Coexistence of laryngoceles and laryngeal carcinoma is still being debated, and there are several suggested theories about the pathophysiological relationship between these two entities. We present the case of a 66-year-old male patient with bilateral laryngomucoceles and laryngeal squamos cell carcinoma. A systematic hystological examination of whole organ sections showed that the submucosal spreading of cancer around the saccular necks on both sides caused stenosis which probably created a one-way valve mechanism allowing air to enter the saccule but not to exit. Progression of the tumor completely obstructed the laryngeal opening, leading to glandular secretion stagnation and formation of laryngomucoceles

Dubravko Manestar

Igor Šegec

Josip Maleš

Ksenija Marjanović

Marinela Rosso

Mićo Rosso

Tatjana Šepić

English

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Coll. Antropol. 34 (2010) 2: 727–730Case reportBilateral Laryngoceles in Association withSquamos Cell Carcinoma: A Case Reportand Literature ReviewMarinela Rosso1, Ksenija Marjanovi}2, Josip Male{1, Tatjana [epi}3, Igor [egec1, Mi}o Rosso4and Dubravko Manestar31 Department of Otorhinolaryngology and Head and Neck Surgery, Osijek University Hospital Center, Osijek, Croatia2 Institute for Pathology and Forensic Medicine, Osijek University Hospital Center, Osijek, Croatia3 Department of Otorhinolaryngology and Head and Neck Surgery, Rijeka University Hospital Center, Rijeka, Croatia4 Department of Gynecology and Obstetrics, Osijek University Hospital Center, Osijek, CroatiaA B S T R A C TCoexistence of laryngoceles and laryngeal carcinoma is still being debated, and there are several suggested theoriesabout the pathophysiological relationship between these two entities. We present the case of a 66-year-old male patientwith bilateral laryngomucoceles and laryngeal squamos cell carcinoma. A systematic hystological examination of wholeorgan sections showed that the submucosal spreading of cancer around the saccular necks on both sides caused stenosiswhich probably created a one-way valve mechanism allowing air to enter the saccule but not to exit. Progression of the tu-mor completely obstructed the laryngeal opening, leading to glandular secretion stagnation and formation of laryn-gomucoceles.Key words: larynx, mucocele, laryngeal cancer, etiologyIntroductionThe laryngeal ventricle is the space between the vocaland ventricular fold. The bilateral upward extensionarising from the anterior end of the laryngeal ventricle isthe laryngeal saccule1. The laryngeal saccule is linedwith respiratory epithelium containing serum-mucus-se-creting glands, which provide lubrication for the vocalfolds. When the saccule is abnormally sized it becomeslaryngocela. The laryngocela is also lined with respira-tory epithelium and is connected to the laryngeal ventri-cle by a saccule neck2. According to the site of presenta-tion, three types of laryngoceles have been described:internal, external and mixed. The internal laryngocele isconfined to the interior of the larynx, and the externallaryngocele extends superiorly through the thyrohyoidmembrane to the neck. Simultaneous existence of inter-nal and external laryngoceles, medial and lateral to thethyrohyoid membrane, is termed a mixed or combinedlaryngocele3,4.Laryngoceles are normaly air-filled, but may be filledwith mucous to form laryngomucocele or can be filledwith pus and form laryngopyocela. Laryngomucoceles re-sults from the proliferation and secretion of mucousglands within an obstructed saccule neck of laryngocele5.Normally, the glandular serum-mucus secretion is evacu-ated through the saccule neck. After laryngeal trauma,neoplastic disorder or chronic inflammation, which cancause obstruction of the saccule neck, these secretionscan stagnate in the laryngocele4,6. The correlation be-tween laryngocele and laryngeal carcinoma is still beingdebated, and there are a few suggested theories aboutthe pathophysiological relationship between laryngomu-coceles and laryngeal carcinoma3,7,8.Case ReportA 66-year-old male approached our department with athree month history of progressive hoarseness, bilateralneck swelling, dysphonia and difficulty breathing. Hehad been a heavy smoker but had no other relevant med-ical history. A clinical examination revealed a 3.3 cm,727Received for publication May 20, 2009soft, painless mass that was extending on both sides ofhis neck beyond and lateral to the thyroid lamina. Nolymphadenopathy was detected during the neck exami-nation. A videolaryngoscopic examination of the larynxrevealed an exophytic mass that extended from the la-ryngeal surface of the epiglottis to both ventricular folds,anterior commisure and anterior part of the vocal folds,with decreased mobility on both sides. A direct laryn-goscopy was carried out under general anesthesia for bi-opsy, and a histopathologic examination revealed the le-sion to be well-differentiated squamos cell carcinoma.Computerized tomography (CT) scans assessed the ex-tent of tumour involvement and neck masses more pre-cisely, and at the supraglottic and glottic level showed abilateral tumour mass with erosion of the ossified thy-roid cartilage. The scans also detected the presence offluid-filled cavities in both laterocecervical regions de-limited by regular walls, about 2.5 cm in diameter, ex-tending up to the level of the hyoid bone (Figure 1).There was no evidence of enlarged cervical lymph nodes.A total laryngectomy and bilateral selective anterolateralneck dissection was performed and both laryngomu-coceles were excised.Macroscopic examination showed a large tumour onthe median laryngeal surface of the epiglottis, ventricu-lar and vocal folds and the deep structures of the larynx(Figure 2). Ossified thyroid cartilage was destroyed by acarcinoma. Macroscopically it seems that a carcinoma in-volves saccule neck on both sides. Hystological examina-tion showed well-differentiated squamos cell carcinomawhich infiltrated the deep structures of the larynx andinvaded ossified thyroid cartilage. Carcinoma has ex-tended subepithelially around the both sacculae necks,infiltrating submucosa of the internal parts of bothlaryngoceles. Overlaying pseudostratified ciliated colum-nar epithelium was intact (Figure 3). Two cervical lymphnode metastases in the right side were detected. The tu-mour was graded at T4N1M09. Four weeks after surgery, the patient underwent post-operative radiotherapy. Two years after treatment, thepatient remained free of tumour recurrence.Discussion and ConclusionThe term laryngocele was introduced by Virchow in1867, but the condition is thought to have first been re-ported in 1829 by the French military surgeon Domi-nique Larrey10. Incidence is 1:2,500,000 population peryear4,11. Peak incidence is in the sixth decade of life.Laryngoceles are unilateral in 85%, with no right- or left-side predominancy, and can be congenital and acquired11.It seems that they develop under conditions of prolongedand pronounced increase in intralaryngeal pressure, whichoccurs with glass blowing, wind instrument playing, sin-ging and persistent coughing6. They can also occur withconditions that produce a flap-valve mechanism, such astumours, inflammatory lesions and trauma4,6,12.Diagnosis of laryngocele was based on clinical find-ings, endoscopic examination of the larynx and imagingM. Rosso et al.: Laryngoceles and Laryngeal Carcinoma, Coll. Antropol. 34 (2010) 2: 727–730728Fig. 1. Axial CT scan showing fluid-filled cavities in the both lat-eral neck regions. Laryngomucoceles are passing through thethyreohyoid membrane at the level of the hyoid bone.Fig. 2. Macroscopically it seems that a carcinoma involves sa-ccule neck on both sides.Fig. 3. Hystological examination showed pseudostratified, cilli-ated columnar epithelium of the narrow stalk (black arrow). Insubmucous there is a well-differentiated squamous cell carcino-ma (white arrow) which narrows laryngeal opening.studies. A CT scan of the neck provided a definitive diag-nosis of laryngocele2,12.Coexistence of laryngoceles and laryngeal carcinomais still being debated and has been reported by severalauthors. It was first described by Marschik in 19278. Thefrequency of laryngoceles in laryngeal cancer specimensvaries between 4.9% and 28.8%, and in the normal larynxis about 2%13–15. This difference demonstrated the role ofcancer in the genesis of laryngoceles. In patients withlaryngocele, because of the high frequency of coexistentcarcinoma, detailed examination of the larynx is ne-cessary14. However, in patients with metastatic necknodes with unknown primary carcinoma, detailed exami-nation of the larynx and looking for laryngocele as a po-tential site of a primary carcinoma is recommended13.There are several suggested theories about the patho-physiological relationship between laryngomucocelesand laryngeal carcinoma. The first theory describes thedistortion of the saccule neck by a carcinoma, which maycreate a one-way valve that inflates and distends thesaccule. It seems that the laryngocele provides a pathwayof least resistance for a tumor to spread. This theory doesnot explain the existence of contralateral laryngocele.The second theory is that carcinoma may arise from thelining epithelium of a large saccule. A preexisting laryn-gocele might provide a preferential site for tumor devel-opment. There is no scientific evidence to support thishypothesis. The third theory postulates an alteration oflaryngeal physiology due to carcinoma, which may in-crease intralaryngeal pressure, possibly as a result of fre-quent coughing, phonatory misuse or changes in laryn-geal neuromuscular mechanics, resulting in the enlarge-ment of the congenitally large saccule3,4,8.In our case laryngeal cancer spread to the medial la-ryngeal surface of the epiglottis, ventricular and vocalfolds, the deep structures of the larynx and both saccularnecks. Although it is thought that the tumour spreadinto the laryngoceles through the saccular necks, a sys-tematic hystological examination of whole organ sectionsshowed that the respiratory epithelium of saccular necksand internal parts of the laryngoceles were intact. Sub-mucosal spreading of cancer around the saccular neckson both sides caused stenosis which probably created aone-way valve mechanism allowing air to enter the sac-cule but not to exit. Prolonged forced coughing, difficultyin expectoration and phonatory alterations might be ad-ditional causative factors in enlargement of large sac-cule. Progression of the tumor completely obstructed thelaryngeal opening, leading to glandular secretion stagna-tion and formation of laryngomucoceles. These findingssuggest the posibility of the first theory of laryngoceleformation and explain that the saccular neck was not apathway of low resistance for the tumor to spread.AcknowledgementsThis paper has been presented at the 7th Congress ofthe European Laryngological Society, held in Barcelona,Spain, from 29th to 31st May 2008.R E F E R E N C E S1. MCDONALD SE, PINDER DK, SEN C, BIRCHALL MA, Eur ArchOtorhinolaryngol, 263 (2006) 237. — 2. GALLIVAN KH, GALLIVAN GJ,J Voice, 16 (2002) 258. — 3. AKBAS Y, UNAL M, PATA YS, Eur ArchOtorhinolaryngol, 261 (2004) 307. — 4. AKDOGAN O, IBRAHIM A, SEL-CUK A, DERE H, B-ENT 3, (2007) 209. — 5. PAPILA I, ACIOGLU E,KARAMAN E, AKMAN C, Eur Arch Otorhinolaryngol, 262 (2005) 473. —6. CASSANO L, LOMBARDO P, RAGONA RM, PASTORE A, Eur ArchOtorhinolaryngol, 257 (2000) 507. — 7. GREGOR RT, LOFTUS B, BALMAJM, COHEN P, HILGERS FJM, Ann Otol Rhinol Laryngol, 103 (1994)732. — 8. HARNEY M, PATIL N, WALSH R, BRENNAN P, WALSH M, JLaryngol Otol, 115 (2001) 590. — 9. GREENE FL, FRITZ AG, BALCHCM, HALLER DG, PAGE DL, FLEMING ID, MORROW M (Eds) AJCCCancer Staging Mannual (Springer-Verlag, New York, 2002). — 10.DUNCAN I, HEIN D, MARKS N, J Laryngol Otol, 113 (1999) 675. — 11.DURSUN G, OZGURSOY OB, BETON S, BATIKHAN H, OtolaryngolHead Neck Surg, 136 (2007) 211. — 12. ALVI A, WEISSMAN J, MYS-SIOREK D, NARULA S, MYERS EN, Am J Otolaryngol, 19 (1998) 251. —13. KORAMPALLI TS, BELLOSO A, HANS PS, IRION K, SHEPPARDIJ, J Laryngol Otol, 122 (2008) 353. — 14. MICHEAU C, LUBOINSKI B,LANCHI P, CACHIN Y, Laryngoscope, 88 (1978) 680. — 15. CARRAT X,FRANCOIS JM, CHARLES D, DEVARS F, TRAISSAC L, Ann Otol Rhi-nol Laryngol, 107 (1998) 703.M. RossoDepartment of Otorhinolaryngology and Head and Neck Surgery, Osijek University Hospital Center, Huttlerova 4,31000 Osijek, Croatiae-mail: rosso.marinela@kbo.hrM. Rosso et al.: Laryngoceles and Laryngeal Carcinoma, Coll. Antropol. 34 (2010) 2: 727–730729OBOSTRANE LARINGOKELE UDRU@ENE S PLANOCELULARNIM KARCINOMOM: PRIKAZSLU^AJA I OSVRT NA LITERATURUS A @ E T A KO istovremenom postojanju laringokela i karcinoma larinksa se jo{ uvijek raspravlja. Postoji nekoliko predlo`enihteorija o patofiziolo{kim odnosima izme|u ova dva entiteta. Predstavljamo slu~aj 66-godi{njeg mu{karca s bilateralnimlaringomukokelama i planocelularnim karcinomom larinksa. Patohistolo{ka analiza pokazala je submukozno {irenjekarcinoma oko vrata mukokele na obje strane {to je vjerojatno prouzro~ilo stenozu i stvorilo jednosmjerni ventil me-hanizam koji omogu}ava ulaz zraka u laringokelu, ali ne i izlaz. Progresija tumora potpuno zatvara laringealni otvor, a{to dovodi do stagnacije `ljezdane sekrecije i stvaranja laringomukokele.M. Rosso et al.: Laryngoceles and Laryngeal Carcinoma, Coll. Antropol. 34 (2010) 2: 727–730730

Bilateral Laryngoceles in Association with Squamos Cell Carcinoma: A Case Report and Literature Review

Bilateral Laryngoceles in Association with Squamos Cell Carcinoma: A Case Report and Literature Review

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