Asphyxiating Death Due to Basal Lingual Cyst (Thyroglossal Duct Cyst) in Two-Month-Old Infant Is Potentially Aggravated After Central Catheterization With Forced Positional Changes

Abstract

There are various causes of possible upper airway obstruction in infants. Particularly, large cysts on the base of tongue may cause severe airway obstruction by a mass effect on the hypopharynx and by displacing the epiglottis. Of these basal lingual cysts, thyroglossal duct cyst is rare but occasionally its remnants can be found at the base of the tongue. Although they are pathologically benign, basal lingual cysts may result in death by asphyxia. There are a few reported cases of asphyxia caused by basal lingual cyst. All died suddenly in the bed or cot while sleeping without any significant symptoms and signs. The authors experienced a unique case of death due to airway obstruction by basal lingual cyst (thyroglossal duct cyst), confirmed by postmortem examination, probably aggravated after central catheterization in 2-month-old infant who had open heart surgery for atrial and ventricular septal defect and report with a literature review.

Endoscopic carbon dioxide laser procedure for an endogenous thyroglossal duct cyst

“…There was a special data, which showed only 10 cases belonged to this situation by the end of 2006. However, another 10 cases were presented in less than 2 years [3][4][5][6][7][8]. We reported the 21st case.…”

Uncommon thyroglossal duct cyst with inward tendency: A recommendation regarding ingrowth type

“…All of the cases could be diagnosed and confirmed easily. However, most conditions occurring at uncommon sites (above the hyoid, at the base of the tongue, behind the hyoid bone, or low in the neck) were misdiagnosed before surgery; some of which in children were confirmed only by the autopsy after death due to the absence of the aforementioned typical characteristics [6,7,12,18].…”

Acute Upper-Airway Obstruction by a Lingual Thyroglossal Duct Cyst and Implications for Advanced Airway Management

“…11 If unidentified, the presence of a lingual thyroglossal duct cyst causing upper airway obstruction may be associated with significant risk for mortality. 12, 13 Several case reports describe an infant death, initially presumed to be due to Sudden Infant Death Syndrome (SIDS), but subsequently thought attributable to large lingual thyroglossal duct cysts. 8, 14, 15 Thus, a high index of suspicion for this possible lesion is necessary in infants who present with signs and symptoms of upper airway obstruction.…”

“…The American Society of Anesthesiologists ‘Practice guidelines for management of the difficult airway’ suggest that while “there is insufficient published evidence to evaluate the effect of a bedside medical history or predicting the presence of a difficult airway…there is suggestive evidence that some features of a patient’s medical history …may be related to the likelihood of encountering a difficult airway”. 17 In addition to a suggestive history, 4, 11, 12 features of the physical examination could also be a clue to the possibility of a difficult airway. 17, 18 Among the suggestive historical clues include difficulty with feeding, stridor, respiratory distress of acute onset, positional change in work of breathing, and history of a difficult intubation.…”