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A Newborn With Stridor and Respiratory Distress

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A 0-day-old female neonate exhibited loud stridor and respiratory distress at birth. The pregnancy was uncomplicated, and prenatal ultrasonography results were normal. After spontaneous vaginal delivery at 37 weeks’ gestation, the neonate immediately developed tachypnea, severe retractions, barking cough, hoarse cry, and dusky appearance with oxygen desaturations to 60%, which required continuous positive airway pressure. Despite respiratory support, she was intubated within the first hour of life. The intubating clinician noted a large flesh-colored laryngeal mass and placed a size 3.5 uncuffed endotracheal tube. Ear, nose, and throat evaluation noted that the neonate was intubated and ventilating easily. No craniofacial abnormalities or obvious oral cavity, oropharyngeal, or neck masses were present. Chest radiography was obtained, and results were normal. The patient was subsequently taken to the operating room for direct laryngoscopy and bronchoscopy. Intraoperative evaluation revealed a large submucosal mass involving the left side of the epiglottis, false fold, aryepiglottic fold, and arytenoid that was soft and fluctuant on palpation (Figure 1).

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C. Lateral saccular cyst

Congenital laryngeal cysts are a rare yet important cause of stridor, respiratory distress, and airway obstruction in neonates. Timing of presentation can vary from shortly after birth, to late infancy, to early childhood.17 Saccular cysts are fluid-filled submucosal lesions that arise from the laryngeal saccule and often appear in the anterior aspect of the ventricle. These cysts do not communicate with the laryngeal lumen, as opposed to laryngoceles, which typically contain air or air-fluid levels because they are open to the lumen. However, some controversy exists in the nomenclature of these lesions because they are histologically similar. DeSanto et al2 classified laryngeal saccular cysts into 2 types: anterior and lateral. Anterior cysts enlarge medially and posteriorly between the true and false vocal folds. Lateral cysts progress in a posterosuperior direction, which results in distention of the false vocal fold and aryepiglottic fold, as seen in this neonate.

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Article Information

Corresponding Author: John Faria, MD, Department of Otolaryngology, University of Rochester, 125 Lattimore Rd, Rochester, NY 14620 (john_faria@urmc.rochester.edu).

Published Online: November 7, 2019. doi:10.1001/jamaoto.2019.3367

Conflict of Interest Disclosures: None reported.

Additional Contributions: We thank the patient’s mother for granting permission to publish this information.

References
1.
Kirse  DJ, Rees  CJ, Celmer  AW, Bruegger  DE.  Endoscopic extended ventriculotomy for congenital saccular cysts of the larynx in infants.  Arch Otolaryngol Head Neck Surg. 2006;132(7):724-728. doi:10.1001/archotol.132.7.724PubMedGoogle ScholarCrossref
2.
DeSanto  LW, Devine  KD, Weiland  LH.  Cysts of the larynx—classification.  Laryngoscope. 1970;80(1):145-176. doi:10.1288/00005537-197001000-00013PubMedGoogle ScholarCrossref
3.
Mitchell  DB, Irwin  BC, Bailey  CM, Evans  JN.  Cysts of the infant larynx.  J Laryngol Otol. 1987;101(8):833-837. doi:10.1017/S0022215100102828PubMedGoogle ScholarCrossref
4.
Massoth  LJ, Digoy  GP.  Flexible carbon dioxide laser-assisted endoscopic marsupialization and ablation of a laryngeal saccular cyst in a neonate.  Ann Otol Rhinol Laryngol. 2014;123(8):541-544. doi:10.1177/0003489414525343PubMedGoogle ScholarCrossref
5.
Xiao  Y, Wang  J, Ma  L, Han  D.  The clinical characteristics of congenital laryngeal saccular cysts.  Acta Otolaryngol. 2016;136(2):168-171. doi:10.3109/00016489.2015.1100327PubMedGoogle ScholarCrossref
6.
Prowse  S, Knight  L.  Congenital cysts of the infant larynx.  Int J Pediatr Otorhinolaryngol. 2012;76(5):708-711. doi:10.1016/j.ijporl.2012.02.025PubMedGoogle ScholarCrossref
7.
Kim  JH, Kim  MH, Ahn  HG, Choi  HS, Byeon  HK.  Clinical characteristics and management of saccular cysts: a single institute experience.  Clin Exp Otorhinolaryngol. 2019;12(2):212-216. doi:10.21053/ceo.2018.00808PubMedGoogle ScholarCrossref
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