Portuguese Version

Year:  2002  Vol. 68   Ed. 5 - (10º)

Artigo Original

Pages: 663 to 666

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Supraglottic cysts of the larynx: ethiologic, clinical and therapeutical aspects

Author(s): Rafael B. Cahali 1,
Silvia A. Zimbres 2,
Domingos H. Tsuji 3,
Michel B. Cahali 4,
Luiz U. Sennes 5

Keywords: laryngeal cysts, classification, respiratory obstruction.

Abstract:
Aim: In this study, the authors describe their experience on diagnosis, etiology, treatment and follow-up of patients with laryngeal cysts from the otolaryngology department of the Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo. Study design: Clinical randomized. Material and method: Trying to emphasize the incidence, the clinical presentation, treatment and results. Nineteen patients were analyzed, showing that the initial clinical presentation of these cysts can represent since a common complaint until severe respiratory obstruction. Conclusion: Therefore, it is necessary the knowledge about this pathology to diagnose properly and provide a better prognosis to the patient.

Introduction

Larynx cysts are a group of rare benign injuries that may cause significant respiratory obstruction and even death if not properly managed1, 2,3. They are difficult to be diagnosed, since test findings are generally unspecific or patients are asymptomatic. Therefore, patients with larynx stridor or dyspnea should be further investigated to prevent complications. 1. It is worth mentioning that these patients may have common complaints to the otorhinolaryngologist such as, dysphonia, dysphagia, globus sensation or even occasional findings that do not demand specific treatment.

In 1970, DeSanto4 classified larynx cysts in ductile and saccular according to its location in the larynx and to the histological examination. Therefore, saccular cysts are located along the saccular plan deeply into the false vocal folds, aryepiglottic fold and anterior ventricle whereas ductal cysts are formed by the distension of obstructed glandular ducts. This classification is widely accepted today and will be used in this study.

Objective

The objective of this study is to report our experience with symptomatic laryngeal cyst patients that have undergone surgery, emphasizing cysts etiological, clinical and topographic characteristics as well as therapeutic management and outcomes.

MATERIAL AND METHOD

This retrospective paper studied 19 patients with treated supraglottic larynx cysts that have undergone surgery at the Clinical Otorhinolaryngology Department of the Clinical Hospital, Medical School of the University of Sao Paulo from January 1990 to January 2000.

The symptoms were assigned to presence of cysts after ruling out and treating all possible symptom causes such as gastroesophageal reflux, chronic pharyngitis and tonsillitis.

In all cases diagnosis was made by laryngoscopy and confirmed by suspension laryngoscopy in laryngeal microsurgery and in the anatomical pathological exam.

Age, gender, physical examination, cyst location, treatment used and outcomes were analyzed and data were and related.

Graph 1. Distribution according to gender



Graph 2. Distribution according to age



Graph 3. Symptoms Identified



Graph 4. Duration of Symptoms

RESULTS

In 19 patients studied, 12 were female and 7 were male (Graph 1). The age range was 2 to 83 years (mean age 36 and median age = 34.9) (Graph 2).

Globus or foreign body sensation was the most common complaint (47.3%). Dysphagia, dysphonia and dyspnea were reported in 5 patients (26.3%), not necessarily the same patients. Urgency tracheostomy was required in three patients with dyspnea. Two patients (10.5%) complained of cervical pain and one patient had a visible mass in oroscopy with laryngeal stridor (Graph 3). Ten patients (52.5%) reported more than one of these symptoms.

The duration of symptoms ranged from 2 months to 12 years, however most of the patients (11 patients = 57.8%) reported 1 year or less of clinical history. Only 3 patients (15.7%) had the symptoms for more than three years (Graph 4).

Most of them were related to the epiglottis; 7 were located in its lingual aspect and one in the laryngeal aspect. In 6 cases it was located in the vallecula, with three cysts located in the ventricular band and 1 located in the posterior ventricular portion, and another one located together with the right arytenoids (Chart 1).

According to the classification of DeSanto et al. 15 ductal cysts (78.8%) and 3 saccular cysts (15.7%) were found, the latter included two internal laryngoceles. One of the ductal cysts of the epiglottis corresponded to a traumatic cyst due to foreign body. Moreover, one of the cysts found in the vallecula was diagnosed as a lymphangioma in the anatomical pathological exam, and did not fit in this classification.

Eighteen out of nineteen cysts were sessile (94.7%) and only 1 was pediculated, forming a valve to glottis (right arytenoid cyst).

All cases have undergone surgical treatment, 18 patients (94.7%) were treated through endoscopy and one was followed by lingual tonsillectomy for exeresis of a cyst in the lingual aspect of the epiglottis. Only one case had the surgery performed by cervicotomy for comprehensive internal laryngocele removal. In this surgery tracheotomy was carried out with median cervical incision, exposure of the left paraglottic spaces with perichondral resection and removal of the upper portion of the left thyroid cartilage. Next, laryngocele dissection was carried out until its origin in the upper portion of the left ventricle.

Postoperative follow-up ranged from 1 to 8 years (mean was 4.4 years). Two patients had recurrence (10.5%). One occurred in a patient with lymphangioma 5 years after surgery. The other recurrence happened with a patient with ductal cyst in the lingual aspect of the epiglottis, he had three recurrences within one year, after the third surgery he was followed up for 5 years with no recurrences. Nine patients (47.3%) did not have any recurrences in more than 5 years of follow-up.

Chart 1. Relation between location of laryngeal cysts and classification.

Discussion

Laryngeal cysts are uncommon and account for 5% of the benign injuries of the larynx. Several hypotheses were discussed in order to explain pathogenesis of these lesions. In 1929, Imperatori advocated they were branchial remains, and in 1943, Dinolt raised the possibility of saccular sequestration. Eventually, DeSanto et. al. in 1970 proposed the classification in ductal and saccular cysts.

In his study, DeSanto did not find gender preference of cysts. In our study we found a higher female prevalence (12 cases) accounting for 63.2% of the studied population.

Moreover, a higher prevalence of laryngeal cysts was found in the age range of 20 to 40 years (42.5% of the cases), and it did not match with other authors, since in their studies this lesion was likely to happen within the 6th decade of life. Only 36.5% of patients were above 40 years old.

In respect to clinical picture, Lam5 and Albert6 described most of the lesions as symptomatic. In patients who had symptoms, they varied according to age, size and location of cysts and were as follows: progressive cough, dysphagia, hoarseness, stridor, dyspnea, globus sensation and pain2,4,5,7. In our study none of the patients were asymptomatic upon diagnosis. The most common symptom found was globus sensation (47.3%), followed by dysphagia, dysphonia and dyspnea, and were reported by the same number of patients (26.3% of the cases). It is worth mentioning that most of the patients (52.5%) had more than one symptom at diagnosis.

Most of the patients (57.9%) had experienced symptoms for a maximum period of 1 year when they came to the service. Only 15.8% of the patients had symptoms for more than 3 years, showing that in general laryngeal cyst complaints are valued by the patients and make them search for medical care.

Several authors reported cases of significant breathing obstruction that may be caused by these cysts1,3,8. Mitchell1 et al. in his study found the need for tracheostomy in 20% of their patients. Three of our patients (60% of patients with dyspnea) have undergone tracheostomy, that is, 15% of all cases. Mortality rate due to laryngeal cysts in children is 40%, according to LaBagnaro9; data on mortality rate in adults were not found in the literature.

In terms of location, laryngeal cysts, Lam5, Albert6 and DeSanto4 stated that cysts are found in the epiglottis, more specifically in its lingual aspect1,5. Besides the epiglottis, the vocal folds are actually referred by Lam as the other location of high prevalence. Wong3 states that the vallecula and the tongue base are the most common locations of laryngeal cysts.

This study did not take into account vocal folds cysts. In the supraglottis region, we preferably found epiglottic cysts (42% of total cases); 87.5% out of this figure were focused in the lingual aspect. Followed by the epiglottis, vallecula accounted for 31.3% of the cysts (See Chart 1). Cysts of the infraglottic region were not found and are considered rare5.

DeSanto classification was used in this study for laryngeal cysts that were divided in saccular and ductal cysts4. Saccular cysts are large and are found along the saccule, deeply to the vocal folds, aryepiglottic fold and anterior ventricle. It is believed that they result form cystic distension of the laryngeal saccule. Laryngoceles are included in this kind of cyst. Ductal cysts are formed by the distension of obstructed glandular ducts and are the most common laryngeal cysts. 4 This study found 3 cases of saccular cysts (15.7% of cases) and 15 cases of ductal cysts (78.8% of cases), confirming the observation of DeSanto that ductal cysts are the most commonly found. Such classification is largely accepted, however it is difficult to be applied in practice. We were not able to place in the classification one cyst found, and it corresponded to a lymphangioma. Pereira10 et al. in 1955 proposed a more detailed classification, in which laryngeal cysts were subdivided in neoplastic and non-neoplastic (or retention cysts). Lymphangioma would be a neoplastic cyst, together with desembryoplastic cysts (that may be congenital or acquired), traumatic (accidental or surgical) and cystic hemangioma. Neoplastic cysts or retention cysts are subdivided in glandular, tonsilloid and lymphatic. Mitchell1 thinks the differentiation between ductal and saccular cysts proposed by DeSanto is of semantic nature, since the natural history and outcome do not change with the classification. 11

Several forms of treatment have been described for laryngeal cysts, namely aspiration, marsupialization, laser ablation and total excision with endoscopy technique or external route. Recurrence is strictly related to maintenance of remains in the cyst wall, therefore total excision is recommended in order to have lower recurrence rates. 7

All patients have undergone total excision of the cysts, 18 through microlaryngoscopy and 1 through external route. Recurrence rate was 10.5%. Other authors confirmed this low recurrence rate. 1,5. Moreover, this recurrence rate could have been even lower if we had used CO2 laser as advocated by Danish 7.

Conclusion

This study reported our experience with laryngeal supraglottis cysts in which a clear understanding of the clinical history and natural outcome of laryngeal cysts are key for specialist physicians to diagnose and properly treat this condition with relatively simple methods.

References

1. Mitchell DB, Irwin BC, Bailey CM, Evans JNG. Cysts of the infant larynx. J Laryngol Otol 1987;101:833-7.
2. Albert DM, Ali Z. Two cases of vallecular cyst presenting with acute stridor. J Laryngol Otol 1985;99:421-5.
3. Wong KS, Li HY, Huang TS. Vallecular cyst synchronous with laryngomalacia: Presentation of two cases. Otolaryngol Head Neck Surg 1995;113:621-4.
4. DeSanto LW, Devine KD, Weiland LH. Cysts of the larynx - Classification. Laryngoscope 1970;80:145-176.
5. Lam HCK, Abdullah VJ, Soo G. Epiglottic cyst. Otolaryngol Head Neck Surg 2000;122:311-2.
6. Albert SWK. Vallecular cyst: repot of four cases - one with co-existing laryngomalacia. J Laryngol Otol 2000;114:224-226.
7. Danish HMN, Meleca RJ, Dworkin JP, Abbarah TR. Laryngeal Obstructing Saccular Cysts: a review of this disease and treatment approach emphasizing complete endoscopic carbon dioxide laser excision. Arch Otolaryngol Head Neck Surg 1998;124:593-596.
8. Newman BH, Taxy JB, Laker HI. Laryngeal cyst in adults. Am J Clin Pathol 1984;81:715-20.
9. LaBagnaro J. Cysts of the base of the tongue in infants: an unusual cause of neonatal airway obstruction. Otolaryngol Head Neck Surg 1989;101:108-11.
10. Pereira C. Quistos do laringe. Rev. Bras Otorrinolaringologia 1955;23:159-200.
11. Ramesar K, Alibizzati C. Laryngeal cysts: clinical relevance of a modified working classification. J Laryngol Otol 1988;102:932-5.

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1 Post-graduate physician, Clinic of Otorhinolaryngology, Hospital das Clínicas, Medical School, University of São Paulo (HCFMUSP).
2 Undergraduate, Medical School, FMUSP.
3 Assistant Physician, Ph.D., Clinic of Otorhinolaryngology, HCFMUSP.
4 Assistant Physician, Clinic of Otorhinolaryngology, HCFMUSP.
5 Ph.D., Professor, Clinic of Otorhinolaryngology, FMUSP.
Study conducted at the Division of the Clinic of Otorhinolaryngology, Medical School, University of São Paulo.

Address correspondence to: Rafael Burihan Cahali - Divisão de Clínica Otorrinolaringológica -
Hospital das Clínicas - FMUSP Av. Dr. Enéas de Carvalho Aguiar, 255, 6º andar, sala 6021
São Paulo - SP Fax (55 11)280-0299

Article submitted on January 24, 2002. Article accepted on August 15, 2002

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