A case of congenital aplasia of the epiglottis in an adult




Abstract


Aplasia or hypoplasia of the epiglottis in an adult is rarely reported congenital anomaly of the larynx. Most patients with epiglottic aplasia have presented fatal symptom of respiratory distress and severe aspiration in infancy or childhood. We present a case of congenital absence of the epiglottis found in a 33-year-old woman complaining of recurrent lingual tonsillitis. Laryngoscopic finding and CT scan revealed an isolated epiglottic aplasia and normal appearance of other laryngeal structure. This is the first description of physiologic evaluation in an epiglottic anomaly to find the compensatory mechanism of aspiration prevention. We discussed this case with a review of literature.



Introduction


The main functions of larynx are respiration, phonation and protection of airway. Since epiglottis is a physiologic important part of larynx, aplasia or hypoplasia of the epiglottis results in difficulties of phonation and swallowing. Aplasia or hypoplasia of the epiglottis is a fairly rare disease.


Symptoms of a malformation of the epiglottis appear usually as dyspnea and dysphagia in infants. Most of the children with epiglottic deformities died due to respiratory failure and aspiration in an early age. On the other hand, it is asymptomatic in an adult. We report a case of congenital aplasia of the epiglottis in an adult without any other congenital anomaly and symptom, and discuss it with a review of literature. This study was deemed exempt by the Busan St. Mary’s Hospital Institutional Review Board.





Case reports


A 33-year old woman with swallowing pain and myalgia visited our otolaryngology office. There was no unusual symptom except an occasional sore throat. Her medical history, life style and family history were unremarkable. She had no history of surgery or trauma. Her tonsils showed an injection with white patch revealing an acute tonsillitis after physical examination. Under laryngoscopic examination, we found incidentally an aplasia of epiglottis ( Fig. 1 -A ). Computer Tomography (CT) scan was performed to find another accompanied anomaly in the larynx but there was no specific abnormality despite the aplasia of the epiglottis ( Fig. 1 -B). A video fluoroscopic barium swallowing test was done with permission of the patient to evaluate whether there were abnormal aspiration and a protective mechanism of the areodigestive tract. The test revealed a lingual tonsil and tongue base with compensatory function of epiglottis for the closure of airway during swallowing and there was no aspiration ( Fig. 2 ). The tonsillitis symptoms improved by the treatment with antibiotics and anti-inflammatory drugs.




Fig. 1


Endoscopic view of the larynx (A) demonstrating aplasia of the epiglottis with hypertrophic lingual tonsil. Sagittal CT scan (B) showing absence of the epiglottis. (arrow).



Fig. 2


Videoflourographic barium swallowing test. The barium-coated bouls is conveyed into oropharynx. Lingual tonsil and tongue base are elevated in preparation for swallow (A, arrow). The bolus is descending into the hypopharynx (B). The wave of pharyngeal contraction has obliterated the oropharyngeal cavity and moved into the esophagus, with the bolus descending further (C). The bolus has disappeared into the esophagus. There is no aspiration or residual bouls (D).





Case reports


A 33-year old woman with swallowing pain and myalgia visited our otolaryngology office. There was no unusual symptom except an occasional sore throat. Her medical history, life style and family history were unremarkable. She had no history of surgery or trauma. Her tonsils showed an injection with white patch revealing an acute tonsillitis after physical examination. Under laryngoscopic examination, we found incidentally an aplasia of epiglottis ( Fig. 1 -A ). Computer Tomography (CT) scan was performed to find another accompanied anomaly in the larynx but there was no specific abnormality despite the aplasia of the epiglottis ( Fig. 1 -B). A video fluoroscopic barium swallowing test was done with permission of the patient to evaluate whether there were abnormal aspiration and a protective mechanism of the areodigestive tract. The test revealed a lingual tonsil and tongue base with compensatory function of epiglottis for the closure of airway during swallowing and there was no aspiration ( Fig. 2 ). The tonsillitis symptoms improved by the treatment with antibiotics and anti-inflammatory drugs.


Aug 24, 2017 | Posted by in OTOLARYNGOLOGY | Comments Off on A case of congenital aplasia of the epiglottis in an adult

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