La tunisie Medicale - 2019 ; Vol 97 ( n°05 ) : 736-738
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Résumé

Le laryngocèle est une lésion bénigne rare du larynx causée par une dilatation anormale du saccule laryngé. Nous rapportons le cas d'un home de 78 ans présentant une masse molle cervicale bilatérale indolore. Le diagnostic de laryngocèle externe bilatérale a été confirmé par scanner et le patient a subi une résection chirurgicale.

Mots Clés
Article

Introduction:
Laryngocele is a rare benign lesion of the larynx caused by an abnormal dilatation of the laryngeal saccule(1). It can extend internally into the airway or externally through the thyrohyoid membrane, and may be present at any age. Laryngoceles are classified as internal, external or combined according to their localization. Despite the few cases of bilateral external laryngoceles reported in the literature,  this  presentation should always be mentioned among differential diagnosis of upper airway problems (2). Through this case report we tried to understand the clinical aspect of bilateral external laryngoceles and to insist on the surgical management using the external cervical approach.
Case report:
A 78-year-old man, with no significant past medical history, was referred to our department with the complaint of progressive hoarseness and bilateral neck swelling for 3 months after two days of intense coughing. He was a chronic smoker (20 cigarettes per day for 30 years). He was a farmer and did not play wind instruments nor have chronic constipation.
Neck examination showed bilateral painless soft mass, covered with normal skin, manually reducible, protrusive after Valsalva manoeuvre (figure 1).
Cervical CT-scan showed bilateral cystic lesions: the left one was 5cm large, associated with an hydroaeric level and the right one was 3cm large fully pneumatized (figure2).
Direct laryngoscopy under general anesthesia revealed a swelling in the left vestibular fold without suspect lesion. We performed systematic biopsies which did not show any evidence of malignancy at the histological examination. Surgical resection of bilateral laryngocele was performed by an external cervical approach (figures 3,4 and 5).  Post-operative recovery was uneventful and the patient was discharged.
The final histological examination confirmed the diagnosis of bilateral laryngocele.
Neither complications nor recurrences were observed after 12 months of follow-up.
Discussion:
Laryngocele is an abnormal dilatation of laryngeal saccule which communicates with the lumen of the larynx (1). It was first described by Dominique Jean Larrey, Napoleon’s surgeon in Egypt in 1829, who found it in muezzins (3), thereafter, Virchow introduced the term “laryngocele” in 1887 (4).
The incidence of laryngocele is estimated to be 1 per 2.5 million of the population per year. This disease is more frequent in men with a peak incidence in the sixth decade of life (1,5).
Causes behind genesis of laryngoceles are not well understood. In adults, an increase in intraluminal pressure in the laryngeal saccule is frequently associated, such as wind  instruments playing, singing, weight lifting or glass blowing, association with laryngeal carcinoma and atmospheric pressure changes during air travel (4,6,7). It may also occur in patients having congenital predisposition8. Laryngocele can occur in the left or the right side. Combined type is the most frequent type (44%-50%) followed by the internal type (30%) then the external type (20%-26%) (2,8). About 85% of laryngoceles are unilateral (4,9). The case presented is bilateral, which is a rare event.
Clinically, laryngocele usually manifest with hoarseness and neck swelling (4). But most of them remain asymptomatic with a post-mortem discovery reaching 6% (2).
The main differential diagnosis includes saccular cysts, branchial cysts, neck abscesses, oncocytic papillary cystadenoma, and lymphadenopathies (9-11).
The diagnosis of laryngocele is based on clinical findings, endoscopic examination of the larynx, and imaging. It should be noted that all patients with upper airway problems should undergo endoscopic evaluation to ascertain the diagnosis. Cervical computed tomography is useful to diagnose and define the type of laryngocele. Internal laryngoceles are limited by the thyrohyoid membrane, while both combined and external laryngoceles lie superficial to the thyrohyoid membrane (10,11).
Treatment of symptomatic cases depends on the size and type of laryngocele. Nowadays, internal laryngoceles are treated in most cases via endoscopic approach using a CO2 laser, while external and combined types are treated usually via an external approach. Some cases of combined laryngocele are treated via endoscopic approach by drawing the lateral external component into the laryngeal lumen (1,9,10). Both laryngoceles were managed at the same time in cases of bilateral forms reported in the literature (4,9,11,12).
Conclusion:
Laryngocele is a usually unilateral disease, but some bilateral cases were reported. Malignancies should be always ruled out due to the fact of association between laryngocele and laryngeal carcinoma. Treatment must be based on the type and the size of the laryngocele and benefit from the advanced new surgical technologies such as laser and robotic surgery.


Figure1:bilateral neck swelling (blue arrows)


Figure2: An axial cervical CT-scan showing bilateral laryngocele (asterisks)


Figure 3: left side laryngocele (blue arrow)


Figure4: right side laryngocele


Figure 5: both laryngoceles resected

References:
1.    Zelenik K, Stanikova L, Smatanova K, Cerny M, Kominek P. Treatment of Laryngoceles: what is the progress over the last two decades? BioMed research international 2014; 2014:819453.
2.    Stocker DJ, Kelly WF, Fitzpatrick TM, Bernet VJ, Burch HB. Bilateral external laryngoceles following radioiodine ablation for Graves disease. Arch Intern Med 2002; 162:2007-2009.
3.    Mitroi M, Capitanescu A, Popescu FCet al. Laryngocele associated with laryngeal carcinoma. Rom J Morphol Embryol 2011; 52:183-185.
4.    Prasad KC, Vijayalakshmi S, Prasad SC. Laryngoceles - presentations and management. Indian J Otolaryngol Head Neck Surg 2008; 60:303-308.
5.    Raine JI, Allin D, Golding-Wood D. Laryngopyocoele presenting with acute airway obstruction. BMJ Case Reports 2014; 2014:bcr2014204102.
6.    Spinosi MC, Mezzedimi C, Monciatti G, Passali D. Internal Laryngocele: Unusual onset in a 91-year-old female patient. Sultan Qaboos University medical journal 2018; 18:e104-e106.
7.    Akbas Y, Ünal M, Pata Y. Asymptomatic bilateral mixed-type laryngocele and laryngeal carcinoma. Eur Arch Otorhinolaryngol 2003; 261.
8.    Keles E, Alpay HC, Orhan I, Yildirim H. Combined laryngocele: a cause of stridor and cervical swelling. Auris, nasus, larynx 2010; 37:117-120.
9.    Aksoy EA, Elsürer Ç, Serin GM, Ünal ÖF. Bilateral internal laryngoceles mimicking asthma. J Res Med Sci 2013; 18:453-456.
10.    Oukessou Y, Abada RL, Roubal M, Mahtar M. Laryngocele: an unusual presentation. Eur Ann Otorhinolaryngol Head Neck Dis 2015; 132:177-178.
11.    Werner RL, Schroeder JW, Castle JT. Bilateral laryngoceles. Head Neck Pathol 2014; 8:110-113.
12.    Suqati AA, Alherabi AZ, Marglani OA, Alaidarous TO. Bilateral combined laryngocele. Saudi Med J 2016; 37:902-903.

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