Laryngocele: Experience at a Tertiary Care Hospital of Eastern India *Santosh Kumar Swain, †Krishna Chandra Mallik, ‡Satyajit Mishra, and §Mahesh Chandra Sahu, *xBhubaneswar, yCuttuck, and zBurla, Odisha, India
Summary: Objectives. To study the details of clinical profile and management of laryngocele at a tertiary care teaching hospital of eastern India. Study Design. A prospective study. Case series of six patients of laryngocele. Methods. Six patients of laryngocele were examined at the Outpatient Department of ENT of Institute of Medical Sciences & SUM Hospital, Bhubaneswar, Odisha, India, between August 2010 and January 2014. The details of the patients, such as age, gender, occupations, clinical presentations, imaging modalities and treatment options, are discussed. Results. The common clinical presentations of laryngocele are hoarseness of voice and swelling in the neck. Sometimes, laryngocele patients are asymptomatic. The patients were in the range of 45–70 years old, among them five were males and one was female. The diagnosis was made clinically and radiologically. Among the six patients, five were treated by surgical approach. Conclusions. Laryngocele is an abnormal dilatation of the laryngeal saccule. It is a very rare clinical condition. Hoarseness of voice and swelling in the neck are common clinical presentations in laryngocele. Imaging studies are essential for making diagnosis, determining the type, localization, extent of laryngocele and for treatment. Surgery is the treatment of choice in laryngocele. Key Words: Laryngocele–Laryngeal saccule–Hoarseness–Laryngopyocele. INTRODUCTION Laryngocele is a rare air-filled cystic dilatation of the laryngeal saccule, which arises from the laryngeal ventricle.1 The laryngeal ventricle of Morgagni is normally a small elliptical recess situated between the false folds above and true folds below. The anterosuperior aspect of this recess ends in a blind pouch, which is called the appendix of the ventricle of Morgagni or saccule. Laryngoceles are of three types, namely internal, external, and mixed or combined according to their relationships with the thyrohyoid membrane.2 If laryngeal sac does not pierce the thyrohyoid membrane and remains within the larynx, it is called as internal laryngocele. When this sac pierces the thyrohyoid membrane and presents in the neck, it becomes an external laryngocele. Mixed or combined laryngocele has both internal and external components. Mixed laryngocele is otherwise called as the laryngocele that is situated both medial and lateral to the thyrohyoid membrane.3,4 Laryngocele is an unusual disease representing more than 20% of the true submucosal lesions of the larynx.5 Larry,6 a surgeon to Napoleon army, first described laryngocele in 1829. He described it as the compressible pouches related to the thyrohyoid membrane among the Muzzins, who shouted Korans from the roof of the mosques. In 1867, Virchow
Accepted for publication September 10, 2014. From the *Department of ENT, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, Kalinga Nagar, Bhubaneswar, Odisha, India; yDepartment of ENT, SCB Medical College, Cuttuck, Odisha, India; zDepartment of ENT, VSS Medical College, Burla, Odisha, India; and the xCentral Research Laboratory, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, Kalinga Nagar, Bhubaneswar, Odisha, India. Address correspondence and reprint requests to Santosh Kumar Swain, Department of ENT, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinga Nagar, Bhubaneswar 751003, Odisha, India. E-mail: [email protected] Journal of Voice, Vol. -, No. -, pp. 1-5 0892-1997/$36.00 Ó 2015 The Voice Foundation http://dx.doi.org/10.1016/j.jvoice.2014.09.013
named this affliction ‘‘Laryngocele ventricularis’’ to describe an air-filled dilatation of the ventricular saccule.7 Laryngoceles are usually unilateral and combined. They may present at any age group but are most commonly seen in the fifth to sixth decades.8 It is more frequent in males than in females at a ratio of 5:1.9 Although very rare, bilateral presentation of laryngocele was reported. There are different theories to explain the development of laryngocele, including a congenital saccule, weakness of laryngeal tissues, and increased intralaryngeal pressure, as seen in wind instrument players, glassblowers, street hawkers, and singers.2 In this study, we presented six cases of laryngocele with details of clinical profile and management (Table 1). METHOD Six patients of laryngoceles were examined at the Outpatient Department of ENT, Institute of Medical Sciences & SUM Hospital, Bubaneswar, Odisha, India, between August 2010 and January 2014. All patients were in the age group of 45–70 years. Out of six patients, five were males and one was female. Among them, five were treated surgically. Case 1 A 56-year-old male attended the outpatient department of ENT with complaints of hoarseness of voice since 1.5 years. He was a teacher by occupation with no history of breathing difficulty, dysphagia, and hemoptysis. He was not a diabetic and hypertensive patient. A small swelling was present on the left side of the neck since 6 months, which was insidious in onset and gradually progressive in nature. On physical examination, a single soft, nontender, and nonpulsatile swelling of about 6 3 5 cm was present in the front of left sternocleidomastoid muscle below the angle of the mandible. The valsalva maneuver
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TABLE 1. Details About the Patient Profile Cases Age (y) Case 1
56
Case 2
45
Case 3
70
Case 4
48
Case 5
65
Case 6
46
Sex Male
Occupation
Clinical Feature
Teacher
Radiological Finding
Hoarseness of voice, Plain X-ray and CT neck swelling scan showing an air-filled sac Male Salesman Neck swelling A large air-filled sac in the X-ray of the neck Male Preacher Hoarseness of voice, CT scan showing neck swelling combined laryngocele Female Housewife Hoarseness of voice Air-filled cavity in the X-ray of the neck Male Musical instrument Neck swelling Air-filled cavity in player plain X-ray Male Politician Neck swelling Air-filled cavity in X-ray and CT scan
produced enlargement of the neck swelling. Fiberoptic laryngoscopy showed normal vocal folds and a small bulging of left false fold with normal mucosa. Fine needle aspiration from the neck swelling yielded only air. X-ray of the soft tissue neck revealed an air-filled cavity on the left side. Considering the possibility of laryngocele, computed tomography (CT) scan was advised. CT scan demonstrated laryngocele in the left side of the neck with no evidence of any underlying intralaryngeal or cervical pathology (Figure 1). The patient was operated under general anesthesia (GA). Case 2 A 45-year-old male patient, salesman by occupation, presented to the outpatient department of ENT with complaints of painless swelling in the upper part of the right side of the neck. The patient did not have breathing difficulty, swallowing difficulty, voice change, or cough. On examination, the patient had a diffuse, nontender, and cystic swelling, which was approximately 4 3 4 cm in size and extended below the lower border of the mandible to the thyroid cartilage in the anterior triangle. The swelling was compressible and enlarged in size with valsalva maneuver and on coughing (Figure 2). Fiberoptic laryngoscopy showed a mild swelling at the level of the false fold. X-ray of the neck (anteroposterior and lateral views) showed a large air-filled sac in the right side of the neck (Figure 3A and B). The patient was operated under GA by external approach (Figure 4A and B). The postoperative period was uneventful. The patient was asymptomatic with no recurrence of the swelling in the subsequent follow-up at out-patient department. Case 3 A 70-year-old male had been suffering from hoarseness of voice since approximately 5 years. He was a preacher in occupation. On examination, he was found to have painless soft mass at the upper right side of the neck, about 4 cm in size, conducting vibration during speech, manually reducible, covered
Side of Neck Involved
Treatment
Left
Surgical excision
Right
Surgical excision
Right
Surgical excision
Right Left
Refused to do surgery Surgical excision
Right
Surgical excision
with skin. Fiberoptic laryngoscopy showed a supraglottic mass covered with normal mucosa. CT scan showed a large combined type of laryngocele. Surgical resection was performed by the external lateral cervical approach and an endoscopic route. Postoperative recovery was uneventful. Case 4 A 48-year-old lady came to the outpatient department of ENT with the complaints of hoarseness of voice since 1 year. She was a housewife with no history of breathing difficulty, dysphagia, and hemoptysis. ENT examinations were found normal. Chest X-ray was normal. On X-ray, soft tissue neck with lateral view revealed an air-filled cavity in the right side of the neck and diagnosis of laryngocele was made. She did not agree to do CT scan and also denied for surgery. Case 5 A 65-year-old man attended the outpatient department of ENT with complaints of swelling on the left side of the neck since 2 years. He was using musical instruments in the past. The swelling was more in size during blowing the nose or at the time of defecation. There was no history of voice change, stridor, or dysphagia. There was a history suggestive of constipation. Neck examination showed a soft cystic swelling on the left side of the neck at the level of hyoid, increased in size with valsalva maneuver. The swelling was nontender with diffuse margin and nonpulsatile. Fiberoptic laryngoscopy showed normal vocal folds. Other ENT examinations were normal. X-ray of soft tissue neck showed an air-filled cavity on the left side. CT scan was not done in this case. The patient was operated under GA. Case 6 A 46-year-old man came to the outpatient department of ENT with complaints of swelling of the neck in the right side, which was more prominent on coughing since 6 months. He was a
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Laryngocele: Experience at a Tertiary Care Hospital
3
confirmed by CT scan also. Patient was operated by external approach under GA.
FIGURE 1. CT scan of the neck with coronal cut (reconstructed view) showing a large laryngocele in the left side. local politician by occupation. There was no history of hoarseness of voice, dysphagia, or stridor. On examination, the neck mass was soft, nonpulsatile, and compressible mass situated at the level of hyoid on the right side of the neck. Indirect laryngoscopy showed normal findings in the larynx. X-ray of the neck showed an air-filled cavity in the right side, and it was
FIGURE 2. Neck swelling because of laryngocele (which is enlarged in size with valsalva maneuver).
DISCUSSION Laryngocele is a rare and benign dilatation of laryngeal saccule, which may extend internally into the airway or externally through the thyrohyoid membrane. The laryngeal ventricle was first described by Gallen (1822) and Morgagni (1841).10 The laryngeal ventricle is normally a small elliptical recess located between the false folds above and true folds below. The anterosuperior aspect of this recess ends in a blind pouch, called the saccule. Laryngocele is an extremely rare condition. The incidence of laryngocele is one in 2.5 million people per year.11 It has different synonyms like sacculus laryngis, appendix ventriculi laryngis, laryngeal pouch, or Hilton’s sac.12 Approximately, two of three laryngoceles are unilateral, and they may be external (30%), internal (20%), or combined (50%).9 A combined laryngocele and an external laryngocele with only an external manifestation have a frequency of approximately 60%, whereas an internal laryngocele has approximately a 40% frequency.13 In the present study, all are unilateral and of external type, except case 3, which is of combined type and also unilateral. The etiology of laryngocele is unknown and unclear.14 There are several explanations for formations of laryngocele. In neonates, they are thought to be remnants of the lateral air sacs seen in other higher primates. In adults, they may represent as congenital enlargement of the saccule or an acquired lesion association with increased intralaryngeal pressure. The association of laryngocele with laryngeal carcinoma and occupations involving long periods of forced expiration support this notion.15 The cause of laryngocele is considered as multifactorial and is associated with the increase in transglottic pressure, a factor usually present in glassblowers or musicians who play wind instruments.16 Sometimes, laryngeal cancer is associated with laryngocele. The pathogenesis for cancer and laryngocele is chronic intralaryngeal pressure. In some other study, the association between laryngocele and cancer is coincidental.17 The association between laryngocele with amyloidosis and ankylosing spondylitis is reported.18 Laryngocele has been seen as an acquired condition, after excision of submandibular gland for chronic sialadenitis and salivary calculus19 and tracheostomy.20 The histopathological examination is important to rule out any associated lesion in the laryngocele. The normal macroscopical picture of laryngocele is a three-dimensional sac-like structure with outside fibrous wall. Microscopically, the inner lining of laryngocele is covered with ciliated pseudostratified columnar epithelium with varied number of goblet cells on a thin basal membrane.21 The commonest clinical features are hoarseness of voice and a swelling in the neck with almost equal frequency. But, it may also present with dysphagia, pain, stridor, and snoring.10 Sometimes, it may be asymptomatic. Cases 1, 3, and 4 complain hoarseness of voice, whereas cases 2, 5, and 6 present with neck swelling. A laryngocele may be infected, which then is
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FIGURE 3. A. X-ray of the neck (anteroposterior view) showing an air-filled sac. B. X-ray of the neck (lateral view) showing an air-filled sac. called a laryngopyocele. Laryngopyocele is seen in 8% of cases.9 Stell and Maran in a review of 139 cases showed that peak incidence is in the sixth decade, and two of three laryngoceles are unilateral; also they may be combined, external, or internal with roughly equal frequency. Laryngocele is more common among males as in our study, but there is no definite explanation for male predominance. Previously, laryngocele has been diagnosed by plain X-ray, xerography, plain film tomography, and contrast laryngography. X-ray of the neck with lateral view may show a wellcircumscribed air collection in laryngocele. It is difficult to differentiating laryngocele from any soft tissue mass in neck when laryngocele is filled with fluid rather than air. Contrast laryngography can show the mass effect of internal laryngoceles, and fluoroscopic visualization of the larynx may help in the diagnosis of complicated laryngoceles.22 Many suggest that CT scan is the gold standard, with magnetic resonance imaging (MRI) playing an adjuvant role in the diagnosis of laryngoceles.23 CT scan has proved to be the most effective method for defining the spatial relationship between the laryngocele, laryngeal structure, and extralaryngeal soft tissue, in differentiating the laryngocele from other cystic mass and the coexistence of a laryngeal cancer.24 Ultrasonography of the neck can be done for initial evaluation of neck mass, mainly for differentiating the nature of lesion as well as defining its contents and location.25 An air-filled laryngocele typically has low signal cystic dilatation of the laryngeal ventricle on MRI.26 MRI should be the imaging of choice if there is a doubt of a concurrent laryngeal tumor with a laryngocele.27 The paralaryngeal space, thyrohyoid membrane, true and false folds are
clearly seen in MRI. In our cases, the radiological diagnosis was done with plain X-ray of the neck and CT scan. Plain X-ray of soft tissue neck with valsalva is a worthwhile screening test for puzzling hoarseness although more detailed information may be gained from laryngeal CT scan. The differential diagnosis of laryngocele includes saccular cyst, branchial cyst, neck abscess, and lymphadenopathy.8,27,28 The treatment option of the laryngocele is surgery. Surgery by external approach is done for large or external laryngocele, whereas endoscopic approach is useful for small or internal laryngocele. External surgical approach gives an excellent exposure during the dissection of the plane between the neck of laryngocele and surrounding paraglottic tissue. This approach also offers less recurrence rate, minimal morbidity, and negligible complications.4,11,29 Endoscopic excision with laser is the treatment of choice in patients with internal laryngocele. It needs less operative time and does minimal damage to endolarynx and vocal folds. The voice quality and swallowing functions can be preserved.11,29 The mixed type of laryngocele can be completely excised via an external approach, and however, a combined external and endoscopic laser approach ensures complete removal.11,29,30 In the present study, we operated all cases (case 4 refused surgery) with only external approach, whereas case 3 was approached through both external and endoscopic approaches.
CONCLUSION Laryngocele is a rare clinical entity. Although it is a benign disease, laryngocele can be a potential cause for respiratory
FIGURE 4. A. Laryngocele seen in operating field. B. Laryngocele removed in toto.
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Laryngocele: Experience at a Tertiary Care Hospital
obstruction that may threaten the patient’s life. Sometimes, the diagnosis is difficult. Imaging studies are essential for making diagnosis, determining the type, localization, extent of laryngocele, and for treatment. If the patient has a hoarseness of voice of longer duration with normal vocal folds, plain X-ray of the neck (anteroposterior view) with and without valsalva maneuver to diagnose smaller laryngocele should be carried out. It is mandatory in patients presenting with a soft neck mass, even if not a wind instrument player or a glassblower, to exclude the possibility of a laryngocele. Recurrences are unusual after surgery. REFERENCES 1. Pruszewicz A, Obrebowski A, Maciejewska B. Bilateral internal laryngocele with open nasality—report of a case. Otolaryngol Pol. 2006;60: 935–938. 2. Verret DJ, DeFatta RJ, Sinard R. Combined laryngocele. Ann Otol Rhinol Laryngol. 2004;113:594–596. 3. Glanz I, Grunebaum M. The radiographic appearances of ventricular laryngocele. Isr J Med Sci. 1971;7:1155–1159. 4. Thome R, Thome DC, De La Cortina RAC. Lateral thyrotomy approach on the paraglottic space for laryngocele resection. Laryngoscope. 2000;110: 447–450. 5. Saleh EM, Mancuso AA, Stringer SP. CT of submucosal and occult laryngeal masses. J Comput Assist Tomogr. 1992;16:87–93. 6. Larry DJ. Laryngocele: a case report and review. J Otolaryngol. 1983;12: 6389–6392. 7. Barman D, Pakira B, Majumder P, Ghosh SK, Banka A, Dutta S. Laryngocele: our experience. Indian J Otolaryngol Head Neck Surg. 2008;60: 210–213. 8. Amin M, Maran AG. The aetiology of laryngocele. Clin Otolaryngol Allied Sci. 1988;13:267–272. 9. Stell PM, Maran AG. Laryngocele. J Laryngol Otol. 1975;89:915–924. 10. Arjun D. Laryngocele (report of two cases). Indian J Otolaryngol Head Neck Surg. 1992;1:29–31. 11. Matino SE, Martinez VV, Leon VX, Quer AM, Burgues VJ, De JM. Laryngocele: clinical and therapeutic study of 60 cases. Acta Otorrinolaringol Esp. 1995;46:279–286. 12. Gallivan GJ, Gallivan KH. Bilateral mixed laryngoceles: simultaneous strobovideolaryngoscopy and external video examination. J Voice. 2002;16: 258–266.
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13. Upile T, Jerjes W, Sipaul F, et al. Laryngocele: a rare complication of surgical tracheostomy. BMC Surg. 2006;6:14. 14. Erdogmus B, Yazici B, Ozturk O, Ataoglu S, Yazici S. Laryngocele in association with ankylosing spondylitis. Wien Klin Wochenschr. 2005; 117:718–720. 15. Noris CW. Pharyngoceles of the hypopharynx. Laryngoscope. 1979;89: 1788–1807. 16. Sobol SM, Bailey SB. Evaluation and surgical management of tumors of the neck: benign tumors. In: Thawley SE, Panje WR, Batsakis JG, Lindberg RD, eds. Comprehensive Management of Head and Neck Tumors. Philadelphia, PA: WB Saunders; 1999:1435–1439. 17. Canalis RF. Observation on the simultaneous occurrence of laryngocele and cancer. J Otolaryngol. 1976;5:207–212. 18. Ayden O, Unstundag E, Iseri M, et al. Laryngeal amyloidosis with laryngocele. J Laryngol Otol. 1999;113:278–281. 19. Abu-Khalat M, Tarawneh M. Laryngocele: an acquired condition. J Laryngol Otol. 1981;95:425–429. 20. Tahwinder U. Laryngocele: a rare complication of surgical tracheostomy. BMC Surg. 2006;6:14. 21. Pennings RJE, Van den Hoogen FJ, Marres HAM. Giant laryngoceles: a cause of upper airway obstruction. Eur Arch Otorhinolaryngol. 2001; 258:137–140. 22. Johnson TH Jr, Fioranelli RJ. Laryngeal cyst (filled laryngocele): laryngographic diagnosis. Radiology. 1969;93:875–877. 23. Akbas Y, Unal M, Pata YS. Asymptomatic bilateral mixed type laryngocele and laryngeal carcinoma. Eur Arch Otorhinolaryngol. 2004;261:307–309. 24. Lancella A, Abbate G, Dosdegani R. Mixed laryngocele: a case report and review of the literature. Acta Otorhinolaryngol Ital. 2007;27:255–257. 25. Youssefzadeh S, Steiner E, Turetschek K, Gritzmann N, Kursten R, Franz P. The sonography of laryngeal cysts. Rofo. 1993;159:38–42. 26. Alvi A, Weissman J, Myssiorek D, Narula S, Myers EN. Computed tomography and magnetic resonance imaging characteristics of laryngocele and its variant. Am J Otolaryngol. 1998;19:251–256. 27. Holinger LD, Barnes DR, Smid LJ, Holinger PH. Laryngocele and saccular cysts. Ann Otol Rhinol Laryngol. 1978;87(5 pt 1):675–685. 28. Griffin J, Ramadan HH, Wetmore SJ. Laryngocele: a case of stridor and airway obstruction. Otolaryngol Head Neck Surg. 1993;108:760–762. 29. Dursun G, Ozgursoy OB, Beton S, Batikhan H. Current diagnosis and treatment of laryngocele in adults. Otolaryngol Head Neck Surg. 2007;36: 211–215. 30. Ettema SL, Carothers DG, Hofmann HT. Laryngocele resection by combined external and endoscopic laser approach. Ann Otol Rhinol Laryngol. 2003;112:361–364.
Summary: Objectives. To study the details of clinical profile and management of laryngocele at a tertiary care teaching hospital of eastern India. Study Design. A prospective study. Case series of six patients of laryngocele. Methods. Six patients of laryngocele were examined at the Outpatient Department of ENT of Institute of Medical Sciences & SUM Hospital, Bhubaneswar, Odisha, India, between August 2010 and January 2014. The details of the patients, such as age, gender, occupations, clinical presentations, imaging modalities and treatment options, are discussed. Results. The common clinical presentations of laryngocele are hoarseness of voice and swelling in the neck. Sometimes, laryngocele patients are asymptomatic. The patients were in the range of 45–70 years old, among them five were males and one was female. The diagnosis was made clinically and radiologically. Among the six patients, five were treated by surgical approach. Conclusions. Laryngocele is an abnormal dilatation of the laryngeal saccule. It is a very rare clinical condition. Hoarseness of voice and swelling in the neck are common clinical presentations in laryngocele. Imaging studies are essential for making diagnosis, determining the type, localization, extent of laryngocele and for treatment. Surgery is the treatment of choice in laryngocele. Key Words: Laryngocele–Laryngeal saccule–Hoarseness–Laryngopyocele. INTRODUCTION Laryngocele is a rare air-filled cystic dilatation of the laryngeal saccule, which arises from the laryngeal ventricle.1 The laryngeal ventricle of Morgagni is normally a small elliptical recess situated between the false folds above and true folds below. The anterosuperior aspect of this recess ends in a blind pouch, which is called the appendix of the ventricle of Morgagni or saccule. Laryngoceles are of three types, namely internal, external, and mixed or combined according to their relationships with the thyrohyoid membrane.2 If laryngeal sac does not pierce the thyrohyoid membrane and remains within the larynx, it is called as internal laryngocele. When this sac pierces the thyrohyoid membrane and presents in the neck, it becomes an external laryngocele. Mixed or combined laryngocele has both internal and external components. Mixed laryngocele is otherwise called as the laryngocele that is situated both medial and lateral to the thyrohyoid membrane.3,4 Laryngocele is an unusual disease representing more than 20% of the true submucosal lesions of the larynx.5 Larry,6 a surgeon to Napoleon army, first described laryngocele in 1829. He described it as the compressible pouches related to the thyrohyoid membrane among the Muzzins, who shouted Korans from the roof of the mosques. In 1867, Virchow
Accepted for publication September 10, 2014. From the *Department of ENT, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, Kalinga Nagar, Bhubaneswar, Odisha, India; yDepartment of ENT, SCB Medical College, Cuttuck, Odisha, India; zDepartment of ENT, VSS Medical College, Burla, Odisha, India; and the xCentral Research Laboratory, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, Kalinga Nagar, Bhubaneswar, Odisha, India. Address correspondence and reprint requests to Santosh Kumar Swain, Department of ENT, IMS & SUM Hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinga Nagar, Bhubaneswar 751003, Odisha, India. E-mail: [email protected] Journal of Voice, Vol. -, No. -, pp. 1-5 0892-1997/$36.00 Ó 2015 The Voice Foundation http://dx.doi.org/10.1016/j.jvoice.2014.09.013
named this affliction ‘‘Laryngocele ventricularis’’ to describe an air-filled dilatation of the ventricular saccule.7 Laryngoceles are usually unilateral and combined. They may present at any age group but are most commonly seen in the fifth to sixth decades.8 It is more frequent in males than in females at a ratio of 5:1.9 Although very rare, bilateral presentation of laryngocele was reported. There are different theories to explain the development of laryngocele, including a congenital saccule, weakness of laryngeal tissues, and increased intralaryngeal pressure, as seen in wind instrument players, glassblowers, street hawkers, and singers.2 In this study, we presented six cases of laryngocele with details of clinical profile and management (Table 1). METHOD Six patients of laryngoceles were examined at the Outpatient Department of ENT, Institute of Medical Sciences & SUM Hospital, Bubaneswar, Odisha, India, between August 2010 and January 2014. All patients were in the age group of 45–70 years. Out of six patients, five were males and one was female. Among them, five were treated surgically. Case 1 A 56-year-old male attended the outpatient department of ENT with complaints of hoarseness of voice since 1.5 years. He was a teacher by occupation with no history of breathing difficulty, dysphagia, and hemoptysis. He was not a diabetic and hypertensive patient. A small swelling was present on the left side of the neck since 6 months, which was insidious in onset and gradually progressive in nature. On physical examination, a single soft, nontender, and nonpulsatile swelling of about 6 3 5 cm was present in the front of left sternocleidomastoid muscle below the angle of the mandible. The valsalva maneuver
2
Journal of Voice, Vol. -, No. -, 2015
TABLE 1. Details About the Patient Profile Cases Age (y) Case 1
56
Case 2
45
Case 3
70
Case 4
48
Case 5
65
Case 6
46
Sex Male
Occupation
Clinical Feature
Teacher
Radiological Finding
Hoarseness of voice, Plain X-ray and CT neck swelling scan showing an air-filled sac Male Salesman Neck swelling A large air-filled sac in the X-ray of the neck Male Preacher Hoarseness of voice, CT scan showing neck swelling combined laryngocele Female Housewife Hoarseness of voice Air-filled cavity in the X-ray of the neck Male Musical instrument Neck swelling Air-filled cavity in player plain X-ray Male Politician Neck swelling Air-filled cavity in X-ray and CT scan
produced enlargement of the neck swelling. Fiberoptic laryngoscopy showed normal vocal folds and a small bulging of left false fold with normal mucosa. Fine needle aspiration from the neck swelling yielded only air. X-ray of the soft tissue neck revealed an air-filled cavity on the left side. Considering the possibility of laryngocele, computed tomography (CT) scan was advised. CT scan demonstrated laryngocele in the left side of the neck with no evidence of any underlying intralaryngeal or cervical pathology (Figure 1). The patient was operated under general anesthesia (GA). Case 2 A 45-year-old male patient, salesman by occupation, presented to the outpatient department of ENT with complaints of painless swelling in the upper part of the right side of the neck. The patient did not have breathing difficulty, swallowing difficulty, voice change, or cough. On examination, the patient had a diffuse, nontender, and cystic swelling, which was approximately 4 3 4 cm in size and extended below the lower border of the mandible to the thyroid cartilage in the anterior triangle. The swelling was compressible and enlarged in size with valsalva maneuver and on coughing (Figure 2). Fiberoptic laryngoscopy showed a mild swelling at the level of the false fold. X-ray of the neck (anteroposterior and lateral views) showed a large air-filled sac in the right side of the neck (Figure 3A and B). The patient was operated under GA by external approach (Figure 4A and B). The postoperative period was uneventful. The patient was asymptomatic with no recurrence of the swelling in the subsequent follow-up at out-patient department. Case 3 A 70-year-old male had been suffering from hoarseness of voice since approximately 5 years. He was a preacher in occupation. On examination, he was found to have painless soft mass at the upper right side of the neck, about 4 cm in size, conducting vibration during speech, manually reducible, covered
Side of Neck Involved
Treatment
Left
Surgical excision
Right
Surgical excision
Right
Surgical excision
Right Left
Refused to do surgery Surgical excision
Right
Surgical excision
with skin. Fiberoptic laryngoscopy showed a supraglottic mass covered with normal mucosa. CT scan showed a large combined type of laryngocele. Surgical resection was performed by the external lateral cervical approach and an endoscopic route. Postoperative recovery was uneventful. Case 4 A 48-year-old lady came to the outpatient department of ENT with the complaints of hoarseness of voice since 1 year. She was a housewife with no history of breathing difficulty, dysphagia, and hemoptysis. ENT examinations were found normal. Chest X-ray was normal. On X-ray, soft tissue neck with lateral view revealed an air-filled cavity in the right side of the neck and diagnosis of laryngocele was made. She did not agree to do CT scan and also denied for surgery. Case 5 A 65-year-old man attended the outpatient department of ENT with complaints of swelling on the left side of the neck since 2 years. He was using musical instruments in the past. The swelling was more in size during blowing the nose or at the time of defecation. There was no history of voice change, stridor, or dysphagia. There was a history suggestive of constipation. Neck examination showed a soft cystic swelling on the left side of the neck at the level of hyoid, increased in size with valsalva maneuver. The swelling was nontender with diffuse margin and nonpulsatile. Fiberoptic laryngoscopy showed normal vocal folds. Other ENT examinations were normal. X-ray of soft tissue neck showed an air-filled cavity on the left side. CT scan was not done in this case. The patient was operated under GA. Case 6 A 46-year-old man came to the outpatient department of ENT with complaints of swelling of the neck in the right side, which was more prominent on coughing since 6 months. He was a
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Laryngocele: Experience at a Tertiary Care Hospital
3
confirmed by CT scan also. Patient was operated by external approach under GA.
FIGURE 1. CT scan of the neck with coronal cut (reconstructed view) showing a large laryngocele in the left side. local politician by occupation. There was no history of hoarseness of voice, dysphagia, or stridor. On examination, the neck mass was soft, nonpulsatile, and compressible mass situated at the level of hyoid on the right side of the neck. Indirect laryngoscopy showed normal findings in the larynx. X-ray of the neck showed an air-filled cavity in the right side, and it was
FIGURE 2. Neck swelling because of laryngocele (which is enlarged in size with valsalva maneuver).
DISCUSSION Laryngocele is a rare and benign dilatation of laryngeal saccule, which may extend internally into the airway or externally through the thyrohyoid membrane. The laryngeal ventricle was first described by Gallen (1822) and Morgagni (1841).10 The laryngeal ventricle is normally a small elliptical recess located between the false folds above and true folds below. The anterosuperior aspect of this recess ends in a blind pouch, called the saccule. Laryngocele is an extremely rare condition. The incidence of laryngocele is one in 2.5 million people per year.11 It has different synonyms like sacculus laryngis, appendix ventriculi laryngis, laryngeal pouch, or Hilton’s sac.12 Approximately, two of three laryngoceles are unilateral, and they may be external (30%), internal (20%), or combined (50%).9 A combined laryngocele and an external laryngocele with only an external manifestation have a frequency of approximately 60%, whereas an internal laryngocele has approximately a 40% frequency.13 In the present study, all are unilateral and of external type, except case 3, which is of combined type and also unilateral. The etiology of laryngocele is unknown and unclear.14 There are several explanations for formations of laryngocele. In neonates, they are thought to be remnants of the lateral air sacs seen in other higher primates. In adults, they may represent as congenital enlargement of the saccule or an acquired lesion association with increased intralaryngeal pressure. The association of laryngocele with laryngeal carcinoma and occupations involving long periods of forced expiration support this notion.15 The cause of laryngocele is considered as multifactorial and is associated with the increase in transglottic pressure, a factor usually present in glassblowers or musicians who play wind instruments.16 Sometimes, laryngeal cancer is associated with laryngocele. The pathogenesis for cancer and laryngocele is chronic intralaryngeal pressure. In some other study, the association between laryngocele and cancer is coincidental.17 The association between laryngocele with amyloidosis and ankylosing spondylitis is reported.18 Laryngocele has been seen as an acquired condition, after excision of submandibular gland for chronic sialadenitis and salivary calculus19 and tracheostomy.20 The histopathological examination is important to rule out any associated lesion in the laryngocele. The normal macroscopical picture of laryngocele is a three-dimensional sac-like structure with outside fibrous wall. Microscopically, the inner lining of laryngocele is covered with ciliated pseudostratified columnar epithelium with varied number of goblet cells on a thin basal membrane.21 The commonest clinical features are hoarseness of voice and a swelling in the neck with almost equal frequency. But, it may also present with dysphagia, pain, stridor, and snoring.10 Sometimes, it may be asymptomatic. Cases 1, 3, and 4 complain hoarseness of voice, whereas cases 2, 5, and 6 present with neck swelling. A laryngocele may be infected, which then is
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Journal of Voice, Vol. -, No. -, 2015
FIGURE 3. A. X-ray of the neck (anteroposterior view) showing an air-filled sac. B. X-ray of the neck (lateral view) showing an air-filled sac. called a laryngopyocele. Laryngopyocele is seen in 8% of cases.9 Stell and Maran in a review of 139 cases showed that peak incidence is in the sixth decade, and two of three laryngoceles are unilateral; also they may be combined, external, or internal with roughly equal frequency. Laryngocele is more common among males as in our study, but there is no definite explanation for male predominance. Previously, laryngocele has been diagnosed by plain X-ray, xerography, plain film tomography, and contrast laryngography. X-ray of the neck with lateral view may show a wellcircumscribed air collection in laryngocele. It is difficult to differentiating laryngocele from any soft tissue mass in neck when laryngocele is filled with fluid rather than air. Contrast laryngography can show the mass effect of internal laryngoceles, and fluoroscopic visualization of the larynx may help in the diagnosis of complicated laryngoceles.22 Many suggest that CT scan is the gold standard, with magnetic resonance imaging (MRI) playing an adjuvant role in the diagnosis of laryngoceles.23 CT scan has proved to be the most effective method for defining the spatial relationship between the laryngocele, laryngeal structure, and extralaryngeal soft tissue, in differentiating the laryngocele from other cystic mass and the coexistence of a laryngeal cancer.24 Ultrasonography of the neck can be done for initial evaluation of neck mass, mainly for differentiating the nature of lesion as well as defining its contents and location.25 An air-filled laryngocele typically has low signal cystic dilatation of the laryngeal ventricle on MRI.26 MRI should be the imaging of choice if there is a doubt of a concurrent laryngeal tumor with a laryngocele.27 The paralaryngeal space, thyrohyoid membrane, true and false folds are
clearly seen in MRI. In our cases, the radiological diagnosis was done with plain X-ray of the neck and CT scan. Plain X-ray of soft tissue neck with valsalva is a worthwhile screening test for puzzling hoarseness although more detailed information may be gained from laryngeal CT scan. The differential diagnosis of laryngocele includes saccular cyst, branchial cyst, neck abscess, and lymphadenopathy.8,27,28 The treatment option of the laryngocele is surgery. Surgery by external approach is done for large or external laryngocele, whereas endoscopic approach is useful for small or internal laryngocele. External surgical approach gives an excellent exposure during the dissection of the plane between the neck of laryngocele and surrounding paraglottic tissue. This approach also offers less recurrence rate, minimal morbidity, and negligible complications.4,11,29 Endoscopic excision with laser is the treatment of choice in patients with internal laryngocele. It needs less operative time and does minimal damage to endolarynx and vocal folds. The voice quality and swallowing functions can be preserved.11,29 The mixed type of laryngocele can be completely excised via an external approach, and however, a combined external and endoscopic laser approach ensures complete removal.11,29,30 In the present study, we operated all cases (case 4 refused surgery) with only external approach, whereas case 3 was approached through both external and endoscopic approaches.
CONCLUSION Laryngocele is a rare clinical entity. Although it is a benign disease, laryngocele can be a potential cause for respiratory
FIGURE 4. A. Laryngocele seen in operating field. B. Laryngocele removed in toto.
Santosh Kumar Swain, et al
Laryngocele: Experience at a Tertiary Care Hospital
obstruction that may threaten the patient’s life. Sometimes, the diagnosis is difficult. Imaging studies are essential for making diagnosis, determining the type, localization, extent of laryngocele, and for treatment. If the patient has a hoarseness of voice of longer duration with normal vocal folds, plain X-ray of the neck (anteroposterior view) with and without valsalva maneuver to diagnose smaller laryngocele should be carried out. It is mandatory in patients presenting with a soft neck mass, even if not a wind instrument player or a glassblower, to exclude the possibility of a laryngocele. Recurrences are unusual after surgery. REFERENCES 1. Pruszewicz A, Obrebowski A, Maciejewska B. Bilateral internal laryngocele with open nasality—report of a case. Otolaryngol Pol. 2006;60: 935–938. 2. Verret DJ, DeFatta RJ, Sinard R. Combined laryngocele. Ann Otol Rhinol Laryngol. 2004;113:594–596. 3. Glanz I, Grunebaum M. The radiographic appearances of ventricular laryngocele. Isr J Med Sci. 1971;7:1155–1159. 4. Thome R, Thome DC, De La Cortina RAC. Lateral thyrotomy approach on the paraglottic space for laryngocele resection. Laryngoscope. 2000;110: 447–450. 5. Saleh EM, Mancuso AA, Stringer SP. CT of submucosal and occult laryngeal masses. J Comput Assist Tomogr. 1992;16:87–93. 6. Larry DJ. Laryngocele: a case report and review. J Otolaryngol. 1983;12: 6389–6392. 7. Barman D, Pakira B, Majumder P, Ghosh SK, Banka A, Dutta S. Laryngocele: our experience. Indian J Otolaryngol Head Neck Surg. 2008;60: 210–213. 8. Amin M, Maran AG. The aetiology of laryngocele. Clin Otolaryngol Allied Sci. 1988;13:267–272. 9. Stell PM, Maran AG. Laryngocele. J Laryngol Otol. 1975;89:915–924. 10. Arjun D. Laryngocele (report of two cases). Indian J Otolaryngol Head Neck Surg. 1992;1:29–31. 11. Matino SE, Martinez VV, Leon VX, Quer AM, Burgues VJ, De JM. Laryngocele: clinical and therapeutic study of 60 cases. Acta Otorrinolaringol Esp. 1995;46:279–286. 12. Gallivan GJ, Gallivan KH. Bilateral mixed laryngoceles: simultaneous strobovideolaryngoscopy and external video examination. J Voice. 2002;16: 258–266.
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