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| CASE REPORT |
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| Year : 2021 | Volume
: 11
| Issue : 2 | Page : 29-32 |
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A novel approach to upper airway cyst in infants: A case series
Rakesh Srivastava
ENT Department, Sushrut Institute of Plastic Surgery, Super Specialty Hospital, Lucknow, Uttar Pradesh, India
| Date of Submission | 20-Dec-2020 |
| Date of Acceptance | 09-Jul-2021 |
| Date of Web Publication | 12-Apr-2022 |
Correspondence Address:
Rakesh Srivastava
ENT Department, Sushrut Institute of Plastic Surgery and Super Specialty Hospital, Lucknow
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jlv.jlv_21_20
 Abstract | | |
There are various anecdotal reports and case series in the literature on congenital laryngeal cyst. These studies described pathology, presentation, and management of these cysts. The current case series is 2-year retrospective analysis of symptomatology and therapeutic difficulties on seven infants with laryngeal cysts. All the babies presented with moderate-to-severe stridor. Vallecular and base of tongue cyst are the most common sites. Flexible laryngobronchoscopy remains the main diagnostic test. The management initiated by the preoperative use of high-flow nasal cannula and followed by decompression (aspiration) and deroofing. Deroofing done by using the various energy sources such as coblation and lasers (carbon dioxide and diode). Coblation is faster, easier to use with no risk of fire, and bleeding.
Keywords: Pediatric laryngeal cyst, infant, high flow nasal cannula
How to cite this article:
Srivastava R. A novel approach to upper airway cyst in infants: A case series. J Laryngol Voice 2021;11:29-32 |
| Introduction | |  |
Laryngeal cysts were first described by Verneuil in 1852. Subsequently, these were reported by Abercrombie in 1881.[1] Congenital laryngeal cysts are rare clinical entities, with an incidence of 1.82/100,000 live births in the Hong Kong.[2] Prowse and Knight reported the incidence of congenital laryngeal cysts 3.49 cases/100,000 live births over a 10-year period (1.40/100,000 for saccular cyst and 2.09/100,000 live births for ductal cysts).[3]
These cysts are found at various sites starting from the base of the tongue, vallecula, aryepiglottic (AE) fold, epiglottis, and saccule of the ventricle. Despite the different anatomical locations of these extralaryngeal cysts, they are covered under the broader heading of laryngeal cysts as they cause airway obstruction by the displacement of different portions of the supraglottic larynx.
Early classification was based on the location of the lesion within the oropharynx/larynx. The term mucous or retention cyst appears regularly in various classifications as they develop from dilatation of the mucous gland after obstruction of its duct. Depending on the site and size of the cyst, it distorts the larynx and pharynx and presents with various symptoms.
The first classification of cystic laryngeal lesions was described by DeSanto et al.[4] The authors divided all laryngeal cysts into saccular, ductal, and thyroid cartilage foraminal cysts. Arens et al. looked at all laryngeal cysts with respect to location and histomorphology. They subsequently divided laryngeal cysts into congenital, retention, and inclusion cysts.[5] Ductal cyst was considered to be caused by the obstruction of submucosal ducts, whereas saccular cyst was caused by the obstruction of saccule of the ventricle.
The common symptoms of these cysts are difficulty in breathing (stridor), cyanotic spells, poor feeding or coughing while taking milk, change in cry/voice, and failure to thrive.[3]
Vallecular cyst sometimes originates from the base of the tongue. They are unilocular arising from any surface of the vallecula. The cyst wall lined by the respiratory epithelium with mucous glands and retained secretions. The origin may be from vallecula, base of the tongue, epiglottis, or AE fold.
Larger sized cysts are potentially dangerous. They compress the epiglottis or AE fold causing upper airway obstruction. Anesthesia in such situation is challenging due to limited visualization of laryngeal inlet. The clinical manifestations of laryngeal cyst are moderate-severe respiratory distress, difficulty in swallowing, and failure to thrive. Differential diagnosis includes hemangioma, lingual thyroid, cystic hygroma, hamartoma, dermoid cyst, teratoma, and thyroglossal cyst.
Wigger and Tang first described subglottic cyst (SGC). These lesions occur almost exclusively in premature infants with a history of endotracheal intubation with an estimated incidence of 1.9/100,000 births.[2],[6]
A SGC originates from the obstruction of mucous glands due to epithelial fibrosis and squamous metaplasia in the healing process. Subepithelial fibrosis and squamous metaplasia can obstruct the ducts of the mucous glands leading into the formation of retention cysts.[7]
| Materials and Methods | | |
This is a retrospective study on seven cases of upper airway cysts in infants from 2017 to 2019. All the cases were referred by the pediatrician with noisy breathing and failure to thrive. Flexible bronchoscopy was done in all cases. Imaging included was HRCT scan (Plain/Contrast) in five cases [Figure 1]a and [Figure 1]b. Imaging not done in two cases who were already intubated. Thyroid scan and thyroid profile were done in one case with the base of tongue cyst. | Figure 1: (a) (Axial), (b) (Coronal) Contrast-enhanced computed tomography scan show vallecular cyst
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Every case discussed with the anesthetist regarding preoperative planning. All the babies were first kept on high-flow nasal cannula (HFNC) at 8 L/min for 10–15 min with head end of operating table elevated to 30°. Premedication done after 15 min of high flow by injection Propofol (2–3 mg/kg) and injection fentanyl (1–2 μg/kg). Oropharynx sprayed with 10% xylocaine spray (Atomizer) and exposure of oropharynx done by the anesthetist using MacIntosh laryngoscope. Surgeon (RS) visualized cyst with 30° nasal endoscope and the aspiration of the cyst done using 18G lumber puncture needle which was curved near the distal third. Decompression and reduction in the size of the cyst improved the airway exposure. Complete procedure done under total intravenous anesthesia with endotracheal tube in the position except in SGC case. Various energy sources were used for deroofing of the cyst. Carbon dioxide (CO2) ((Lumenis Acupulse, Tel aviv) laser was used to deroof SGC [Figure 2]a and [Figure 2]b. Other energy sources used were Coblation (Smith and Nephew Healthcare limited) with Evac70 wand was used for AE fold and vallecula cyst dissection [Figure 3]a and [Figure 3]b, and Diode Laser (angiodynamics, diomed) for base of tongue tumor (BOT) cyst. HFNC was used during the extubation phase for 10–15 min with gradual tapering of oxygen flow. | Figure 2: Subglottic cyst (a – Aspiration of subglottic cyst; b – Multiple cyst; c – Carbon dioxide laser deroofing)
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 | Figure 3: (a) Aspiration of vallecular cyst; (b) Deroofing using coblator
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| Results | | |
The most common findings in all babies were moderate-to-severe stridor [Table 1]. Two babies were referred from other hospital in intubated condition (Huge AE fold cyst, vallecular cyst) [Figure 4]a and [Figure 4]b. Flexible bronchoscopy helped to confirm the size, site, consistency of the cyst, and any other synchronous lesions. The following chart shows the age of presentation, site of cyst, symptoms, aspiration, procedure, energy source, and duration of procedure. | Table 1: Site of cyst, symptoms, aspiration of cyst, surgery, energy source, and time duration in surgery
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[Table 1] shows site of cyst, symptoms, aspiration of cyst, surgery, energy source, and time duration in surgery.
Other symptoms were failure to thrive and coughing while feeding. The preoperative use of HFNC, local anesthetic spray, and intravenous sedation helped in easy decompression of cyst. Coordination among the anesthesia and surgeon team is important in such cases. Deroofing was done by CO2 laser (lumenis acupulse, Telaviv) using the scanner mode in three cases [Figure 2]c. Three cases were done by Coblator and one case was done by diode laser. Laryngomalacia (Type-3) was observed in one case with severe retroflexion of suprahyoid epiglottis which was disproportionate to the size of BOT cyst. This patient was treated by deroofing of cyst with epiglottopexy using Lichtenberger needle carrier. Suture (2/0 vicryl round body) was used which was tightened and buried under the neck skin [Figure 5]a and [Figure 5]b.
SGC was secondary to the history of intubation for lower respiratory infection for 3 days. Following extubation, baby presented with respiratory distress after 3 weeks. One baby was preterm and twins presented with right AE fold cyst. The baby was having coughing on taking milk with positional stridor.
Recurrence was noticed in one case with huge right AE fold cyst. The recurrence happened within 3 weeks of primary surgery. The cyst was again deroofed with the removal of redundant mucosa using CO2 laser. The average time taken for the surgery from aspiration to completion was the highest with the CO2 laser [Table 1]. The reasons are adjustment of the laser beam, protection of surrounding structures from fire, and angulation of beam, especially in BOT/vallecula cyst.
In the immediate postoperative period, all patients were given antibiotics, steroids, and proton-pump inhibitor (Lansoprazole 1 mg/kg) or H2 blocker (Ranitidine syrup 2 mg/kg). Nebulization with budesonide and ipratropium bromide twice a day for 5 days in SGC case.
| Discussion | | |
Laryngeal cysts are among the rare congenital anomalies in infants, largely treated as laryngomalacia in the pediatric practice. An average delay of 1 or 2 months noticed from the onset of symptoms to the diagnosis. Airway gets compromised due to pressure of cyst over the supraglottic larynx. All cases in the present series presented with respiratory distress. Stridor was the most common presenting complaint in 70% of cases proven in a study by Prowse and Knight followed by coughing, cyanotic episodes, poor feeding, voice change, and failure to thrive.[3]
Associated laryngomalacia in one case was apparent due to severe retroflexion of suprahyoid epiglottis with each inspiration during flexible bronchoscopy. It was difficult to diagnose floppiness of the epiglottis during perioperative period.
Surgeon (RS) and his team of anesthetists are using HFNC in almost all cases of pediatric airway intervention routinely.[8] HFNC improves the apnea time. Combination of local anesthetic spray, mild intravenous sedation, rapid response, and aspiration of the cyst make intubation easier for the anesthetist. The use of HFNC during SGC dissection gives free field for the instrumentation. Aspiration of cyst to reduce the size of cyst for easy intubation and removal of cyst was done by Lee et al.[1]
The classical procedure of marsupialization of the cyst is not possible in airway cysts due to narrow area of dissection and suturing difficulties. Deroofing using various energy devices such as laser, coblator, or even cold instruments was quite effective. The respiratory epithelium of the cyst wall was burned, and redundant mucosa of the collapsed cyst was removed. Coblation is faster due to ease of use, better visibility, angulation of wand, two hand technique, no risk of fire, and bleeding. Coblation wand can be bent according to the site of dissection. It ablates the tissue by generating the field of ionized sodium molecules at around 40°C–70°C. It results in less tissue trauma, minimal bleeding, and better postoperative recovery.[9] The duration of surgery was more with CO2 laser due to time required in adjustment of the laser beam, protection of surrounding structures from fire, and angulation of beam, especially in BOT/Vallecula cyst.
SGC is seen more often in neonate and infant with a history of endotracheal intubation. Difficulty in breathing usually starts after few weeks of extubation and sometimes after few months. SGC reported with the shortest duration of 1 day intubation history was also reported in the literature.[10] The largest retrospective study of SGC cases were reported from Great Ormond Street hospital in 2002. All patients had a history of intubation ranging from 1 to 126 days (average 10 days). SGC is most commonly seen in preterm babies. Various modalities of instrumentation including CO2 laser have been used with reported recurrence rate of 43% on follow-up bronchoscopy between 3 and 6 months.[11]
Detailed evaluation of SGC with rigid scope using Lindholm Benjamine laryngoscope was done by Aksoy et al. (2012) on nine patients. It was observed that SGC was multiple in all cases and located over the posterior and lateral wall. Multiple cyst in subglottis was also noticed in the present series [Figure 2]b. They were treated with aspiration followed by endoscopic deroofing using CO2 laser and balloon tracheoplasty. In Aksoy et al. series, three cases had a history of intubation, and in the remaining six cases, the cause might have been gastroesophageal reflux.[12] Ransom et al. used Microdebrider for SGC in eight cases with recurrence in two patients.[13]
In his study on eight cases of vallecular cyst, Pak et al. performed complete excision in four cases and deroofing in remaining four cases.[2] In the present case series, severe respiratory distress and difficult laryngeal exposure prevented complete excision of the cyst. Securing the airway was prime consideration under such condition.
| Conclusion | | |
Laryngeal cysts in infants are relatively rare. They are detected late in India due to delayed diagnosis. The decompression of the cyst under HFNC with mild sedation was found to be relatively safe technique in such cases. Coordination among operating and anesthesia team is of paramount importance during intubation. Various energy sources were used with similar efficacy.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Lee WS, Tsai CS, Lin CH, Lee CC, Hsu HT. Airway obstruction caused by a congenital epiglottic cyst. Int J Pediatr Otorhinolaryngol 2000;53:229-33.  |
| 2. | Pak MW, Woo JK, van Hasselt CA. Congenital laryngeal cysts: Current approach to management. J Laryngol Otol 1996;110:854-6. |
| 3. | Prowse S, Knight L. Congenital cysts of the infant larynx. Int J Pediatr Otorhinolaryngol 2012;76:708-11. |
| 4. | DeSanto LW, Devine KD, Weiland LH. Cysts of the larynx--Classification. Laryngoscope 2015;125:2629. |
| 5. | Arens C, Glanz H, Kleinsasser O. Clinical and morphological aspects of laryngeal cysts. Eur Arch Otorhinolaryngol 1997;254:430-6. |
| 6. | Wigger HJ, Tang P. Fatal laryngeal obstruction by iatrogenic subglottic cyst. J Pediatr 1968;72:815-20. |
| 7. | Hawkins DB. Hyaline membrane disease of the neonate prolonged intubation in management: Effects on the larynx. Laryngoscope 1978;88:201-24. |
| 8. | Srivastava R, Agarwal AC, Agarwal S, Pathak M. High flow nasal cannula: A game changer in airway surgery. Indian J Otolaryngol Head Neck Surg 2019;71:299-303. |
| 9. | Gogia S, Agarwal SK, Agarwal A. Vallecular cyst in neonates: Case series-a clinicosurgical insight. Case Rep Otolaryngol 2014;2014:764860. |
| 10. | Chigicherla S, Tiwari S, Sharma RD, Nathani R. Subglottic cyst causing upper airway obstruction. Int J Otorhinolaryngol Head Neck Surg 2020;6:1203-5. |
| 11. | Lim J, Hellier W, Harcourt J, Leighton S, Albert D. Subglottic cysts: The Great Ormond Street experience. Int J Pediatr Otorhinolaryngol 2003;67:461-5. |
| 12. | Aksoy EA, Elsürer C, Serin GM, Unal OF. Evaluation of pediatric subglottic cysts. Int J Pediatr Otorhinolaryngol 2012;76:240-3. |
| 13. | Ransom ER, Antunes MB, Smith LP, Jacobs IN. Microdebrider resection of acquired subglottic cysts: Case series and review of the literature. Int J Pediatr Otorhinolaryngol 2009;73:1833-6. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1]
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