IJAIMS

Anil Gangwar et al 10.5005/jp-journals-10050-10030 Case Report

A Rare Case of Vallecular Cyst 1Anil Gangwar, 2Vaibhav Kuchhal, 3Priyanshu Srivastava

ABSTRACT Endoscopic laryngoscopy is necessary to visual- Vallecular cyst is an uncommon but potentially dangerous ize vallecular cyst when suspected clinically. Other condition causing stridor and has been associated with sudden conditions leading to neonatal stridor should be ruled resulting in death due to its anatomical out, such as and other laryngotracheal location in neonates. It may also present with feeding problems abnormalities. Marsupialization with a CO2 laser is the resulting in failure to thrive. Endoscopic laryngoscopy is 1 necessary to visualize vallecular cyst when suspected most successful treatment. clinically. Other conditions leading to neonatal stridor should be ruled out, such as laryngomalacia and other laryngotracheal CASE REPORT abnormalities. Marsupialization with a CO2 laser is the most successful treatment. We report a case of a 6-month-old A 6-month-old male infant was referred to the ear, nose male infant referred to our ear, nose and throat department of and throat (ENT) department of Dr. Susheela Tiwari Dr. Susheela Tiwari Hospital, Government Medical College, Hospital, Government Medical College, Haldwani, for Haldwani, for the evaluation of inspiratory stridor, feeding the evaluation of inspiratory stridor, feeding difficulty, difficulty, suprasternal retraction, and failure to thrive. The patient was misdiagnosed and treated conservatively by a suprasternal retraction, and failure to thrive. pediatrician. On rigid laryngoscopic examination, an anteriorly He was born at 38 weeks of gestation by vaginal deliv- displacing cystic mass on the tongue base on the left side was ery at a local hospital, weighing 2.10 kg, without perinatal detected. The cyst was removed intoto using bipolar cautery. problem. There was hoarseness when he cried, but he did Immediately after surgery, inspiratory stridor, suprasternal retraction, and feeding difficulty improved gradually. One week not have significant respiratory distress. After 4 months later, follow-up rigid laryngoscopy showed no abnormal finding he developed noisy breathing, and some days later stridor except episodic influx of . developed. The patient’s father consulted the pediatri- Keywords: Laryngomalacia, Laryngoscopy, Lymphadenopathy, cian of a private hospital and treatment started along the Vallecular cyst. lines of and the patient was shifted to the pediatric intensive care unit. After 2 weeks of treatment, How to cite this article: Gangwar A, Kuchhal V, Srivastava P. A Rare Case of Vallecular Cyst. Int J Adv Integ Med Sci the patient was discharged. Few weeks later, the patient 2016;1(2):88-90. again developed stridor. He was brought to the emergency room and then admitted to the neonatal intensive care Source of support: Nil unit for treatment. After nasal continuous positive airway Conflict of interest: None pressure (CPAP) support, severe respiratory distress was relieved. However, symptoms, such as hoarseness, noisy INTRODUCTION breathing, and feeding difficulty persisted. Vallecular cyst is a rare but potentially dangerous cause He was evaluated with several studies, including of stridor in neonates and infants.1 Vallecular cyst is chest computed tomography, but there was no abnor- usually asymptomatic or there are only subtle symptoms, mal finding present. Ten days after weaning from nasal such as voice change or a lump in the throat. In contrast, CPAP, there was a sudden desaturation episode again, cysts may lead to stridor and/or respiratory distress in which was relieved by positive pressure ventilation via neonates and young infants because of their relatively manual bag masking. For evaluation of upper airway, small airway and cause severe airway obstruction and he was transferred to our department. On admission, even death.1 It also has the potential to excite the retching his body weight was 2.10 kg, height was 49 cm, and head reflex, which may induce gagging and vomiting. They circumference was 36 cm. His body temperature was leads to failure to thrive.1 37.1°C, pulse rate was 172/min, respiratory rate was 41/ min, and blood pressure was 74/42 mm Hg. Conjunctivae and sclerae were within normal limits. The and 1,3Resident, 2Professor and Head the oral cavity were also normal. No lymphadenopathy 1-3Department of ENT, Government Medical College, Haldwani or any mass was detected. The chest expansion Uttarakhand, India was symmetrical, but there was inspiratory stridor and Corresponding author: Anil Gangwar, Resident, Department suprasternal retraction. Heart beats were regular without of ENT, Government Medical College, Haldwani, Uttarakhand any audible murmur. The abdomen was soft and flat with India, Phone: +919997357812, e-mail: [email protected] normal bowel sound. Liver and spleen were not palpable. 88 IJAIMS

A Rare Case of Vallecular Cyst

Fig. 1: Magnetic resonance imaging neck Fig. 2: During cauterization

Fig. 3: Excised specimen Fig. 4: Histopathology

There was no rash or petechiae on skin exam. All cranial Immediately after surgery, inspiratory stridor, supra- nerve function were intact. sternal retraction, and feeding difficulty improved gradu- The results of laboratory test were hemoglobin ally. One week later, follow-up rigid laryngoscopy showed 12 gm/dL, white blood cell count 7000/μl with a normal no abnormal finding except episodic influx of epiglottis. differential cell count, and platelet count 410,000/μl. The He was discharged and follow-up was done in the outpa- chest X-ray was normal. On rigid laryngoscopic examina- tient clinic. He was healthy at home, had no airway and tion, an anteriorly displacing cystic mass on the tongue feeding problems, and gained weight steadily. base left side was detected. Magnetic resonance imaging (MRI) neck was done, which showed retention cyst Histopathological Report about 1.6 × 1 × 1.2 cm in anteroposterior, transverse, and Histopathologic examination showed a cyst lined by superoinferior dimensions in the left vallecula insinuat- squamous epithelium with chronic active inflammation ing into the left base of the tongue, medially abutting the (hematoxylin and eosin, × 40) (Fig. 4). epiglottis (Fig. 1). Under sedation, laryngoscopy confirmed the cystic DISCUSSION mass between the base of the tongue and the epiglottis. Intubation was tried by an anesthesiologist, but the tube Laryngeal cysts are a uncommon cause of stridor in was not negotiable. Cyst was aspirated first and then infants. They are more commonly seen in adults. They intubation was done. After that, cyst was removed using have been classified based on location, histology, size, bipolar cautery (Figs 2 and 3). After cautery, the position and contents. The vallecula is the depression behind of the epiglottis was recovered. Removed specimen was the base of the tongue between the median and lateral sent for histopathological examination. epiglottic folds on each side. Vallecular cysts belong to International Journal of Advanced & Integrated Medical Sciences, April-June 2016;1(2):88-90 89 Anil Gangwar et al the category of ductal cysts described in DeSanto’s early Simple aspiration of the cyst is not advised because of its classification system of laryngeal cysts.2 They classified high recurrence rate.8 laryngeal cysts into ductal, saccular, and thyroid forami- In our case, vallecular cyst was diagnosed in a nal subtypes. Ductal cysts are caused by mucus retention 6-month-old male infant with symptoms of noisy respira- in the submucosal collecting ducts.2 Vallecular cysts may tion, recurrent upper infection (URTI). contribute to upper airway obstruction by posterior dis- Patient was misdiagnosed and treated conservatively by placement of the supraglottis. pediatrician. Clinical presentations of vallecular cysts include stridor, feeding problems, failure to thrive, gastroesopha- CONCLUSION geal reflux, apnea/cyanosis, chest retractions, hoarse When an infant presents with noisy breathing or stridor, cry, as well as respiratory distress and life-threatening a complete ENT examination is required to rule out 3 Laryngomalacia can coexist with vallecular events. upper airway abnormalities. Awareness and a high level cysts.1 Diagnosis is by direct laryngoscopy, though of suspicion are required for a quick diagnosis of such a imaging studies, particularly MRI, can provide impor- lesion as it can predispose an infant to recurrent URTI, tant information.4 Management is by transoral surgical feeding problems, respiratory distress, or failure to thrive. removal or marsupialization. About 60% of children with stridor have laryngeal REFERENCES obstruction, such as laryngomalacia, vocal cord paraly- 1. Yao TC, Chiu CY, Wu KC, Wu LJ, Huang JL. Failure to thrive sis, , hemangioma, or laryngeal cysts; caused by the coexistence of vallecular cyst, laryngomala- 25% have lesions in the upper airway, including choanal cia and gastroesophageal reflux in an infant. Int J Pediatr atresia, macroglossia, facial anomalies; 15% are due to Otorhinolayngol 2004 Nov;68(11):1459-1464. tracheobronchial lesions, such as or vas- 2. DeSanto LW, Devine KD, Weiland LH. Cysts of the cular compression, and others.5,6 In 1881, Abercrombie classification. Laryngoscope 1970 Jan;80(1):145-176. provided the first description of a laryngeal cyst.7 Mitchell 3. LaBagnara J. Cysts of the base of the tongue in infants: an unusual cause of neonatal airway obstruction. Otolaryngol et al in 1987, published the largest single series consisting Head Neck Surg 1989 Jul;101(1):108-111. of 20 cases experiencing of the Hospital for Sick Children, 4. Tuncer U, Aydogan LB, Soylu L. Vallecular cyst: a cause of London over a 15-year period.8 In 1980, Holinger reported failure to thrive in an infant. Int J Pediatr Otorhinolaryngol on 219 children presenting with stridor to two Chicago 2002 Sep;65(2):133-135. pediatric units over four years. Two of these cases (0.9%) 5. Wong KS, Li HY, Huang TS. Vallecular cyst synchronous with laryngomalacia: presentation of two cases. Otolaryngol were due to laryngeal cysts.9 A total of 12 to 45% of Head Neck Surg 1995 Nov;113(5):621-624. laryngomalacia presents with other associated airway 6. Wang W, Tovar JA, Eizaguirre I, Aldazabal P. Airway obstruc- abnormality, such as laryngeal cyst.5 Thus, in neonatal tion and gastroesophageal reflux: an experimental study stridor, evaluation of the airway anatomy and differential on the pathogenesis of the association. J Pediatr Surg 1993 diagnosis from other causes of stridor are important to Aug;28(8):995-998. 7. Gluckman PG, Chu TW, van Hasselt CA. Neonatal val- prevent any mortality and morbidity from this cause. lecular cysts and failure to thrive. J Laryngol Otol 1992 Newman classified laryngeal cysts as epithelial, May;106(5):448-449. tonsillar and oncocytic cysts (1984: Modified working 8. Mitchell DB, Irwin BC, Bailey CM, Evans JNG. Cyst of the classification).10,11 Two major hypotheses to explain the infant larynx. 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Vallecular cysts native modalities, such as endoscopic marsupialization, in newborns and young infants. Pediatr Pulmonol 1999 excision and de-roofing have been developed recently.1 Apr;27(4):282-285.

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