Vallecular Cyst as a Cause of obstructive Sleep apnea in an infant

Abstract

A 3-month-old baby was diagnosed with obstructive sleep apnea (OSA) on polysomnography (PSG) with a high apnea hypopnea index (AHI). On further investigations he was found to have a vallecular cyst that was successfully treated. We discuss the clinical presentation of vallecular cysts and the importance of polysomnography in identifying this rare condition. REpoRt oF CaSEA 3-month-old baby with a history of incomplete right cleft lip and alveolus was referred by the Craniofacial Surgery team for PSG for evaluation prior to surgical closure. Patient was born full term via C-section for fetal decelerations with an uneventful perinatal course. He had stridor, snoring, and gasping during sleep since birth. A PSG was performed during daytime nap. He slept for 64 minutes with an arousal/awakening index of 5.6 per hour. He was found to have 45 obstructive apneas, 2 obstructive hypopneas, and 4 mixed apneas (AHI 64.8/h, baseline oxygen saturation 94% to 96%, desaturation index 89/h, SpO 2 nadir 75%, P ET CO 2 highest 47 torr). Due to OSA severity, he was admitted for further investigation.On physical exam, he was well developed, noticeably stridulous, with high arched palate and right incomplete lip with alveolar cleft. He had a pectus excavatum deformity with mild subcostal retractions but good bilateral air entry and clear breath sounds.Chest x-ray incidentally showed a right middle lobe infi ltrate, and he was found to be positive for metapneumovirus. He was treated for presumed aspiration pneumonia, and refl ux management was initiated for clinical evidence of refl ux. He was noted to have laryngomalacia on bedside laryngoscopy performed by the otorhinolaryngologist. He was discharged home on oxygen supplementation at half liter per minute (Lpm) via nasal cannula. He had a subsequent hospitalization for RSV bronchiolitis within two weeks of discharge.Subsequently, on outpatient follow-up with the otorhinolaryngologist, full offi ce laryngoscopy revealed a vallecular cyst (Figure 1) cyst was successfully marsupialized. Six weeks later, a repeat PSG during daytime nap was performed; it showed marked improvement of his obstructive apneas, and reduction of AHI from 64.8 to 7.2 (baseline oxygen saturation 96% to 98%; desaturation index 12/h; SpO 2 nadir 76%, P ET CO 2 highest 44 torr). His need for supplemental oxygen was reduced to 0.25 Lpm. He had his cleft lip successfully repaired at 9 months of age, and a follow-up PSG during daytime nap was completely normal (AHI 0.2/h; baseline oxygen saturation 95% to 97%; desaturation index 1.2/h; SpO 2 nadir 91%, P ET CO 2 highest 45 torr).

Clinical efficacy of low-temperature radiofrequency ablation of pharyngolaryngeal cyst in 84 Chinese infants

“…[ 1 – 3 ] In the present study, 34 pharyngolaryngeal cysts were located at the tongue base, 25 on the lingual surface of epiglottis, 15 in the epiglottic vallecula, 2 at valleculae linguae, 6 at the aryepiglottic fold, and 2 cases in the subglottic region. The previous reports also detected laryngeal vallecular cyst [ 15 ] and Thornwald cyst. [ 16 ] This suggests that the pharyngolaryngeal cyst can occur in any region of pharyngolarynx.…”

“…Pharyngolaryngeal cysts frequently induce airway obstruction and may even cause suffocation in infants. [ 15 – 17 ] Hence, it is very important to diagnose these patients accurately and in a timely manner. The previous reports as well as our own experience suggests that the diagnosis of pharyngolaryngeal cysts is usually made on palpation of neck, blood gas analysis, B-mode ultrasonography of thyroid, diagnostic imaging (CT or MRI), and laryngoscopy.…”

Scoping review of head and neck neoplasms presenting with obstructive sleep apnoea: the importance of flexible nasendoscopy

“…The majority of included patients had their neoplasms treated surgically, with 57 patients (72.2%) treated with surgery alone 9–12,14–61 . Two patients (2.5%) were treated with both surgery and radiotherapy, 13,62 and one patient (1.3%) treated with each of surgery and chemotherapy 63 and surgery and chemoradiotherapy 64 .…”

Bilateral vallecular cysts

“… 4 , 5 The patient presented in this study was asymptomatic; however some authors had described important symptoms as dysphagia, 3 stridor and dyspnea. 1 Therefore, we highlighted the importance of the endoscopic exam in patients with these symptoms.…”

“…This classification is based on histological features, content and site. 1 The entire larynx can harbor cysts, the most common sites being, in order of frequency, the vocal folds, epiglottis, and valeculla, 2 where 10% of the cases occur. 3 …”