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Table of Contents
CASE REPORT
Year : 2011  |  Volume : 1  |  Issue : 1  |  Page : 27-29

Congenital vallecular cyst with laryngomalacia: A report of two cases


1 Department of Laryngology, Deenanath Mangeshkar Hospital, Pune, India
2 Department of Laryngology, Deenanath Mangeshkar Hospital, Pune; Department of ENT, Karnataka Institute of Medical Sciences, Hubli, India
3 Department of Laryngology, Bharati VidyaPeeth Medical College, Pune, India

Date of Web Publication7-Feb-2011

Correspondence Address:
Sachin Gandhi
Department of Laryngology, Deenanath Mangeshkar Hospital, Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2230-9748.76134

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   Abstract 

Vallecular cyst in an infant is uncommon, and it finds infrequent mention in the literature as well. However when present, it accounts for severe inspiratory stridor, causing airway compromise, and sometimes even death. Laryngomalacia is the commonest congenital anomaly of the larynx, which is present after birth giving rise to inspiratory stridor. Laryngomalacia results as a collapse of supraglottic structures of the larynx. Clinical co-presentation of vallecular cyst and laryngomalacia is very rare. This is a report of two cases of 2-month-old infants who presented with the history of noisy breathing and refusal to feeds since 10-15 days. CT scan of the neck revealed a huge cyst in the vallecula compromising the airway. Microlaryngoscopic-guided aspiration and marsupilization of the cyst was done using CO 2 laser and the release of aryepiglottic fold was done using microlaryngeal scissors. Respiratory and feeding problems dramatically improved in these patients by immediate surgical intervention.

Keywords: Vallecular cyst, laryngomalacia, CO 2 laser, aryepiglottopexy


How to cite this article:
Gandhi S, Raza S A, Thekedar P, Mishra P. Congenital vallecular cyst with laryngomalacia: A report of two cases. J Laryngol Voice 2011;1:27-9

How to cite this URL:
Gandhi S, Raza S A, Thekedar P, Mishra P. Congenital vallecular cyst with laryngomalacia: A report of two cases. J Laryngol Voice [serial online] 2011 [cited 2020 Jul 9];1:27-9. Available from: http://www.laryngologyandvoice.org/text.asp?2011/1/1/27/76134


   Introduction Top


Vallecular cyst is a rare but well recognized cause of stridor and death in newborns and infants. [1] Though laryngomalacia is the most common congenital anomaly of the larynx, clinical copresentation of vallecular cyst and laryngomalacia is very rare. [2] We herein report two cases of infant with a vallecular cyst with laryngomalacia referred from the pediatric ICU to the department of Laryngology.


   Case Reports Top


Case 1

A 2-month-old infant referred from PICU to the Department of Laryngology, of this tertiary care hospital, with the history of noisy breathing and feeding difficulty since 15 days. There was no history of fever, cough, or choking. The child was born after full-term normal delivery with a birth weight of 3 kg. On examination, the child had inspiratory stridor. There was tachypnoea with a respiratory rate of 48 per minute and in-drawing of suprasternal notch. The accessory muscles of respiration were active and there was in-drawing of xiphi-sternum and intercostal spaces. Computed tomogram of the neck revealed a well-defined cystic lesion measuring 3×2 cm occupying the vallecula, pushing the epiglottis downward causing airway compromise [Figure 1].
Figure 1: CT Scan showing the presence of vallecular cyst.

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The patient was taken up for microlaryngoscopy under general anesthesia. We had difficulty in intubating the patient as the cyst was obscuring the view of the laryngeal inlet. The cyst was aspirated using a wide bore needle which revealed mucoid material. This facilitated the visualization of the endolarynx [Figure 2]. Under rigid telescopic guidance, the endolarynx was visualized and the patient was intubated with high-frequency jet ventilation. Marsupialization of the cyst was done using CO 2 laser. Division of the aryepiglottic folds using microlaryngeal scissors was done at the same sitting for laryngomalacia. The patient tolerated the procedure well and was under observation for 24 h with endotracheal tube in situ. The stridor gradually subsided over a period of 48 h. Culture from the aspirate revealed Klebsiella pneumoniae sensitive to ofloxacin and levofloxacin.
Figure 2: (a) Vallecular cyst (b) aspiration of vallecular cyst (c) marsupilization of the cyst.

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Case 2

A 2-month-old infant was referred from PICU with history of inspiratory stridor, choking sensation and difficulty in feeding since 10 days. Child was the first child in the family, was born after a full-term normal delivery with a birth weight of 3.1 kg. On examination, the child had pallor, tachycardia, with respiratory rate of 64/min. There was in-drawing of suprasternal notch and intercostal spaces. Clinical diagnosis of laryngomalacia was made and the patient was taken up for microlaryngoscopy under general anesthesia. Examination of the endolarynx revealed vallecular cyst compromising the airway. Aspiration of the cyst was done using a wide bore needle and it was filled with thick mucoid material. Marsupialization of the cyst was done using CO 2 Laser. Aryepiglottopexy was not done at the same sitting as there was edema [Figure 3]. While intubated patient was kept on systemic steroids. The child was extubated after 24-36 h, but the child was not maintaining oxygen saturation at repeated intervals. Later division of the aryepiglottic fold using microlaryngeal scissors was done and the child was kept under observation for 24 h with endotracheal tube in situ and then weaned off endotracheal tube and oxygen. The patient tolerated the procedure well. Culture of the mucoid aspirate revealed no organisms.
Figure 3: Marsupilization of the cyst.

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   Discussion Top


Vallecular cyst with laryngomalacia is very rare and this association is a concomitant finding according to some. Retention cysts of the larynx are squamous or columnar; both forms originate from obstructed seromucinous salivary glands. The squamous variant is common on the lingual surface of epiglottis, valleculae, and on the aryepiglottic fold. Vallecular cyst causes airway obstruction by mass effect in the hypopharynx and also by inferior and posterior displacement of epiglottis leading to supraglottic obstruction. [3] This is particularly significant when vallecular cyst is associated with laryngomalacia where the airway collapse contributed by the negative airway as a result of obstruction gets exacerbated particularly during inspiration. [4]

These cysts may reach a considerable size before being diagnosed, but minor cysts are often incidental finding at routine laryngological examination. Antenatal diagnosis using ultrasound scan at around 28 weeks gestations is possible, which may help clinician to plan for a high-risk delivery at a tertiary care center where facilities for emergency tracheostomy or surgery and ventilation of neonate are available.[5]

After birth it is important to take a careful history targeted to identify situations or positions that worsen or improve obstructive symptom, which helps in differentiating cyst from laryngomalacia. However history may not be the only relying factor in such cases. Computed tomography is essential to determine the cyst size, location, contents, and to plan optimal route of passage of the endotracheal tube. Role of fiber optic evaluation has been mentioned in certain studies to evaluate and to assess for any other associated pathology; however both of our patients had severe stridor and so it was not considered safe to take them up for fiber optic evaluation. Instead they were taken up for direct laryngoscopic evaluation. [6] It is essential to distinguish solid and cystic lesion as cystic lesions may rupture at the time of intubation.

When present in neonate/infancy period, they account for severe inspiratory stridor and failure to thrive. This increases the morbidity and mortality of the disease and hence needs to be tackled on an emergency basis. Marsupialization of the cyst was done using CO 2 laser to prevent recurrence. Aryepiglottopexy was done for laryngomalacia using microlaryngeal scissors. A study by Ku in 2000 of four cases supports the fact that use of CO 2 laser is an efficient tool for vallecular cyst marsupialization. However in his series, he had only one case with coexistent laryngomalacia, which improved after removal of the cyst. [4] There are some reports on literature in which the stridor of the patient with coexistent vallecular cyst and laryngomalacia improved just after vallecular cyst excision. [7],[8] In the first case reported here, after the marsupialization of vallecular cyst, very severe form of laryngomalacia was seen and so aryepiglottopexy was done in same sitting. While in the other case, the severity of laryngomalacia was not very high so a trial of extubation was given after the cyst excision; however the child did not maintain proper saturation when extubated, so in second sitting aryepiglottopexy was done and child was symptom-free after that. We are of the firm belief that marsupilization along with aryepiglottopexy is the ideal treatment of choice in such cases in where there is severe form of laryngomalacia. The use of high-frequency jet ventilation via small canula is an excellent means of anesthesia to overcome the problems of intubation when compared to routine endotracheal tubes, as the latter are usually thick and interfere with the surgery.


   Conclusion Top


Vallecular cyst with laryngomalacia is a rare entity, which affects mainly infants. Timely intervention ensures relief of compromised airway thereby avoiding possible tracheostomy. Use of CO 2 laser is justified as it facilitates removal of the cyst, prevents edema, and recurrence. High-frequency jet ventilation is an added advantage to overcome the problems of intubation especially in pediatric age group and pediatric surgeries related to larynx and the airway. During the follow-up period up to 6 months, both the patients were free from stridor and had good weight gain.

 
   References Top

1.Gutierrez JP, Berkowitz RG, Robertson CF. Vallecular cysts in newborns and young infants. Pediatr Pulmonol 1999;27:282-5.  Back to cited text no. 1
    
2.Wong KS, Li HY, Huang TS. Vallecular cyst synchronous with laryngomalacia: Presentation of two cases. Otolaryngol Head Neck Surg 1995;113:621-4  Back to cited text no. 2
    
3.LaBagnara 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  Back to cited text no. 3
    
4.Ku AS. Vallecular cyst: Report of four cases--one with co-existing laryngomalacia. J Laryngol Otol 2000;114:224-6.  Back to cited text no. 4
[PUBMED]    
5.Cuillier F, Samperiz S, Testud R, Fossati P. Antenatal diagnosis and management of Vallecular cyst. Ultrasound Obstet Gynecol 2002;20:623-6.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Chow PY, Nag DK, Poon G, Hui Y. Vallecular cyst in a neonate. Hong Kong Med J 2002;8:464.  Back to cited text no. 6
    
7.Yao TC, Chiu CY, Wu KC, Wu LJ, Huang JL. Failure to thrive caused by the coexistence of vallecular cyst, laryngomalacia and gastroesophageal reflux in an infant. Int J Pediatr Otorhinolaryngol 2004;68:1459-64.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Sands NB, Anand SM, Manoukian J. Series of congenital vallecular cysts: A rare yet potentially fatal cause of upper airway obstruction and failure to thrive in the newborn. J Otolaryngol Head Neck Surg 2009;38:6-10.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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