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Idiopathic acquired supraglottic web: a case report.

Abstract

We report a case of an idiopathic acquired supraglottic web in an 83-year-old man. The web was managed with a combination of dilation and unilateral C[O.sub.2] laser excision. Subsequent to removal, the web recurred three times; it was removed in the same fashion twice and left alone once. To the best of our knowledge, this is the first reported case of a truly idiopathic acquired laryngeal web.

Introduction

Laryngeal webs are abnormal connections of epithelium covered fibrous tissue that span two structures within the larynx. The introduction of indirect laryngoscopy in the mid-19th century led to the first reported case of a laryngeal web in vivo by Zurhelle in 1869.1 Many more cases have been reported since then, but laryngeal webs are still a relatively uncommon finding--especially in the supraglottic area.

We report a new case of supraglottic web. To the best of our knowledge, this is the first reported case of a truly idiopathic acquired laryngeal web.

Case report

An 83-year-old man presented with a complaint of dysphonia of 8 weeks' duration. He was a retired mechanical engineer with no significant medical history. Flexible nasendoscopyrevealed mild erythema of the vocal folds. The patient was reassured and advised voice rest.

At follow-up 3-months later, the patient's symptoms had worsened. In addition to a high-pitched voice, he now had a mild degree of stridor. Repeat flexible nasendoscopy revealed thickened aryepiglottic folds and a small but distinct supraglottic web that had not been present during the previous examination (figure 1). The web was located anteriorly, and it was noted to prolapse during inspiration. No intervention was administered at this time.

Over the next 9 months, the patient's dysphonia and stridor continued to progress, and he began to complain of shortness of breath. Findings on extensive investigation of his cardiac and pulmonary function were unremarkable. Hematologic and biochemical investigations were performed to rule out the presence of autoimmune and granulomatous diseases, including Wegener granulomatosis and sarcoidosis. Screenings for elevated levels of extractable nuclear antigens, double-stranded DNA, cytoplasmic-staining antineutrophil cytoplasmic antibodies (c-ANCA), and angiotensin-converting enzyme were negative. Repeat flexible nasendoscopyrevealed that the web now covered approximately 70% of the larynx. Beneath the larynx, both vocal folds were mobile.

Intraoperatively, the web was 4 mm thick, and it had originated 17 mm above the true vocal folds. The lumen of the web was dilated from 14 to 32 Fr with esophageal bougies, then C[O.sub.2] laser microlaryngoscopy was used to excise the web from the right side only. The appearance of the laryngeal ventricles, subglottic area, true vocal folds, and false vocal folds was normal. Histopathologic analysis of the excised web tissue detected no evidence of granulomatous disease, acid-fast bacilli, or fungal infection.

Following surgery (figure 2), the patient's symptoms were substantially improved. However, upon review 1 month postoperatively, the stridor had returned. Flexible nasendoscopy revealed the presence of another anterior supraglottic web. This one covered the anterior 25% of the larynx. The web again was managed with dilation and unilateral laser excision, but it recurred three times during the next 13 months. Twice it was managed in the same way, but treatment was deferred the third time, as this web involved less than 20% of the larynx. Some 14 months after the most recent removal, the patient was asymptomatic.

[FIGURE 1 OMITTED]

Discussion

The prevalence of congenital laryngeal webs worldwide is approximately 1 in every 10,000 births. (2) Acquired laryngeal webs are more common, with a prevalence of approximately 1.5 per 10,000 births. (3) Acquired webs can be caused by endotracheal intubation and trauma. (4) In addition, there have been isolated reports of cases caused by pseudomembranous tracheobronchitis secondary to Bacillus cereus infection, (5) as well as a relationship between microwebs and refractory nodules. (6) The larynx can also be affected by (1) Crohn disease lesions that involve the glottic area, (7) (2) mucous membrane pemphigoid, (8) and (3) infiltrate typical of plasmacytosis. (9) Finally, they may also arise following laser surgery on the larynx (10) or as a complication of laryngopharyngeal reflux. (11)

[FIGURE 2 OMITTED]

We did not carry out dual-channel pH monitoring in this case because our patient reported no change of circumstances regarding his medications, diet, and weight, and he had never complained of any symptoms suggestive of gastroesophageal reflux disease. In all three of the excised specimens we examined in this case, microscopy revealed focal active inflammation with mature plasma cells. The progressive nature of the patient's symptoms and the fact that the size of the webs steadily increased reinforce the fact that these webs were acquired.

Our treatment involved dilating the web and excising one side of it with a C[O.sub.2] laser. We felt that excising both sides of a web would result in a higher chance of recurrence or synechiae formation. Some laryngeal webs have been successfully treated in a single stage with endoscopic lysis and topical application of mitomycin C. (12) This antibiotic is a potent alkylating agent. However, there has been a reported case of laryngeal carcinoma in a nonsmoking adult that might have been related to topical mitomycin C use. (13) We offered mitomycin C application to our patient. However, after reading about it on the Internet, he declined to give consent because he was wary of having a cytotoxic agent used for a benign condition.

Acknowledgment

We thank Mrs. H.E. Cruickshank for her comments during the preparation of this work.

References

(1.) Zurhelle E. Ein Fall von congenitaler Laryngostenose. Berl Klin Wchnschr 1869;6:544.

(2.) Siggers BR, Ross O, Randall C, Connett G. A rare cause of upper airway obstruction in a 5-year-old girl: A laryngeal web. Paediatr Anaesth 2003;13(8):722-4.

(3.) McGuirt WF, Salmon J, Blalock D. Normal speech for patients with laryngeal webs: An achievable goal. Laryngoscope 1984;94(9): 1176-9.

(4.) Lundy DS, Casiano RR, Shatz D, et al. Laryngeal injuries after short- versus long-term intubation. J Voice 1998;12(3):360-5.

(5.) Strauss R, Mueller A, Wehler M, et al. Pseudomembranous tracheobronchitis due to Bacillus cereus. Clin Infect Dis 2001;33(5):E39-41.

(6.) Ford CN, Bless DM, Campos G, Leddy M. Anterior commissure microwebs associated with vocal nodules: Detection, prevalence, and significance. Laryngoscope 1994;104(11 Pt 1): 1369-75.

(7.) Bradley PJ, Ferlito A, Devaney KO, Rinaldo A. Crohffs disease manifesting in the head and neck. Acta Otolaryngol 2004; 124(3): 237-41.

(8.) Alexandre M, Brette MD, Pascal F, et al. A prospective study of upper aerodigestive tract manifestations of mucous membrane pemphigoid. Medicine (Baltimore) 2006;85(4):239-52.

(9.) Timms MS, Sloan P, Balzan AP. Idiopathic plasmacytosis ofthe oral and supraglottic mucosa. J Laryngol Otol 1988;102(7):646-8.

(10.) Wetmore SJ, Key JM, Suen JY. Complications of laser surgery for laryngeal papillomatosis. Laryngoscope 1985;95(7 Pt 1):798-801.

(11.) Holland BW, Koufman JA, Postma GN, McGuirt WF Jr. Laryngopharyngeal reflux and laryngeal web formation in patients with pediatric recurrent respiratory papillomas. Laryngoscope 2002; 112 (11):1926-9.

(12.) Roh JL. Application of mitomycin C after endoscopic lysis of congenital laryngeal web combined with epiglottic hypoplasia in a middle-aged man. Acta Otolaryngol2006;126(4):438-41.

(13.) Agrawal N, Morrison GA. Laryngeal cancer after topicalmitomycin application. J Laryngol Otol2006;120(12):2075-6.

Corresponding author: Mr. Anastasios Kanatas, Specialist Registrar, Department of Oral and Maxillofacial Surgery, Leeds Teaching Hospitals, Great George St., Leeds LS13EX, UK. Email: a.kanatas@ doctors.org.uk

David Pegg, MBChB, BSc(Hons); Anastasios Kanatas, MRCS, PhD; Zvoru Makura, MBChB, FRCSEd

From the Department of Anaesthesia (Dr. Pegg) and the Department of Ear, Nose, and Throat (Mr. Makura), Leeds General Infirmary, Leeds, U.K.; and the Department of Oral and Maxillofacial Surgery, Leeds Teaching Hospitals and St. lames Institute of Oncology, Leeds (Mr. Kanatas).
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Title Annotation:ORIGINAL ARTICLE
Author:Pegg, David; Kanatas, Anastasios; Makura, Zvoru
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:Oct 1, 2011
Words:1295
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