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Potential Causative Factors for Saccular Disorders: Association with Smoking and Other Laryngeal Pathologies
ARTICLE IN PRESS Potential Causative Factors for Saccular Disorders: Association with Smoking and Other Laryngeal Pathologies *,†,1Oded Cohen, *,1Sharon Tzelnick, *,†Yael Shapira Galitz, *,†Hagit Shoffel-Havakuk, *Moshe Hain, *Doron Halperin, and *Yonatan Lahav, *Rehovot and †Jerusalem, Israel Summary: Objective. To describe risk factors, clinical presentation, and outcome of patients with saccular disorders. Study Design. Case control with chart review Methods. A single center retrospective study. Case group included all adult patients, presenting with saccular disorders (saccular cyst or laryngocele), between the years 2010 and 2015. A matched group of patients with vocal fold cyst served as the control. Results. Twenty-nine patients met the inclusion criteria: 15 males and 14 females; the mean age was 60.5(±11.2). The median follow-up period was 10 months (range 2–48). Overall, 75.9% (22) had a positive smoking history; 55.2% (16) were active and 20.7% (6) were past smokers. The median pack- years of all smokers in the saccular disorder group was 40 (range 1–67). Saccular disorder patients demonstrated significantly higher prevalence of active smoking when compared to control patients (55.2% versus 17.9%, P = 0.014). Sixty-nine percent of the patients had some synchronous vocal fold comorbidity. The leading vocal fold comorbidity was Reinke’s edema in 41% (12). Synchronous vocal fold comorbidities were significantly more prevalent in smokers compared with nonsmokers—82% (18 of 22) and 29% (2 of 7), respectively (P = 0.008). Surgical treatment was performed on 26 patients; all of whom underwent complete resection, either by endoscopic (92%), external (4%), or combined external and endoscopic (4%) approaches. There was a single case of recurrence (4%), 10 months following initial resection. Conclusion. Saccular disorders are associated with smoking and synchronous vocal fold comorbidity. Complete resection is recommended as surgical outcome is excellent. Key Words: Saccular–Laryngocele–Smoking–Vocal folds–Endoscopic.
INTRODUCTION Saccular cyst and laryngocele are uncommon pathologies of the laryngeal saccule and ventricle, known as “saccular disorders”.1,2 Historical case series1–5 have classified and delineated management principles, which have changed greatly in recent years.6–8 The laryngeal saccule is located between the vestibular and vocal folds (VeF and VoF, respectively), exvaginating to form a blind sac adjacent to the paraglottic space. Its physiologic function is unclear. Lubrication of the VoF has been hypothesized, although this function seems to have greater physiologic importance in apes.9 Saccular cyst is a dilation of the anatomical laryngeal saccule, filled with mucus, as a result of its orifice obstruction. Hence, it does not communicate with the laryngeal lumen.1,2 Saccular cysts are classified based on their relation to the VeF and VoF1,2: (1) anterior cysts lie between the VeF and VoF; and (2) lateral saccular cysts extend posterosuperiorly into the VeF and aryepiglottic folds. Clinical presentation may include various vocal Accepted for publication January 11, 2017. Financial support or funding: None. Conflict of interest: None. 1 These authors contributed equally to this study. From the *Department of Otolaryngology, Head and Neck Surgery, Kaplan Medical Center, Rehovot, Israel; and the †Hebrew University and Hadassah Medical School, Jerusalem, Israel. Address correspondence and reprint requests to Oded Cohen, Department of Otolaryngology, Head and Neck Surgery, Kaplan Medical Center, POB 1, Rehovot 76100, Israel. E-mail: [email protected] Journal of Voice, Vol. ■■, No. ■■, pp. ■■-■■ 0892-1997 © 2016 The Voice Foundation. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jvoice.2017.01.004
complaints, cough and globus,1–8 and rarely, airway obstruction.2 The management of saccular cysts has shifted in recent years as complete endoscopic resection using CO2 laser6 has replaced previous methods that included endoscopic excisional biopsy, endoscopic unroofing, and external approach.1,2 Laryngocele is an abnormal dilation of the saccule and the ventricle above the level of the thyroid cartilage, which communicates with the laryngeal lumen.1,2 It is classified by both its anatomic relation to the thyrohyoid membrane and by its content. An internal laryngocele lies medial to the thyrohyoid membrane whereas an external one lies lateral to it. A combined laryngocele is situated on both sides of the thyrohyoid membrane. Unlike saccular cyst, laryngocele is filled with air.1,2 If the orifice is obstructed and filled with pus, it is termed laryngopyocele. Hence, internal laryngopyoceles are indistinguishable from infected saccular cysts. 2,3 The etiology of laryngocele remains unclear up to date, yet three theories have been promoted: (1) atavistic remnants of lateral air sacs, (2) congenital anomaly, and (3) chronic exposure to increased translaryngeal pressure.5 The presentation of an internal laryngocele is similar to that of a saccular cyst, whereas an external laryngocele presents as a cervical mass. Initially, an open surgical approach was recommended.1,2 However, recent publications advocate endoscopic approach alone or combined with an open approach as indicated.7,8 In this study, we review adult patients presenting with saccular disorders, examine possible new risk factors that may
ARTICLE IN PRESS 2 contribute to the development of these disorders, and discuss the surgical outcome and benefits of its excision. MATERIALS AND METHODS After obtaining Institutional Review Board approval, we performed a single-institute, retrospective, matched case-control study. Patients who appeared with a mass suspected for a saccular disorder at our laryngology service between the years 2010 and 2015 were enrolled to the case group. The control group comprised of twenty nine matched patients who have undergone direct laryngoscopy (DL) and had a histological diagnosis of vocal fold cyst following excision. Matching was based on gender, age, and comorbidities. Saccular disorders were diagnosed and classified according to De Santo et al and Holinger et al.1,2 Exclusion criteria included all patients whose final pathology was not compatible with a saccular disorder. Recorded data included gender, age, comorbidities, and smoking history. For the case group, we also recorded nonmalignant laryngeal copathologies, previous radiation, neck surgery or direct laryngoscopy, history of laryngeal carcinoma, presenting symptoms, operative approach, macroscopic findings, side and classification, final pathology report, recurrence, complications, and length of follow-up. Socioeconomic status was categorized according to residential address as documented, following the Israeli Central Bureau of Statistics’ 2004 Peripherality Index of Local Authorities.10 Laryngeal comorbidities were defined as ipsi- or contralateral according to their relation to the saccu-
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lar disorder .This was also defined if a side was stated as dominant in a bilateral comorbidity (eg, Reinke’s edema). Relative size was measured and divided according to the degree of concealment by the saccular disorder: (1) up to 25%; (2) 25%–50%; (3) 50%–75%; and (4) more than 75% of the ipsilateral VeF and Vof were affected by the disorder (either the VeF was distended or the VoF was obscured by the lesion) (Figure 1; A1; B1). Smoking status was defined as follows: ex-smokers were subjects who had smoked daily and ceased smoking over 1 month before initial clinic meeting. Nonsmokers were those who had never smoked.11 Smokers were subdivided into light, moderate, and heavy smokers (<10, 10–20, and > 20 cigarettes per day, respectively).11,12 Prior to surgery, all patients underwent evaluation by an expert laryngologist (Y.L.), including videostroboscopic examination either by flexible video rhinolaryngoscope ENF-VQ (Olympus, Center Valley, PA, USA) or Karl Stortz 10 mm, 700 rigid endoscope (Karl Stortz Gmbh & Co. Tuttlingen, Germany). Surgical Procedures All surgical excisions were performed under general anesthesia, using a 5 mm laser endotracheal tube (Mallinckrodt, Covidien, Mansfield, MA, USA) and Wolf laryngoscope and suspension system (Richard Wolf Medical Instruments Corporation, Vernon Hills, IL, USA). Except for a single case of external approach, in all other cases, CO2 laser (Lumenis, Yokneam Industrial Park, Israel) was used in a super-pulse mode, 3–6 W. All saccular
FIGURE 1. Pre- and postoperative evaluation of saccular disorders. A1. anterior saccular cyst concealing 100% of vocal folds and 75% of vestibular fold. A2. After endoscopic translaryngeal resection (see Materials and Methods). B1. Combined laryngocele. B2. After endoscopic transhypopharyngeal resection (see Materials and Methods).
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disorders were completely excised. Three surgical approaches were used for the excision of the saccular disorders (Figure 1; A1 and A1; B1 and B2). Endoscopic translaryngeal approach included an en bloc resection of the lesion with the vestibular fold. Dissection along the inner perichondrium of the thyroid cartilage toward the floor of the ventricle allowed for a complete resection. Endoscopic transhypopharyngeal approach was used in large expanding saccular disorders. Initial laser incision was performed through the medial wall of the pyriform sinus toward the para- and pre-epiglottic spaces, until identification of the lesion and its dissection from the intact VeF and vallecular mucosa. Transcervical external approach was used for combined laryngoceles. The skin incision was made at the level of the thyrohyoid membrane area, followed by careful dissection until the lesion was identified and resected. This procedure was performed either by open external approach alone or combined with endoscopic transhypopharyngeal laser resection. Statistical analysis was performed with SPSS software, version 21.0 (IBM Corp., Armonk, NY, USA). Categorical variables were described using frequency and percentage. Variables that were normally distributed were described using mean and standard deviation (SD) and abnormally distributed variables were described using median and range. Association between noncategorical variables was calculated using Student t test. Differences in smoking history (detailed above) were studied using chi-squared test. Association between categorical variables was calculated using chi-squared test. Association between nonparametric variables was performed using Mann-Whitney U test, as appropriate. Two-tailed P value <0.05 was considered significant. RESULTS The initial review revealed 32 patients with lesions suspected for saccular disorders. Three patients who were initially thought to have a saccular disorder had a final pathology of hemangioma, lymphangioma, and Warthin’s tumor of the larynx, and were excluded. A total of 29 patients were included in the study group. Table 1 presents a summary of demographic and medical history of the study group. The male to female ratio was 15:14. Mean age was 60.5 (±11.2) years. Eighty-two percent of the patients were of high socioeconomic status. Only 17% of our cohort had more than 2 systemic comorbidities, whereas 31% had none. Dyslipidemia (nine patients) and hypertension (eight patients) were the most common. The median body mass index was 27 (range 18–34). Twenty-two patients (75.9%) had a positive smoking history: 55.2% (16) were active and 20.7% (6) were past smokers. Among active smokers, five were light, six were moderate, and five were heavy smokers. Among past smokers, three were heavy and three were light-moderate smokers.11,12 The median pack-years of all smokers in the saccular disorder group was 40 (range 1–67). Differences between patients with saccular disorders (case group) and patients with vocal fold cysts (control group) are presented in Table 2. Patients with saccular disorders demonstrated significantly higher prevalence of active smoking when compared to vocal fold cysts (55.2% versus 19.7%, P = 0.014, respectively), and borderline significance was found when overall history of smoking (past or current smoking) was compared
TABLE 1. Demographic and Medical Data of Study Population (n = 29) Category Gender Female Male Age (years, mean [SD]) Body mass index (median, range) Socioeconomic status Upper class High middle class Lower class Unknown* Systemic comorbidities 2< 1–2 None Smoking status Active smokers Past smokers Nonsmokers
48% (14/29) 52% (15/29) 60.5 (±11.2) 27 (18–34) 72% (21/29) 10% (3/29) 14% (4/29) 4% (1/29) 17% (5/29) 52% (15/29) 31% (9/29) 55% (16/29) 21% (6/29) 24% (7/29)
between the groups (75.8% versus 53.5%, P = 0.072, respectively). Next, we excluded all patients with a history of laryngeal carcinoma and/or neck irradiation (six patients, detailed below) that potentially may have confounded the association with active smoking aforementioned. Fifty-six percent of the included patients with saccular disorders not secondary to a possible known etiology (idiopathic saccular disorders) were active smokers and 21.7% were former smokers. The median pack-year of this subgroup was 52.5 years (range 1–60). Active smoking was also significantly more prevalent in this subgroup when compared to the control group (56.5% versus 19.7%, P = 0.015). A previous history of head and neck carcinoma was found in six patients (20.6%): four were laryngeal squamous cell carcinoma (SCC) (three T1N0M0, one T3N0M0). Regarding the original treatment of the carcinomas, all T1 carcinomas were treated by endoscopic resection. The T3 carcinoma underwent total laryngectomy. Two patients had other malignancies: one patient had laryngeal mucoepidermoid carcinoma and another patient had papillary thyroid carcinoma. Both patients were treated by surgery and external beam irradiation to the neck. Six patients had a surgical history of previous DL. The median time from the DL surgery to the diagnosis of saccular disorder was 9 months (range 1–21). Twenty patients (68.9%) had synchronous VoF comorbidities. The leading VoF comorbidity was Reinke’s edema (12 patients, 41.3%). Eight patients had multiple synchronous laryngeal comorbidities at presentation. Table 3 summarizes synchronous laryngeal comorbidities of the study group. When compared to nonsmokers, smoking patients demonstrated significantly higher prevalence of overall laryngeal comorbidities -both malignant and nonmalignant (81.8% versus 28.5%, P value = 0.008); as well as nonmalignant laryngeal comorbidities alone (72.7% versus 14.2%, P value = 0.006). Smokers did not exhibit an extensive disease when compared
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TABLE 2. Comparison of Demographics and Smoking Habits Between Patients with Saccular Disorders and Vocal Fold Cyst Category Age, years (mean ± SD) Female gender Comorbidities 2< 1–2 None Smoking status Active smokers Past smokers Nonsmokers Smoking history† Positive Negative
Saccular Disorders (n = 29)
Vocal Fold Cysts (n = 29)
P Value*
60.5 (±11.2) 48.3% (14/29)
58.2 (SD ± 7.14) 53.6% (15/28)
0.362 0.889
17.2% (5/29) 51.8% (15/29) 31% (9/29)
17.9% (5/28) 64.2% (18/28) 17.9% (5/28)
0.322
55.2% (16/29) 21.7% (6/29) 24.1% (7/29)
17.9% (5/28) 35.8% (10/28) 46.3% (13/28)
0.014
75.8% (22/29) 24.2% (7/29)
53.5% (15/28) 13/28 (46.4%)
0.072
* P value was calculated using t test for age, and chi-squared test for all other parameters. † Positive smoking history refers to patients who are past or active smokers grouped together.
to nonsmokers, as no statistical difference was found between lesions obstructing less and more than 50% of the VeF/VoF (50% versus 0%, P value = 0.182). Figure 2 describes the clinical presentation of saccular disorder patients. Dysphonia was the leading presenting symptom (62%). Voice-related symptoms (eg, dysphonia, aphonia, or voice fatigue) were found in 65.5 % (19 of 29). Five patients were asymptomatic: three were diagnosed on laryngoscopy performed during routine follow-up visits and two were diagnosed as an incidental finding in cervical imaging. Correlation between symptoms and the lesions’ relative size was analyzed. Symptomatic patients were more likely to have a lesion concealing more than 50% of the VoF (50% versus 0%, P value = 0.061). No statistical difference was found in relation to the VeF (50% versus 0%, P value = 0.436).
Twenty-six patients underwent surgical excision of their saccular disorder. Another patient with undiagnosed asymptomatic laryngocele had undergone total laryngectomy for advanced laryngeal cancer, with the laryngocele found on preoperative computed tomography scans and confirmed on histopathologic examination; of the two remaining patients, one with laryngocele refused surgery and the other one underwent surgery in another facility. Of the 26 patients who underwent excisional surgery, 20 (72.4%) were diagnosed with saccular cysts and 6 patients (27.6%) were diagnosed with laryngoceles. Of saccular cysts, nine (45%) were on the right side, eight on the left (40%), and three (15%) were bilateral. Fourteen cysts (70%) were defined as anterior and six (30%) were lateral. One patient had SCC within the cystic wall. Of the six patients diagnosed with laryngocele, four had combined laryngocele and
TABLE 3. Summary of Synchronous Laryngeal Comorbidities in the Cohort Pathology Reinke’s edema† (polypoid corditis) Laryngeal SCC Vocal fold dysplasia Vocal fold sulcus Vocal fold nodule Atrophic vocal fold Vocal fold hematoma Postoperative web Vocal fold paralysis Supraglottic stenosis Vocal fold polyp Vocal fold cyst Total‡
Patients (%)
Bilateral
Ipsilateral*
Contralateral*
12 (41.3) 4 (13.8) 4 (13.8) 2 (6.9) 2 (6.9) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4)
6 1 1 1 1 10
5 3 2 1 1 1 1 1 1 16
1 1 1 1 1 5
* Side was defined with relation to the saccular lesion—see Materials and Methods section. † All cases of Reinke’s edema were bilateral, yet in six cases differences between sides were documented. ‡ Eight patients had multiple metasynchronous laryngeal comorbidities at presentation. Abbreviation: SCC, squamous cell carcinoma.
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FIGURE 2. Clinical presentation of saccular disorder patients. two had internal laryngocele. Altogether, five patients had an infected saccular disorder (defined either as infected saccular cyst or laryngopyocele). Endoscopic resection was performed on 24 patients (92.3%). Twenty-one patients (87.5%) were operated on using a translaryngeal approach, and three patients (12.5%) were operated on using the transhypopharyngeal approach. Transcervical external approach was used for two combined laryngoceles: combined with transhypopharyngeal approach for one case and as a sole procedure for the other case. Postoperative median follow-up time was 10 (range 2–48) months. A single case of recurrence was noted 10 months following a resection of a right internal laryngocele. Simple granulation at the vestibular fold stump was observed in nine patients (34.6%) 1 month following excision (Figure 3). A single patient required resection of a symptomatic granulation 3.5 months following initial surgery. All other granulations resolved spontaneously by 12 months following surgery. One patient had a temporary right vocal hypokinesia, diagnosed 2 weeks following a resection of a combined laryngopyocele. This was not noticed in her follow-up 8 weeks following surgery. DISCUSSION Although recognized for many years, the pathogenesis and risk factors for saccular disorders are not completely understood. Our study summarizes our institute’s experience of patients diagnosed with these disorders. Relying on a relatively large series of patients, this study aims to examine possible new etiologies and risk factors, as well as to propose recommended management and follow-up for these disorders. Historically, classifications have been based on anatomic origin, location, and content.1,2 In their pioneering work, De Santo et al1 and Holinger et al2 have set the grounds for our knowledge regarding saccular disorders. Since their publications, many case series have focused on the management of these lesions, rather than on risk factors and pathogenesis.4–8,13,14 In neonates, saccular disorders are considered to be a congenital malformation/ atresia or atavistic remnants.2 In adults, trauma, neoplasia, inflammation, and fibrosis that occludes the saccular orifice were
related to saccular cysts’ formation, whereas laryngocele pathogenesis is associated with increased ventricular pressure.1,2 Increased ventricular pressure has been related to certain occupations,15 yet Amin et al,16 who measured 200 cadaver hemilarynges, concluded that the “blowing” theory is unlikely and improbable. In our study, the great majority of patients (76%) had a positive smoking history, a rate that is significantly higher than the general population (22% for men and 15% for women).17 To further establish our hypothesis in which smoking may contribute
FIGURE 3. In-office evaluation of postoperative granulations. A. Bilateral granulations seen in the vestibular fold in a patient 1 month following resection. B. Following outpatient clinic visit: left side— complete resolution; right side—90% absorption.
ARTICLE IN PRESS 6 to the pathogenesis of saccular disorders, we compared rates of smoking patients with another benign laryngeal pathology, one that has no known correlation with smoking (the vocal fold cyst group). Despite differences in pathogenesis and risk factors between the two pathologies, we assumed that similar smoking prevalence should be expected, as both pathologies have never been associated with smoking before. After matching for age, gender, and comorbidities, a significant difference was found in active smoking prevalence between the groups. This was also validated after exclusion of potential confounding patients with a history of laryngeal cancer and/or neck irradiation. Despite its limitations, we believe that this comparison contributes and supports our theory in which smoking may play a role in the pathogens of saccular disorder, probably by inducing inflammatory reactions within the larynx. Not surprisingly, the great majority of smoking patients presented with additional laryngeal comorbidity, with bilateral diffuse polyposis being the most common. High rates of smokers were also reported by Holinger et al (61% out of 31 patients with saccular cysts2) and Martinez et al (58% out of 12 patients8). Marom et al described two cases of saccular disorders and suggested that smoking may play a role in the pathogenesis.18 After reviewing previous case series and reports that suggested possible etiologies for saccular disorders, we found an ample number of patients with documented smoking history.12,16,19–26 Seven cases of saccular disorders in our series were a result of previously known reported etiologies, which may lead to direct blockage of the orifice: carcinoma of the larynx,27 radiation to the neck,6 and following laryngeal/neck surgery.28 We suggest that smoking may play a significant role in the pathogenesis of saccular disorders, especially when coexisting with other comorbidities, such as bilateral diffuse polyposis (Reinke’s edema). Two proposed mechanisms by which smoking may contribute to the formation of saccular disorders are as follows: (1) irritation of the laryngeal mucosa by smoke, leading to overproduction of mucus by minor glands and goblet cells secretions and possible obstruction of the ventricle; and (2) direct obstruction of the ventricle caused by Reinke’s edema. A recent study on cigarette smoking mechanism has shown that during one cycle of smoking, the larynx is the most exposed anatomical site (55% of the cycle period), thus supporting the possible explanation of mucosal irritation and blockage.29 The epidemiologic data of our study group are in concordance with previous studies. Saccular disorders seem to have no gender preference. In adults, it seems to present mostly within the sixth to seventh decade of life.2,3,6 Our study is the first to try and characterize the general health condition of these patients. The lack of multicomorbidities in our study group may suggest that the pathology is a result of local rather than systemic factors. The vast majority of our study’s population was of high socioeconomic status. Whether socioeconomic status has a direct impact on the pathogenesis or prevalence of saccular disorders is a matter for further studies and validation. Like previous studies, the most common complaints of saccular disorders were voice related in nature, dysphonia being the most common. Like Lawrence and De Santo,3 we found that the
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presence of symptoms in patients with a saccular disorder correlates with its size. In his publication, Dursun et al recommended CO2 laser resection for internal laryngoceles and an external approach for external and combined laryngoceles.7 Martinez Devesa et al8 reported 12 cases of laryngocele managed endoscopically using CO 2 laser; two of them were combined. Our combined laryngoceles were excised with a combined approach, similar to the preliminary report of Ettema et al.13 The combined approach allows carful separation of the cyst from the delicate laryngeal and pharyngeal areas under the microscope, before a complete transcervical removal. This approach prevents unintended cyst rupture. Recurrence of saccular disorders following surgical excision is uncommon, ranging from 0% to 22%,1,2,5,7,8,26,30,31 although the series vary in both follow-up length and sample size. Whereas some reported early recurrence within the first 2 months,6 other reported cyst recurrence 18–24 months following resection.31 Our single recurrence was diagnosed 10 months following resection. Nearly half of the patients were diagnosed with a simple granulation following surgery, yet postoperative granulation is to be expected after the removal of the vestibular vocal fold. Only two interventions were needed following resection: a removal of a hemorrhagic granulation in the outpatient clinic 5 months following surgery and one patient who underwent DL for granuloma excision. Our results suggest that complications that may require intervention usually appear within the first months following surgery, and therefore support the approach that shortterm follow-up should be more frequent, as suggested by Young et al.6 Our study is not without limitations; it is a retrospective, singlesurgeon (Y.L.) study. As with previous major series, our series is still limited by its sample size, and hence statistical analysis is mostly descriptive. Finally, clinical suspicion of saccular disorder is based on endoscopic evaluation. Three patients who were initially thought to have a saccular disorder had a different final pathology. This joins previous reports of mantle cell lymphoma,32 laryngeal chondroma,33 supraglottic schwannoma,34 and leiomyosarcoma35 presenting as saccular disorders. Moreover, the possibility of a hidden malignancy cannot be overlooked, as represented in our series by one patient with SCC within the cyst wall. Considering the excellent outcome of endoscopic resection, we believe that all saccular disorders should be recommended for surgical excision, regardless of size or clinical presentation as first thought.3 Whether a recurrence of a saccular disorder should be resected is a matter of joint consultation with the patient, especially if asymptomatic. CONCLUSION Smoking and its derivative pathologies may play a significant role in the pathogenesis of saccular disorders in addition to other known causes for direct obstruction of the saccule. Surgical excision of saccular disorders has shown excellent results with a minor rate of recurrence, and low rate of complications. Considering the possibility of a different neoplastic pathology masquerading as a saccular disorder, we recommend that all
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disorders should be completely excised. Follow-up should be maintained tightly at first.
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17. Ministry of Health, The 2015 Minister of Health National Smoking Status Report. Public Health Service, Israel. May 2016. 18. Marom T, Roth Y, Cinamon U. Laryngocele: a rare long-term complication following neck surgery? J Voice. 2011;25:272–274. 19. Desai DP, Portugal LG, Walner DL, et al. Bilateral saccular cysts after cocaine use. Otolaryngol Head Neck Surg. 1999;120:754–757. 20. Schild JA, Grybauskas VT, Mafee MF. Laryngeal amyloidosis and laryngocele. Ann Otol Rhinol Laryngol. 1983;92(4 pt 1):337–339. 21. Lancella A, Abbate G, Dosdegani R. Mixed laryngocele: a case report and review of the literature. Acta Otorhinolaryngol Ital. 2007;27:255–257. 22. Mitroi M, Ca˘pita˘nescu A, Popescu FC, et al. Laryngocele associated with laryngeal carcinoma. Rom J Morphol Embryol. 2011;52:183–185. 23. Aidonis I, Skalimis A, Kirodimos E. Surgical and anesthetic considerations of laryngeal saccular cyst: a case report. J Med Case Rep. 2011;5:283. 24. Griffith CC, Goodstein LA, Tuluc M, et al. A rare case of carcinoma cuniculatum involving the larynx in association with a saccular cyst. Head Neck Pathol. 2015;9:421–425. 25. Pennings RJ, van den Hoogen FJ, Marres HA. Giant laryngoceles: a cause of upper airway obstruction. Eur Arch Otorhinolaryngol. 2001;258:137–140. 26. Cassano L, Lombardo P, Marchese-Ragona R, et al. Laryngopyocele: three new clinical cases and review of the literature. Eur Arch Otorhinolaryngol. 2000;257:507–511. 27. Naiboglu B, Aslan I, Oysu C, et al. Laryngocoele as an unusual complication of supracricoid partial laryngectomy: evaluation of 62 cases. J Laryngol Otol. 2010;124:646–649. 28. Celin SE, Johnson J, Curtin H, et al. The association of laryngoceles with squamous cell carcinoma of the larynx. Laryngoscope. 1991;101:529–536. 29. Shoffel-Havakuk H, Halperin D, Haimovich Y, et al. Analysis of cigarette smoking mechanism by real time video-endoscopic documentation. Inhal Toxicol. 2015;27:495–501. 30. Hogikyan ND, Bastian RW. Endoscopic CO2 laser excision of large or recurrent laryngeal saccular cysts in adults. Laryngoscope. 1997;107:260– 265. 31. Thabet MH, Kotob H. Lateral saccular cysts of the larynx. Aetiology, diagnosis and management. J Laryngol Otol. 2001;115:293–297. 32. Groom KL, Ruhl DS, Sniezek JC. Mantle cell lymphoma presenting as a saccular cyst. Otolaryngol Head Neck Surg. 2012;146:173–174. 33. Papila I, Aciog˘lu E, Karaman E, et al. Laryngeal chondroma presenting as a laryngopyocele. Eur Arch Otorhinolaryngol. 2005;262:473–476. 34. Rosen FS, Pou AM, Quinn FB Jr. Obstructive supraglottic schwannoma: a case report and review of the literature. Laryngoscope. 2002;112:997–1002. 35. Helmberger RC, Croker BP, Mancuso AA. Leiomyosarcoma of the larynx presenting as a laryngopyocele. AJNR Am J Neuroradiol. 1996;17:1112–1114.
INTRODUCTION Saccular cyst and laryngocele are uncommon pathologies of the laryngeal saccule and ventricle, known as “saccular disorders”.1,2 Historical case series1–5 have classified and delineated management principles, which have changed greatly in recent years.6–8 The laryngeal saccule is located between the vestibular and vocal folds (VeF and VoF, respectively), exvaginating to form a blind sac adjacent to the paraglottic space. Its physiologic function is unclear. Lubrication of the VoF has been hypothesized, although this function seems to have greater physiologic importance in apes.9 Saccular cyst is a dilation of the anatomical laryngeal saccule, filled with mucus, as a result of its orifice obstruction. Hence, it does not communicate with the laryngeal lumen.1,2 Saccular cysts are classified based on their relation to the VeF and VoF1,2: (1) anterior cysts lie between the VeF and VoF; and (2) lateral saccular cysts extend posterosuperiorly into the VeF and aryepiglottic folds. Clinical presentation may include various vocal Accepted for publication January 11, 2017. Financial support or funding: None. Conflict of interest: None. 1 These authors contributed equally to this study. From the *Department of Otolaryngology, Head and Neck Surgery, Kaplan Medical Center, Rehovot, Israel; and the †Hebrew University and Hadassah Medical School, Jerusalem, Israel. Address correspondence and reprint requests to Oded Cohen, Department of Otolaryngology, Head and Neck Surgery, Kaplan Medical Center, POB 1, Rehovot 76100, Israel. E-mail: [email protected] Journal of Voice, Vol. ■■, No. ■■, pp. ■■-■■ 0892-1997 © 2016 The Voice Foundation. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jvoice.2017.01.004
complaints, cough and globus,1–8 and rarely, airway obstruction.2 The management of saccular cysts has shifted in recent years as complete endoscopic resection using CO2 laser6 has replaced previous methods that included endoscopic excisional biopsy, endoscopic unroofing, and external approach.1,2 Laryngocele is an abnormal dilation of the saccule and the ventricle above the level of the thyroid cartilage, which communicates with the laryngeal lumen.1,2 It is classified by both its anatomic relation to the thyrohyoid membrane and by its content. An internal laryngocele lies medial to the thyrohyoid membrane whereas an external one lies lateral to it. A combined laryngocele is situated on both sides of the thyrohyoid membrane. Unlike saccular cyst, laryngocele is filled with air.1,2 If the orifice is obstructed and filled with pus, it is termed laryngopyocele. Hence, internal laryngopyoceles are indistinguishable from infected saccular cysts. 2,3 The etiology of laryngocele remains unclear up to date, yet three theories have been promoted: (1) atavistic remnants of lateral air sacs, (2) congenital anomaly, and (3) chronic exposure to increased translaryngeal pressure.5 The presentation of an internal laryngocele is similar to that of a saccular cyst, whereas an external laryngocele presents as a cervical mass. Initially, an open surgical approach was recommended.1,2 However, recent publications advocate endoscopic approach alone or combined with an open approach as indicated.7,8 In this study, we review adult patients presenting with saccular disorders, examine possible new risk factors that may
ARTICLE IN PRESS 2 contribute to the development of these disorders, and discuss the surgical outcome and benefits of its excision. MATERIALS AND METHODS After obtaining Institutional Review Board approval, we performed a single-institute, retrospective, matched case-control study. Patients who appeared with a mass suspected for a saccular disorder at our laryngology service between the years 2010 and 2015 were enrolled to the case group. The control group comprised of twenty nine matched patients who have undergone direct laryngoscopy (DL) and had a histological diagnosis of vocal fold cyst following excision. Matching was based on gender, age, and comorbidities. Saccular disorders were diagnosed and classified according to De Santo et al and Holinger et al.1,2 Exclusion criteria included all patients whose final pathology was not compatible with a saccular disorder. Recorded data included gender, age, comorbidities, and smoking history. For the case group, we also recorded nonmalignant laryngeal copathologies, previous radiation, neck surgery or direct laryngoscopy, history of laryngeal carcinoma, presenting symptoms, operative approach, macroscopic findings, side and classification, final pathology report, recurrence, complications, and length of follow-up. Socioeconomic status was categorized according to residential address as documented, following the Israeli Central Bureau of Statistics’ 2004 Peripherality Index of Local Authorities.10 Laryngeal comorbidities were defined as ipsi- or contralateral according to their relation to the saccu-
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lar disorder .This was also defined if a side was stated as dominant in a bilateral comorbidity (eg, Reinke’s edema). Relative size was measured and divided according to the degree of concealment by the saccular disorder: (1) up to 25%; (2) 25%–50%; (3) 50%–75%; and (4) more than 75% of the ipsilateral VeF and Vof were affected by the disorder (either the VeF was distended or the VoF was obscured by the lesion) (Figure 1; A1; B1). Smoking status was defined as follows: ex-smokers were subjects who had smoked daily and ceased smoking over 1 month before initial clinic meeting. Nonsmokers were those who had never smoked.11 Smokers were subdivided into light, moderate, and heavy smokers (<10, 10–20, and > 20 cigarettes per day, respectively).11,12 Prior to surgery, all patients underwent evaluation by an expert laryngologist (Y.L.), including videostroboscopic examination either by flexible video rhinolaryngoscope ENF-VQ (Olympus, Center Valley, PA, USA) or Karl Stortz 10 mm, 700 rigid endoscope (Karl Stortz Gmbh & Co. Tuttlingen, Germany). Surgical Procedures All surgical excisions were performed under general anesthesia, using a 5 mm laser endotracheal tube (Mallinckrodt, Covidien, Mansfield, MA, USA) and Wolf laryngoscope and suspension system (Richard Wolf Medical Instruments Corporation, Vernon Hills, IL, USA). Except for a single case of external approach, in all other cases, CO2 laser (Lumenis, Yokneam Industrial Park, Israel) was used in a super-pulse mode, 3–6 W. All saccular
FIGURE 1. Pre- and postoperative evaluation of saccular disorders. A1. anterior saccular cyst concealing 100% of vocal folds and 75% of vestibular fold. A2. After endoscopic translaryngeal resection (see Materials and Methods). B1. Combined laryngocele. B2. After endoscopic transhypopharyngeal resection (see Materials and Methods).
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disorders were completely excised. Three surgical approaches were used for the excision of the saccular disorders (Figure 1; A1 and A1; B1 and B2). Endoscopic translaryngeal approach included an en bloc resection of the lesion with the vestibular fold. Dissection along the inner perichondrium of the thyroid cartilage toward the floor of the ventricle allowed for a complete resection. Endoscopic transhypopharyngeal approach was used in large expanding saccular disorders. Initial laser incision was performed through the medial wall of the pyriform sinus toward the para- and pre-epiglottic spaces, until identification of the lesion and its dissection from the intact VeF and vallecular mucosa. Transcervical external approach was used for combined laryngoceles. The skin incision was made at the level of the thyrohyoid membrane area, followed by careful dissection until the lesion was identified and resected. This procedure was performed either by open external approach alone or combined with endoscopic transhypopharyngeal laser resection. Statistical analysis was performed with SPSS software, version 21.0 (IBM Corp., Armonk, NY, USA). Categorical variables were described using frequency and percentage. Variables that were normally distributed were described using mean and standard deviation (SD) and abnormally distributed variables were described using median and range. Association between noncategorical variables was calculated using Student t test. Differences in smoking history (detailed above) were studied using chi-squared test. Association between categorical variables was calculated using chi-squared test. Association between nonparametric variables was performed using Mann-Whitney U test, as appropriate. Two-tailed P value <0.05 was considered significant. RESULTS The initial review revealed 32 patients with lesions suspected for saccular disorders. Three patients who were initially thought to have a saccular disorder had a final pathology of hemangioma, lymphangioma, and Warthin’s tumor of the larynx, and were excluded. A total of 29 patients were included in the study group. Table 1 presents a summary of demographic and medical history of the study group. The male to female ratio was 15:14. Mean age was 60.5 (±11.2) years. Eighty-two percent of the patients were of high socioeconomic status. Only 17% of our cohort had more than 2 systemic comorbidities, whereas 31% had none. Dyslipidemia (nine patients) and hypertension (eight patients) were the most common. The median body mass index was 27 (range 18–34). Twenty-two patients (75.9%) had a positive smoking history: 55.2% (16) were active and 20.7% (6) were past smokers. Among active smokers, five were light, six were moderate, and five were heavy smokers. Among past smokers, three were heavy and three were light-moderate smokers.11,12 The median pack-years of all smokers in the saccular disorder group was 40 (range 1–67). Differences between patients with saccular disorders (case group) and patients with vocal fold cysts (control group) are presented in Table 2. Patients with saccular disorders demonstrated significantly higher prevalence of active smoking when compared to vocal fold cysts (55.2% versus 19.7%, P = 0.014, respectively), and borderline significance was found when overall history of smoking (past or current smoking) was compared
TABLE 1. Demographic and Medical Data of Study Population (n = 29) Category Gender Female Male Age (years, mean [SD]) Body mass index (median, range) Socioeconomic status Upper class High middle class Lower class Unknown* Systemic comorbidities 2< 1–2 None Smoking status Active smokers Past smokers Nonsmokers
48% (14/29) 52% (15/29) 60.5 (±11.2) 27 (18–34) 72% (21/29) 10% (3/29) 14% (4/29) 4% (1/29) 17% (5/29) 52% (15/29) 31% (9/29) 55% (16/29) 21% (6/29) 24% (7/29)
between the groups (75.8% versus 53.5%, P = 0.072, respectively). Next, we excluded all patients with a history of laryngeal carcinoma and/or neck irradiation (six patients, detailed below) that potentially may have confounded the association with active smoking aforementioned. Fifty-six percent of the included patients with saccular disorders not secondary to a possible known etiology (idiopathic saccular disorders) were active smokers and 21.7% were former smokers. The median pack-year of this subgroup was 52.5 years (range 1–60). Active smoking was also significantly more prevalent in this subgroup when compared to the control group (56.5% versus 19.7%, P = 0.015). A previous history of head and neck carcinoma was found in six patients (20.6%): four were laryngeal squamous cell carcinoma (SCC) (three T1N0M0, one T3N0M0). Regarding the original treatment of the carcinomas, all T1 carcinomas were treated by endoscopic resection. The T3 carcinoma underwent total laryngectomy. Two patients had other malignancies: one patient had laryngeal mucoepidermoid carcinoma and another patient had papillary thyroid carcinoma. Both patients were treated by surgery and external beam irradiation to the neck. Six patients had a surgical history of previous DL. The median time from the DL surgery to the diagnosis of saccular disorder was 9 months (range 1–21). Twenty patients (68.9%) had synchronous VoF comorbidities. The leading VoF comorbidity was Reinke’s edema (12 patients, 41.3%). Eight patients had multiple synchronous laryngeal comorbidities at presentation. Table 3 summarizes synchronous laryngeal comorbidities of the study group. When compared to nonsmokers, smoking patients demonstrated significantly higher prevalence of overall laryngeal comorbidities -both malignant and nonmalignant (81.8% versus 28.5%, P value = 0.008); as well as nonmalignant laryngeal comorbidities alone (72.7% versus 14.2%, P value = 0.006). Smokers did not exhibit an extensive disease when compared
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TABLE 2. Comparison of Demographics and Smoking Habits Between Patients with Saccular Disorders and Vocal Fold Cyst Category Age, years (mean ± SD) Female gender Comorbidities 2< 1–2 None Smoking status Active smokers Past smokers Nonsmokers Smoking history† Positive Negative
Saccular Disorders (n = 29)
Vocal Fold Cysts (n = 29)
P Value*
60.5 (±11.2) 48.3% (14/29)
58.2 (SD ± 7.14) 53.6% (15/28)
0.362 0.889
17.2% (5/29) 51.8% (15/29) 31% (9/29)
17.9% (5/28) 64.2% (18/28) 17.9% (5/28)
0.322
55.2% (16/29) 21.7% (6/29) 24.1% (7/29)
17.9% (5/28) 35.8% (10/28) 46.3% (13/28)
0.014
75.8% (22/29) 24.2% (7/29)
53.5% (15/28) 13/28 (46.4%)
0.072
* P value was calculated using t test for age, and chi-squared test for all other parameters. † Positive smoking history refers to patients who are past or active smokers grouped together.
to nonsmokers, as no statistical difference was found between lesions obstructing less and more than 50% of the VeF/VoF (50% versus 0%, P value = 0.182). Figure 2 describes the clinical presentation of saccular disorder patients. Dysphonia was the leading presenting symptom (62%). Voice-related symptoms (eg, dysphonia, aphonia, or voice fatigue) were found in 65.5 % (19 of 29). Five patients were asymptomatic: three were diagnosed on laryngoscopy performed during routine follow-up visits and two were diagnosed as an incidental finding in cervical imaging. Correlation between symptoms and the lesions’ relative size was analyzed. Symptomatic patients were more likely to have a lesion concealing more than 50% of the VoF (50% versus 0%, P value = 0.061). No statistical difference was found in relation to the VeF (50% versus 0%, P value = 0.436).
Twenty-six patients underwent surgical excision of their saccular disorder. Another patient with undiagnosed asymptomatic laryngocele had undergone total laryngectomy for advanced laryngeal cancer, with the laryngocele found on preoperative computed tomography scans and confirmed on histopathologic examination; of the two remaining patients, one with laryngocele refused surgery and the other one underwent surgery in another facility. Of the 26 patients who underwent excisional surgery, 20 (72.4%) were diagnosed with saccular cysts and 6 patients (27.6%) were diagnosed with laryngoceles. Of saccular cysts, nine (45%) were on the right side, eight on the left (40%), and three (15%) were bilateral. Fourteen cysts (70%) were defined as anterior and six (30%) were lateral. One patient had SCC within the cystic wall. Of the six patients diagnosed with laryngocele, four had combined laryngocele and
TABLE 3. Summary of Synchronous Laryngeal Comorbidities in the Cohort Pathology Reinke’s edema† (polypoid corditis) Laryngeal SCC Vocal fold dysplasia Vocal fold sulcus Vocal fold nodule Atrophic vocal fold Vocal fold hematoma Postoperative web Vocal fold paralysis Supraglottic stenosis Vocal fold polyp Vocal fold cyst Total‡
Patients (%)
Bilateral
Ipsilateral*
Contralateral*
12 (41.3) 4 (13.8) 4 (13.8) 2 (6.9) 2 (6.9) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4) 1 (3.4)
6 1 1 1 1 10
5 3 2 1 1 1 1 1 1 16
1 1 1 1 1 5
* Side was defined with relation to the saccular lesion—see Materials and Methods section. † All cases of Reinke’s edema were bilateral, yet in six cases differences between sides were documented. ‡ Eight patients had multiple metasynchronous laryngeal comorbidities at presentation. Abbreviation: SCC, squamous cell carcinoma.
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FIGURE 2. Clinical presentation of saccular disorder patients. two had internal laryngocele. Altogether, five patients had an infected saccular disorder (defined either as infected saccular cyst or laryngopyocele). Endoscopic resection was performed on 24 patients (92.3%). Twenty-one patients (87.5%) were operated on using a translaryngeal approach, and three patients (12.5%) were operated on using the transhypopharyngeal approach. Transcervical external approach was used for two combined laryngoceles: combined with transhypopharyngeal approach for one case and as a sole procedure for the other case. Postoperative median follow-up time was 10 (range 2–48) months. A single case of recurrence was noted 10 months following a resection of a right internal laryngocele. Simple granulation at the vestibular fold stump was observed in nine patients (34.6%) 1 month following excision (Figure 3). A single patient required resection of a symptomatic granulation 3.5 months following initial surgery. All other granulations resolved spontaneously by 12 months following surgery. One patient had a temporary right vocal hypokinesia, diagnosed 2 weeks following a resection of a combined laryngopyocele. This was not noticed in her follow-up 8 weeks following surgery. DISCUSSION Although recognized for many years, the pathogenesis and risk factors for saccular disorders are not completely understood. Our study summarizes our institute’s experience of patients diagnosed with these disorders. Relying on a relatively large series of patients, this study aims to examine possible new etiologies and risk factors, as well as to propose recommended management and follow-up for these disorders. Historically, classifications have been based on anatomic origin, location, and content.1,2 In their pioneering work, De Santo et al1 and Holinger et al2 have set the grounds for our knowledge regarding saccular disorders. Since their publications, many case series have focused on the management of these lesions, rather than on risk factors and pathogenesis.4–8,13,14 In neonates, saccular disorders are considered to be a congenital malformation/ atresia or atavistic remnants.2 In adults, trauma, neoplasia, inflammation, and fibrosis that occludes the saccular orifice were
related to saccular cysts’ formation, whereas laryngocele pathogenesis is associated with increased ventricular pressure.1,2 Increased ventricular pressure has been related to certain occupations,15 yet Amin et al,16 who measured 200 cadaver hemilarynges, concluded that the “blowing” theory is unlikely and improbable. In our study, the great majority of patients (76%) had a positive smoking history, a rate that is significantly higher than the general population (22% for men and 15% for women).17 To further establish our hypothesis in which smoking may contribute
FIGURE 3. In-office evaluation of postoperative granulations. A. Bilateral granulations seen in the vestibular fold in a patient 1 month following resection. B. Following outpatient clinic visit: left side— complete resolution; right side—90% absorption.
ARTICLE IN PRESS 6 to the pathogenesis of saccular disorders, we compared rates of smoking patients with another benign laryngeal pathology, one that has no known correlation with smoking (the vocal fold cyst group). Despite differences in pathogenesis and risk factors between the two pathologies, we assumed that similar smoking prevalence should be expected, as both pathologies have never been associated with smoking before. After matching for age, gender, and comorbidities, a significant difference was found in active smoking prevalence between the groups. This was also validated after exclusion of potential confounding patients with a history of laryngeal cancer and/or neck irradiation. Despite its limitations, we believe that this comparison contributes and supports our theory in which smoking may play a role in the pathogens of saccular disorder, probably by inducing inflammatory reactions within the larynx. Not surprisingly, the great majority of smoking patients presented with additional laryngeal comorbidity, with bilateral diffuse polyposis being the most common. High rates of smokers were also reported by Holinger et al (61% out of 31 patients with saccular cysts2) and Martinez et al (58% out of 12 patients8). Marom et al described two cases of saccular disorders and suggested that smoking may play a role in the pathogenesis.18 After reviewing previous case series and reports that suggested possible etiologies for saccular disorders, we found an ample number of patients with documented smoking history.12,16,19–26 Seven cases of saccular disorders in our series were a result of previously known reported etiologies, which may lead to direct blockage of the orifice: carcinoma of the larynx,27 radiation to the neck,6 and following laryngeal/neck surgery.28 We suggest that smoking may play a significant role in the pathogenesis of saccular disorders, especially when coexisting with other comorbidities, such as bilateral diffuse polyposis (Reinke’s edema). Two proposed mechanisms by which smoking may contribute to the formation of saccular disorders are as follows: (1) irritation of the laryngeal mucosa by smoke, leading to overproduction of mucus by minor glands and goblet cells secretions and possible obstruction of the ventricle; and (2) direct obstruction of the ventricle caused by Reinke’s edema. A recent study on cigarette smoking mechanism has shown that during one cycle of smoking, the larynx is the most exposed anatomical site (55% of the cycle period), thus supporting the possible explanation of mucosal irritation and blockage.29 The epidemiologic data of our study group are in concordance with previous studies. Saccular disorders seem to have no gender preference. In adults, it seems to present mostly within the sixth to seventh decade of life.2,3,6 Our study is the first to try and characterize the general health condition of these patients. The lack of multicomorbidities in our study group may suggest that the pathology is a result of local rather than systemic factors. The vast majority of our study’s population was of high socioeconomic status. Whether socioeconomic status has a direct impact on the pathogenesis or prevalence of saccular disorders is a matter for further studies and validation. Like previous studies, the most common complaints of saccular disorders were voice related in nature, dysphonia being the most common. Like Lawrence and De Santo,3 we found that the
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presence of symptoms in patients with a saccular disorder correlates with its size. In his publication, Dursun et al recommended CO2 laser resection for internal laryngoceles and an external approach for external and combined laryngoceles.7 Martinez Devesa et al8 reported 12 cases of laryngocele managed endoscopically using CO 2 laser; two of them were combined. Our combined laryngoceles were excised with a combined approach, similar to the preliminary report of Ettema et al.13 The combined approach allows carful separation of the cyst from the delicate laryngeal and pharyngeal areas under the microscope, before a complete transcervical removal. This approach prevents unintended cyst rupture. Recurrence of saccular disorders following surgical excision is uncommon, ranging from 0% to 22%,1,2,5,7,8,26,30,31 although the series vary in both follow-up length and sample size. Whereas some reported early recurrence within the first 2 months,6 other reported cyst recurrence 18–24 months following resection.31 Our single recurrence was diagnosed 10 months following resection. Nearly half of the patients were diagnosed with a simple granulation following surgery, yet postoperative granulation is to be expected after the removal of the vestibular vocal fold. Only two interventions were needed following resection: a removal of a hemorrhagic granulation in the outpatient clinic 5 months following surgery and one patient who underwent DL for granuloma excision. Our results suggest that complications that may require intervention usually appear within the first months following surgery, and therefore support the approach that shortterm follow-up should be more frequent, as suggested by Young et al.6 Our study is not without limitations; it is a retrospective, singlesurgeon (Y.L.) study. As with previous major series, our series is still limited by its sample size, and hence statistical analysis is mostly descriptive. Finally, clinical suspicion of saccular disorder is based on endoscopic evaluation. Three patients who were initially thought to have a saccular disorder had a different final pathology. This joins previous reports of mantle cell lymphoma,32 laryngeal chondroma,33 supraglottic schwannoma,34 and leiomyosarcoma35 presenting as saccular disorders. Moreover, the possibility of a hidden malignancy cannot be overlooked, as represented in our series by one patient with SCC within the cyst wall. Considering the excellent outcome of endoscopic resection, we believe that all saccular disorders should be recommended for surgical excision, regardless of size or clinical presentation as first thought.3 Whether a recurrence of a saccular disorder should be resected is a matter of joint consultation with the patient, especially if asymptomatic. CONCLUSION Smoking and its derivative pathologies may play a significant role in the pathogenesis of saccular disorders in addition to other known causes for direct obstruction of the saccule. Surgical excision of saccular disorders has shown excellent results with a minor rate of recurrence, and low rate of complications. Considering the possibility of a different neoplastic pathology masquerading as a saccular disorder, we recommend that all
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disorders should be completely excised. Follow-up should be maintained tightly at first.
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