wilson disease and for a rheumatologic cause of the dysphagia were negative. The patient was treated for gastroesophageal reflux disease (GERD) for several months, but his symptoms did not resolve. A formal neurologic evaluation by a pediatric neurologist was also undertaken, but it did not provide any explanation for the patient's prolonged dysphagia.

The patient then underwent an upper endoscopy, which revealed that the lingual tonsils were greatly enlarged and that there was a slight irregularity of the mucosa in the upper one-third of the esophagus. Findings on the remainder of the examination were normal. Biopsies taken of the distal esophageal mucosa during the endoscopy did not reveal any eosinophilic esophagitis. He was then referred to a pediatric otolaryngologist, who performed transnasal fiberoptic laryngoscopy. This examination revealed that the extremely large lingual tonsils had filled the entire vallecula and were causing the epiglottis to retroflex and approximate the posterior pharyngeal wall (figure 1). Magnetic resonance imaging (MRI) of the neck with and without contrast revealed that the tonsil enlargement was symmetrical (figure 2).

[FIGURE 1 OMITTED]

[FIGURE 2 OMITTED]

Based on the findings of this extensive workup, the only explanation for the patient's dysphagia was lingual tonsil hypertrophy. In order to rule out lymphoma of the lingual tonsil and to provide relief of the upper airway obstruction, it was recommended that he undergo surgical removal of the lingual tonsils. He was taken to the operating room, where he underwent nasotracheal intubation. After a complete evaluation of the airway, biopsies of the lingual tonsil were obtained and sent to pathology. The patient was then placed into suspension with alaryngoscope placed just proximal to the vallecula. A rigid 25[degrees] endoscope was placed through the laryngoscope, and the enlarged lingual tonsils were thus well visualized. A Coblation wand was used to completely ablate the lingual tonsil tissue from the vallecula and to achieve hemostasis (figure 3). All visible lymphoid follicles were removed. The patient was extubated without incident and monitored in the hospital overnight. Pathology revealed benign follicular hyperplasia. The patient did well postoperatively, and his diet was easily advanced from clear liquids to soft solids by postoperative day 2.

At follow-up, the patent's dysphagia remained resolved. Flexible transnasal laryngoscopy showed that his vallecula and tongue base were still clear of obstructing lingual tonsil tissue, and his epiglottis had resumed its natural upright position. He reported that swallowing was much easier and that he was able to eat a regular diet. By 6 months postoperatively, he had regained some of his lost weight.

[FIGURE 3 OMITTED]

Discussion

Lingual tonsil hypertrophy as a cause of dysphagia is unusual. The symptoms caused by this "silent" part of the Waldeyer ring are usually vague. In addition to obstructive sleep apnea, (1) globus sensation, (2) and persistent cough, (3) they include persistent sore throat and, in rare cases, airway obstruction. (6-8) These symptoms are frequently seen in patients with other disorders, particularly GERD. In fact, our patient was treated for GERD (unsuccessfully) for several months prior to his endoscopic examination.

The large volume of tissue present in the vallecula in our patient could have certainly caused his dysphagia. The size of the lingual tonsils forced the epiglottis to retroflex toward the pharyngeal wall. This might have made elevation of the larynx difficult during deglutition, accounting for the sensation of dysphagia. The fact that our patient was able to swallow liquids suggests that his dysphagia to solids was likely the result of the sheer volume of tissue present; he was not able to adequately move a solid bolus past the tissue-filled vallecula.

Lingual tonsillectomy is infrequently performed. Often-cited reasons for the low case volume are the risk of airway edema, difficulty in achieving adequate tongue-base exposure, poor visualization, a bloody operative field, and severe postoperative pain. On the other hand, as the use of Coblation surgery has become more popular, its feasibility as a procedure for performing lingual tonsillectomy and the positive outcomes associated with it have been documented in the otolaryngology literature. (9,10) The major benefits of Coblation tonsillectomy are its relative ease of use and dissection and the excellent hemostasis it provides. The lingual tonsillectomy in our patient was relatively easy to perform, it was achieved in a nearly bloodless field, and it was followed by an easy recovery with symptom resolution. The surgical technique that we used represented a combination of the approaches described by Maturo and Mair (9) and by Robinson et al. (10) The suspension laryngoscope facilitates the complete view of the vallecula, while the 25 [degrees] endoscope provides a magnified and angled view of the surgical field.

In light of the nonspecific symptoms and the difficulty encountered in examining the area, a high index of suspicion is necessary to adequately diagnose and treat dysphagia caused by lingual tonsil hypertrophy. Advances in endoscopic instrumentation and Coblation technology enable a safe and thorough removal of the tissue with symptom resolution.

References

(1.) Olsen KD, Suh KW, Staats BA. Surgically correctable causes of sleep apnea syndrome. Otolaryngol Head Neck Surg 1981;89(5):726-31.

(2.) Caylakli F, Akkuzu B, Avci S. Lingual tonsillar hypertrophy: A case report. Kulak Burun Bogaz Ihtis Derg 2004; 13 (1-2):28-30.

(3.) Lewis M, McClay JE, Schochet R Lingual tonsillectomy for refractory paroxysmal cough. Int J Pediatr Otorhinolaryngol 2000;53(1):63-6.

(4.) Fitzgerald P, O'Connell D. Massive hypertrophy o f the lingual tonsils: An unusual cause of dysphagia. Br J Radiol 1987;60(713):505-6.

(5.) Bock IM, Trask DK. Coblation-assisted lingual tonsillectomy for dysphagia secondary to tongue base hypertrophy. Ann Otol Rhinol Laryngol 2008; 117(7):506-9.

(6.) Cohle SD, Jones DH, Puri S. Lingual tonsillar hypertrophy causing failed intubation and cerebral anoxia. Am J Forensic Med Pathol 1993;14(2):158-61.

(7.) Dindzans LI, Irvine BW, Hayden RE. An unusual case of airway obstruction, l Otolaryngol 1984;13(4):252-4.

(8.) Lin AC, Koltai PJ. Persistent pediatric obstructive sleep apnea and lingual tonsillectomy. Otolaryngol Head Neck Surg 2009;141(1):81-5.

(9.) Maturo SC, Mair EA. Coblation lingual tonsillectomy. Otolaryngol Head Neck Surg 2006;135(3):487-8.

(10.) Robinson S,Ettema SL, Brusky L, Woodson BT. Lingual tonsillectomy using bipolar radiofrequency plasma excision. Otolaryngol Head Neck Surg 2006;134(2):328-30.

Sarah E. Mowry, MD; Marvin Ament, MD; Nina L. Shapiro, MD

From the Division of Otolaryngology-Head and Neck Surgery (Dr. Mowry and Dr. Shapiro) and the Division of Pediatric Gastroenterology (Dr. Ament), David Geffen School of Medicine, UCLA Medical School, Los Angeles.

Corresponding author: Nina L. Shapiro, MD, UCLA SURG-HD & NK, Box 951624, 62-158A CHS, Los Angeles, CA 90095-1624. E-mail: nshapiro@ucla.edu

Financial disclosure: Dr. Shapiro reported having received an honorarium as a speaker/consultant for ArthroCare.

Funding/support: No funding was received to support this project.
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Article Details
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Title Annotation:ORIGINAL ARTICLE
Author:Mowry, Sarah E.; Ament, Marvin; Shapiro, Nina L.
Publication:Ear, Nose and Throat Journal
Article Type:Clinical report
Geographic Code:1USA
Date:Mar 1, 2010
Words:1507
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