TY - JOUR
AU - Abbarah, Thabet R.
AB - Airway-obstructing saccular cysts in adults are rare laryngeal anomalies. Treatment with tracheotomy may be needed for control of the airway, often followed by marsupialization of the cyst wall. Unfortunately, recurrence rates are high following marsupialization. We describe 2 patients with saccular cysts obstructing the airway and discuss airway management and the results following complete endoscopic carbon dioxide laser excision. Both patients had normal voice and swallowing function postoperatively and are disease free.Saccular cysts of the larynx in adults are uncommon anomalies that rarely become large enough to produce airway obstruction. When such a lesion occurs, the airway must be adequately secured, often by tracheotomy, before excision is attempted. Endoscopic marsupialization of the dome of the cyst with laser ablation of the remaining cyst wall or excision of the cyst by an external approach are well-documented treatment options.Recurrences with the marsupialization technique are common, however, and there is increased morbidity associated with the external approach.Coupling the microscope with the carbon dioxide (CO2) laser and using the bivalve laryngoscope (Richard Wolf Medical Instruments, Rosemont, Ill) (Figure 1) to provide superior visualization of the supraglottic larynx now permit precise endoscopic removal of an entire cyst, without disruption of the cyst wall in most cases and with little morbidity to surrounding laryngeal structures.We describe 2 patients who presented with airway-obstructing saccular cysts, review this uncommon anomaly, and describe our results following complete endoscopic excision.Figure 1.The bivalve laryngoscope provides excellent exposure of the supraglottic larynx. The tip of the upper blade may be positioned under the laryngeal surface of the epiglottis to provide better exposure of the false vocal and pharyngoepiglottic folds. A wide malleable retractor may be used to protect the buccal mucosa. Reprinted with permission from Shapiro et al.REPORT OF CASESCASE 1A 76-year-old woman was seen in the Detroit Receiving Emergency Department, Detroit, Mich, with progressive dyspnea, hoarseness, and dysphagia during the past month. She denied having an upper respiratory tract infection, was afebrile, and had a normal leukocyte count. Fiberoptic laryngoscopy revealed a right-sided mucosal-covered mass involving the false vocal, aryepiglottic, and pharyngoepiglottic folds. The mass totally obstructed the view of the laryngeal inlet. A computed tomographic (CT) scan revealed a fluid-filled cyst measuring 3×4 cm (Figure 2) with extension through the thyrohyoid membrane. An airway-obstructing saccular cyst was the presumed diagnosis, and the patient underwent a tracheotomy under local anesthesia to provide a stable airway.Figure 2.Axial computed tomographic scan revealing a 3×4-cm right-sided saccular cyst involving the false vocal, aryepiglottic, and pharyngoepiglottic folds. Note extension through the thyrohyoid membrane.Postoperative videostroboscopic images taken the following day revealed that the cyst had partially decompressed (Figure 3). Four days after the tracheotomy, the patient underwent a microlaryngeal endoscopic CO2laser excision of the saccular cyst with the use of a bivalve laryngoscope with Lewy suspension to provide optimal visualization of the supraglottic larynx (Figure 1). The laryngoscope was positioned with the tip of the upper blade against the laryngeal surface of the epiglottis and the lower blade resting against the posterior pharyngeal wall. Once in position, the front and back of the bivalve laryngoscope were carefully opened to provide a wide field of view. The Lewy suspension was periodically released to prevent venous congestion of the tongue. With the laser in a continuous mode at a power setting ranging from 2 to 5 W, the false vocal fold mucosa was incised along its entire length, exposing the medial border of the cyst. The incision was extended at its midpoint into the pharyngoepiglottic fold, revealing the lateral margin of the cyst, which was noted to rest against the inner thyroid perichondrium. The cyst wall was carefully followed circumferentially using a combination of laser excision and delicate blunt dissection with a peanut sponge to separate the cyst from surrounding normal structures. The entire cyst and a portion of overlying false vocal fold mucosa were removed, leaving the true vocal fold and endolaryngeal mucosa undisturbed. The cyst bed was left to heal by secondary intention.Figure 3.Videolaryngoscopic view of the larynx after spontaneous partial decompression of a right-sided saccular cyst. The small arrow indicates the epiglottis; medium arrow, a saccular cyst; and large arrow, the tracheotomy tube.The patient had improved voice quality postoperatively with normal vocal fold mobility and was tolerating a normal diet without aspiration on the second postoperative day. She was discharged home on postoperative day 3 with a capped tracheotomy tube, and subsequently the tracheotomy tube was removed. The findings of a histologic examination of the surgical specimen were consistent with a saccular cyst. Videostroboscopic evaluation 2 weeks after the surgical procedure revealed a granulating tissue bed; at 1 month, a completely healed larynx with normal voice, airway, and swallowing function; and at 8 months, no evidence of recurrent disease (Figure 4). The patient remains symptom free without evidence of recurrent disease 16 months after her procedure.Figure 4.Postoperative videolaryngoscopic view of the larynx 8 months after complete endoscopic carbon dioxide laser excision of a right-sided saccular cyst.CASE 2A 42-year-old man was seen at another institution with a 6-month history of progressive dysphagia, hoarseness, and respiratory distress when lying supine. Fiberoptic laryngoscopy revealed a large mucosal-covered mass of the left false vocal and aryepiglottic folds that was obstructing the laryngeal inlet. The patient underwent a tracheotomy under local anesthesia for initial airway control, followed by marsupialization of a presumed saccular cyst. Postoperatively the left-sided laryngeal fullness persisted, and he subsequently underwent a second procedure to marsupialize the cyst. The left-sided supraglottic fullness recurred, and the patient was referred to the Detroit Medical Center, Harper Hospital, for treatment of a recurrent saccular cyst.A CT scan and videostroboscopy revealed a large 5×5-cm fluid-filled mass involving the left false vocal, aryepiglottic, and pharyngoepiglottic folds (Figure 5and Figure 6). The mass extended from the inferior border of the hyoid bone superiorly to the ventricle inferiorly. The lateral margin of the cyst rested against the inner thyroid perichondrium. Microlaryngeal endoscopic CO2laser (2 W) excision of the cyst was performed using a bivalve laryngoscope to provide visualization of the supraglottis (Figure 1). To provide better visualization for complete removal of the cyst, the superior two thirds of the wall were initially removed. The remaining anterolateral cyst wall was then completely excised. The pathology report was consistent with a laryngeal cyst. Postoperatively the patient had mild paresthesias and weakness of the left side of the tongue but tolerated a normal diet without signs of aspiration and had improved voice quality with normal vocal fold function. He was discharged home after 23 hours of observation, the cannula was removed after 2 weeks, and the patient had complete resolution of tongue paresthesias and weakness after 1 month. He remains symptom free with normal voice quality and swallowing function 8 months after the procedure (Figure 7).Figure 5.Axial computed tomographic scan revealing a 5×5-cm left-sided recurrent saccular cyst involving the false vocal, aryepiglottic, and pharyngoepiglottic folds.Figure 6.Videolaryngoscopic view of the larynx before excision of a left-sided saccular cyst.Figure 7.Postoperative videolaryngoscopic view of the larynx 6 weeks after complete endoscopic carbon dioxide laser excision of a left-sided saccular cyst.COMMENTThe saccule is a mucous membrane–lined pouch containing numerous mucus-secreting glands that communicates with the anterior portion of the roof of the ventricle and provides lubrication to the vocal folds. It rises vertically and is bounded by the false vocal fold medially, the base of the epiglottis anteriorly, and the inner surface of the thyroid cartilage laterally. Saccular cysts and laryngoceles are abnormal dilatations of the saccule. Saccular cysts have no communication with the laryngeal lumen and are mucus filled, whereas laryngoceles communicate with the laryngeal lumen and are typically air filled.A laryngocele may become temporarily distended with secretions, and if this becomes infected, a laryngopyocele is formed.DeSanto et alreviewed 238 cases of laryngeal cysts and formulated a classification system of 3 cyst types: ductal, saccular, and thyroid cartilage foraminal cysts. Ductal cysts accounted for 75% of all cysts noted in this study and were described as superficial mucosal lesions occurring anywhere in the larynx as a result of the retention of fluid in the collecting ducts or mucus glands. A saccular cyst type was found in nearly 25% of the cases and was described as a submucosal, mucus-filled dilatation of the laryngeal saccule due to blockage of the saccular orifice in the ventricle. There is only 1 case of a thyroid cartilage foraminal cyst reported in the literature, and this is a different disease from either the ductal or saccular cyst types.Saccular cysts can be further classified into anterior or lateral cysts.Anterior saccular cysts extend medially and posteriorly from the occluded opening of the saccule and protrude through the ventricle and into the laryngeal inlet between the true and false vocal folds. Lateral saccular cysts extend from the occluded opening of the saccule but submucosally penetrate superiorly and posteriorly into the false vocal, aryepiglottic, and pharyngoepiglottic folds.Laryngeal cysts account for approximately 5% of all benign laryngeal lesions.They occur most commonly in the sixth decade and no gender predominance exists. Saccular cysts may be asymptomatic and diagnosed as an incidental radiographic finding needing no further intervention, or the patient may have inspiratory stridor, possibly leading to sudden death due to asphyxiation.Progressive cough, dysphagia, hoarseness, dyspnea, and foreign body sensation are the most common presenting symptoms.Diagnosis is often made by flexible laryngoscopy combined with a CT scan, which provides excellent detail of these lesions with regard to location and size. The CT scan can also help differentiate a saccular cyst, which is typically fluid filled, from a laryngocele, which will be air filled.Saccular cysts may develop due to obstruction of the saccular orifice from squamous cell carcinoma. This is based on single case reports, however, and no series exists describing the incidence of saccular cyst in relation to laryngeal cancer. The relationship between laryngocele formation and squamous cell carcinoma of the larynx has been better documented, and the incidence varies between 5% and 28%.Based on these associations, a CT scan of the neck combined with direct laryngoscopy and possible biopsy is indicated to evaluate the possibility of coexistent laryngeal carcinoma.Standard treatment options for saccular cysts include aspiration of the cyst contents, endoscopic marsupialization of the dome of the cyst with laser ablation of the remaining cyst wall, or excision of the cyst by an external approach.Civantos and Holingerdescribed the treatment of saccular cysts in infants and children. They recommended complete endoscopic excision of the cyst if an initial attempt at aspiration failed. The authors emphasized that marsupialization of the cyst often resulted in recurrences and the need for repeated surgical excisions that often caused excessive glottic scarring and the possible development of subglottic stenosis. They recommended total excision of the cyst using a microlaryngoscopic technique and CO2laser ablation of any remaining cyst wall. Unfortunately, the endoscopic approach described offered only marginal visualization of the lesion, with the risk of leaving cyst remnant behind and therefore an increased risk of recurrent disease. Myssiorek and Perskydescribed the use of the CO2laser to marsupialize internal laryngoceles and saccular cysts, but again, visualization was likely marginal and recurrence a possibility because of the potential for leaving cyst wall remnants. After a failed endoscopic excision, Civantos and Holingerrecommend an external approach with excision of the entire cyst through the thyrohyoid membrane. More recently, Hogikyan and Bastianreported their results after complete endoscopic CO2laser excision of large or recurrent laryngeal saccular cysts in 7 adults, with follow-up periods ranging from 7 months to 2 years. They used a modified Bouchayer (Micro-France, Bourbon L' Archambault, France) operating laryngoscope to provide exposure of the cyst for all their cases, but noted that a bivalve laryngoscope was used in some instances.Coupling the microscope with the CO2laser and using the bivalve laryngoscope to provide superior visualization of the supraglottic larynx permit a more precise endoscopic removal of the entire cyst, without disruption of the cyst wall, little morbidity to endolaryngeal structures, no risk to the superior laryngeal nerve, and no external incisions. Removal of the entire cyst will likely decrease the incidence of recurrent disease and result in less overall morbidity.Endotracheal intubation with a small-caliber laser tube can be safely performed for patients with stable airways. The 2 patients in this study presented with impending airway obstruction, necessitating a tracheotomy for airway stabilization before removal of the cyst.Partial decompression of the cyst likely resulted in better visualization of, and therefore easier access to, the lesion for removal in case 1. Initial partial resection of the superior two thirds of the cyst in case 2 allowed for better visualization of the anterolateral extent of the cyst wall and therefore complete removal.Despite periodic relaxation of the bivalve laryngoscope and Lewy suspension apparatus during the procedure, patient 2 suffered mild paresthesias and weakness of the left side of the tongue postoperatively, which subsequently completely resolved. This complication is likely due to the force of tongue retraction produced by both the bivalve laryngoscope and Lewy suspension apparatus, resulting in a stretch injury (neurapraxia) of the lingual and hypoglossal nerves. Although the bivalve laryngoscope gives wide exposure of the supraglottic larynx, the risk of lingual or hypoglossal nerve injury should be discussed with patients.Both patients had normal voice quality and swallowing function after endoscopic CO2laser excision of their saccular cysts. They were discharged home on normal diets, the cannulas were removed shortly after discharge, and they remain disease free.DMyssiorekMPerskyLaser endoscopic treatment of laryngoceles and laryngeal cysts.Otolaryngol Head Neck Surg.1989;100:538-541.JBBoothHGBirckOperative treatment and postoperative management of saccular cyst and laryngocele.Arch Otolaryngol.1981;107:500-502.JCSniezekREJohnsonSGRamirezDKHayesLaryngoceles and saccular cysts.South Med J.1996;89:427-430.RFWardJJonesJAArnoldSurgical management of congenital saccular cysts of the larynx.Ann Otol Rhinol Laryngol.1995;104:707-710.FCivantosLHolingerLaryngoceles and saccular cysts in infants and children.Arch Otolaryngol Head Neck Surg.1992;118:296-300.JShapiroSMZeitelsMPFriedLaser surgery for laryngeal cancer.Operative Technique Otolaryngol Head Neck Surg.1992;3:84-92.NDHogikyanRWBastianEndoscopic CO2laser excision of large or recurrent laryngeal saccular cysts in adults.Laryngoscope.1997;107:260-265.LWDeSantoKDDevineLHWeilandCysts of the larynx.Laryngoscope.1970;80:145-176.BHNewmanJBTaxyHILakerLaryngeal cysts in adults.Am J Clin Pathol.1984;81:715-720.MADadaLaryngeal cyst and sudden death.Med Sci Law.1995;35:72-74.CMicheauBLuboinskiPLanchiYCachinRelationship between laryngoceles and laryngeal carcinomas.Laryngoscope.1978;88:680-688.Accepted for publication July 25, 1997.Corresponding author: Robert J. Meleca, MD, Department of Otolaryngology–Head and Neck Surgery, Wayne State University School of Medicine, 4201 St Antoine Ave, 5E UHC, Detroit, MI 48201.
TI - Laryngeal Obstructing Saccular Cysts
JF - JAMA Otolaryngology - Head & Neck Surgery
DO - 10.1001/archotol.124.5.593
DA - 1998-05-01
UR - https://www.deepdyve.com/lp/american-medical-association/laryngeal-obstructing-saccular-cysts-BcWza46zxT
SP - 593
EP - 596
VL - 124
IS - 5
DP - DeepDyve
ER -