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Bilateral internal laryngoceles mimicking asthma.
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PMID:  24174956     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Laryngocele is an air-filled, abnormal dilation of the laryngeal saccule that extends upward within the false vocal fold, in communication with the laryngeal lumen. A case of 43-year-old male with bilateral internal laryngoceles, who has been treated as asthma for 4 years, is presented. The patient had dyspnea, cough, and excessive phlegm for a month and a late onset stridor. Flexible nasopharyngolaryngoscopy showed bilateral cystic enlargements of the false vocal folds and true vocal folds could not be visualized. Laryngeal CT without contrast enhancement showed bilateral internal laryngoceles. Submucosal total excision of bilateral cystic masses including parts of false vocal folds was performed. The symptoms resolved immediately after surgery. Although the incidence of internal laryngocele is rare, it should be remembered in the differential diagnosis of upper airway problems and diagnostic flexible nasopharnygolaryngoscopy is routinely indicated for airway evaluation in at-risk patients.
Authors:
Elif A Aksoy; Cağdaş Elsürer; Gediz M Serin; O Faruk Unal
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Journal of research in medical sciences : the official journal of Isfahan University of Medical Sciences     Volume:  18     ISSN:  1735-1995     ISO Abbreviation:  J Res Med Sci     Publication Date:  2013 May 
Date Detail:
Created Date:  2013-10-31     Completed Date:  2013-10-31     Revised Date:  2013-11-04    
Medline Journal Info:
Nlm Unique ID:  101235599     Medline TA:  J Res Med Sci     Country:  Iran    
Other Details:
Languages:  eng     Pagination:  453-6     Citation Subset:  -    
Affiliation:
Department of Otorhinolaryngology Head and Neck Surgery, Acıbadem University School of Medicine, Turkey.
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Journal Information
Journal ID (nlm-ta): J Res Med Sci
Journal ID (iso-abbrev): J Res Med Sci
Journal ID (publisher-id): JRMS
ISSN: 1735-1995
ISSN: 1735-7136
Publisher: Medknow Publications & Media Pvt Ltd, India
Article Information
Copyright: © Journal of Research in Medical Sciences
open-access:
Received Day: 06 Month: 3 Year: 2011
Revision Received Day: 24 Month: 6 Year: 2012
Accepted Day: 03 Month: 7 Year: 2012
Print publication date: Month: 5 Year: 2013
Volume: 18 Issue: 5
First Page: 453 Last Page: 456
PubMed Id: 24174956
ID: 3810585
Publisher Id: JRMS-18-453

Bilateral internal laryngoceles mimicking asthma
Elif A. Aksoyaff1
Çağdaş Elsürer1
Gediz M. Serinaff1
Ö. Faruk Ünalaff1
Department of Otorhinolaryngology Head and Neck Surgery, Acıbadem University School of Medicine, Turkey
1Department of Otorhinolaryngology Head and Neck Surgery, Acıbadem Health Care Group, Acibadem Fulya Hospital, Turkey
Correspondence: Address for correspondence: Dr. Elif Ayanoglu Aksoy, Department of Otorhinolaryngology Head and Neck Surgery, Acıbadem University School of Medicine, Acıbadem Maslak Hospital, Büyükdere Cad. No:40 Maslak, Istanbul, Turkey. E-mail: elifayanoglu@yahoo.com

INTRODUCTION

Laryngocele is an air-filled, abnormal dilation of the laryngeal saccule that extends upward within the false vocal fold, in communication with the laryngeal lumen.[1] Different theories regarding the development of laryngoceles include a congenital large saccule, weakness of laryngeal tissues, and increased intralaryngeal pressure, as observed in glass blowers, wind instrument players, street hawkers, and singers.[2]

Clinical relevance is rare, but laryngoceles may present themselves as hoarseness, neck mass, airway obstruction, and even, a neoplasm.[3] Laryngoceles commonly present unilaterally.[1] Up to date there are total five bilateral internal laryngocele cases reported in literature.[4, 5] The case presented here is the sixth bilateral internal laryngocele case who has been treated as asthma for 4 years.


CASE REPORT

A 43-year-old male admitted to the pulmonology clinic of Acibadem Healthw Care Group, Acibadem Masalk Hospital, Istanbul, Turkey, in 2011, with dyspnea, cough, and excessive phlegm for a month and a late onset stridor. The patient had a history of asthma and he had been treated as asthma for 4 years. The response of the patient to antiasthmatic drugs was not sufficient and asthmatic attacks were frequent. A bronchoscopy was performed and upper airway obstruction was detected. Atalectatic bronchial segments were found. Sixth bronchi of the right inferior lung lobe was totally obstructed. The patient was referred to the Ear Nose and Throat (ENT) Clinic for upper airway evaluation. Flexible nasopharyngolaryngoscopy showed bilateral cystic enlargements of the false vocal folds and true vocal folds could not be visualized properly [Figures 1-3]. Supraglottic area was narrow. Laryngeal CT without contrast enhancement showed bilateral internal laryngoceles. Hypodense areas causing expansion at the level of aryepiglottic folds were detected [Figures 4 and 5]. Endolaryngeal laser surgery was done under general anesthesia. Submucosal total excision of bilateral cystic masses including parts of false vocal folds was performed. The symptoms resolved immediately after surgery. Airway was open and true vocal folds were visible during the postoperative endoscopic laryngeal examinations of the patient [Figures 6-8, Video 1]. The patient did not experience any respiratory event during the follow-up period after surgery. His last visit, free of symptoms, was at the sixth postoperative month.


DISCUSSION

Laryngoceles are dilatations of the laryngeal saccule within the ventricle of Morgagni. They were first described in 1829 by Larrey. They consist of a membranous sac located between the false vocal fold and the inner aspect of the thyroid cartilage.[2, 3] Laryngoceles have been reported to be five to seven times more frequent in males, with a peak incidence in the sixth decade of life. Eighty-five percent of laryngoceles have been found to be unilateral with no right or left-side predominancy.[1]

The case presented here is also male but he had bilateral internal laryngoceles, which is an extremely rare situation. Devesa et al.[4] presented a case series of nine laryngoceles, four of which were bilateral internal laryngoceles. Up to date including the bilateral laryngocele case reported by Pruszewicz et al.[5] in 2006, there are total five bilateral internal laryngocele cases reported in literature. The case presented here is the sixth bilateral internal laryngocele case.

An internal laryngocele is confined to the interior of the larynx and extends posterosuperiorly into the false vocal fold and the aryepiglottic fold. Internal laryngoceles appear on laryngoscopy as a smooth swelling of the supraglottis.[3]

The cause of laryngocele is unknown. It is associated with chronic cough, blowing in musical instruments, glass blowing, and laryngeal carcinoma. Its origins involve both congenital and acquired factors. In adults, a congenital defect or an anatomical variation of the sacculus may be the cause, as are acquired factors such are the cases of pharyngeal or laryngeal carcinomas, and people whom occupation or leisure involve raising intralaryngeal pressure, such as blowing musical instruments.[6] Some reports have suggested a link between previous neck surgery and laryngocele. Progression of a clinically significant laryngocele several years following tracheotomy was attributed to local trauma during the original tracheotomy placement, which could have caused an underlying weakness, defect, or mechanical obstruction of the laryngeal saccule.[7] Laryngoceles were reported in approximately 3% of supraglottic laryngectomy patients and it is related to the incomplete resection of the ventricle.[8]

Internal laryngoceles may interfere in speech production and cause snoring or hoarseness, and even upper airway obstruction as the case hereby presented. Other symptoms are a foreign body sensation, sore throat, and cough.[6] The case presented here is of concern because it is bilateral and has been misdiagnosed as asthma.

The diagnosis of a laryngocele is based on clinical findings, endoscopic examination of larynx, and imaging studies. The endoscopic examination and direct laryngoscopy reveal a false vocal fold and arytenoid swelling overlied with normal laryngeal mucosa. It should be noted that all the patients with symptoms of airway problems should at least go through an upper airway endoscopic evaluation to rule out upper airway obstruction. A flexible laryngoscopy is imperative in evaluation of the patient's airway, and this way the diagnosis may be achieved in the office setting.

Laryngeal MRI or CT can be used as an imaging technique. In MRI, an air-filled laryngocele typically appears as a low- signal cystic dilatation of the laryngeal ventricle.[1, 3]

Differential diagnosis includes saccular cyst, branchial cyst, neck abscess, and lymphadenopathy. Saccular cysts do not communicate with the laryngeal lumen, and it is usually filled with fluid.[6]

Various modalities of treatment have been used for resection of laryngoceles.[1, 9, 10] Although external approach for laryngocele resection is a traditional treatment method, use of CO2 laser in microlaryngoscopic surgery has become a preferred method in suitable cases.[1] Latest consensus for laryngocele treatment favors external approach for large or external laryngoceles, while endoscopic endolaryngeal resection for internal laryngoceles.[1] Internal laryngoceles may be removed endoscopically. Dursun et al.[1] have used CO2 laser for excision of internal laryngoceles. They have reported less operation times, minimal damage of endolarynx and vocal folds. They have reported that quality of voice and swallowing functions could be preserved in all patients, and none of their patients required tracheotomy and/or prolonged hospitalization. In the case presented here, the same endolaryngeal laser excision technique is used. The patient had a safe postoperative period without airway complications. He was discharged at the first postoperative day without any complications.

Furthermore, with the removal of the laryngoceles, he had an immediate improvement. The only postoperative complaint was pain while swallowing and it lasted only for a few days after surgery.

Although the incidence of internal laryngocele is rare, it should be remembered in the differential diagnosis of upper airway problems. With this case presented here, we want to emphasize the importance of diagnostic flexible nasopharnygolaryngoscopy in diagnosing upper airway pathologies. Laryngoscopy is routinely indicated for airway evaluation in at-risk patients.


See Video on: www.journals.mui.ac.ir/jrms Click here to view as Video 1

Notes

Source of Support: Nil

Conflict of Interest: None declared.

ACKNOWLEDGMENT

Special thanks to Çağlar Çuhadaroğlu, MD, professor of Pulmonology, for his support and help.


REFERENCES
1. Dursun G,Ozgursoy OB,Beton S,Batikhan H. Current diagnosis and treatment of laryngocele in adultsOtolaryngol Head Neck SurgYear: 2007136211517275541
2. Marom T,Roth Y,Cinamon U. Laryngocele: A rare long-term complication following neck surgeryJ VoiceYear: 201125272420430574
3. Keles E,Alpay HC,Orhan I. Combined Laryngocele: A cause of stridor and cervikal swellingAuris Nasus LarynxYear: 2010371172019410402
4. Devesa PM,Gufoor K,Lyod S,Howard D. Endoscopic CO2 laser management of LaryngoceleLaryngoscopeYear: 200211214263012172256
5. Pruszewicz A,Obrebowski A,Maciejewska B. Bilateral internal laryngocele with open nasality-report of a caseOtolaryngol PolYear: 20066093853
6. Felix JA,Felix F,de Mello LF. Laryngocele: A cause of upper airway obstructionBrass J OtorhinolaryngolYear: 2008741436
7. Upile T,Jerjes W,Sipaul F,El Maaytah M,Singh S,Howard D,et al. Laryngocele: A rare complication of surgical tracheostomyBMC SurgYear: 2006271417129390
8. Naudo P,Laccourreye O,Weinstein G,Jouffre V,Laccourreye H,Brasnu D. Complications and functional outcome after supracricoid partial laryngectomy with cricohyoidoepiglottopexyOtolaryngol Head Neck SurgYear: 199811812499450842
9. Thome R,Thome DC,De La Cortina RA. Lateral thyrotomy approach on the paraglottic space for laryngocele resectionLaryngoscopeYear: 20001104475010718436
10. Myssiorek D,Madnani D,Delacure MD. The external approach for submucosal lesions of the larynxOtolaryngol Head Neck SurgYear: 2001125370311593174

Figures

[Figure ID: F1]
Figure 1 

Flexible nasopharyngolaryngoscopic view of bilateral internal laryngoceles



[Figure ID: F2]
Figure 2 

Flexible nasopharyngolaryngoscopic view of bilateral internal laryngoceles



[Figure ID: F3]
Figure 3 

Flexible nasopharyngolaryngoscopic view of bilateral internal laryngoceles



[Figure ID: F4]
Figure 4 

Axial CT section of larynx showing bilateral internal laryngoceles



[Figure ID: F5]
Figure 5 

Sagittal CT section of larynx showing bilateral internal laryngoceles



[Figure ID: F6]
Figure 6 

Postoperative rigid endoscopic view of larynx. Note the healing of the left false vocal fold



[Figure ID: F7]
Figure 7 

Postoperative flexible nasopharyngolaryngoscopic view of larynx



[Figure ID: F8]
Figure 8 

Postoperative flexible nasopharyngolaryngoscopic view of larynx



Article Categories:
  • Case Report

Keywords: Internal laryngoceles, laryngoscopy, upper airway endoscopy.

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