1024 Large Posterior Lingual Thyroglossal Duct Cyst Pneumatically Splinted with Auto-Continuous Positive Airway Pressure at Low Pressures

Abstract

Most common age of presentation for thyroglossal duct cysts (TDCs) is the first decade of life, however there is similar incidence of TDC in children and adults. Potential vertical locations of TDCs are: lingual, suprahyoid (including submental), thyrohyoid (between the hyoid bone and thyroid cartilage), or suprasternal. Large lingual TDCs are uncommon, usually situated posteriorly, and may cause significant airway obstruction, and in tragic cases sudden infant death, or fatal asphyxia in adults, perhaps by valve effect on the epiglottis, leading to closure of the trachea by a pressed epiglottis. Large lingual TDCs may potentially be pneumatically splinted with positive airway pressure therapy, given the compressible fluid filled nature of the TDC. The patient is a 58-year-old Iranian American man with long-standing snoring that progressively worsened 4 years ago. Home sleep apnea testing (HSAT) showed an apnea hypopnea index (AHI) of 51.1 events per hour, and patient was started on auto-CPAP with pressure range of 5–20 cm delivered through a nasal mask. Auto-continuous positive airway pressure (auto-CPAP) download for 50 days prior to TDC surgical resection showed an average AHI of 3.6 events per hour, with auto-CPAP mean pressure 7.4 cm H20, and average device pressure ≤ 90% of usage time 9.3 cm H20. Recently while attempting to undergo elective cosmetic facial surgery, his anesthesiologist found his “tongue too large,” and the surgery was cancelled. Patient denied neck mass, throat pain or soreness, voice hoarseness, or cutaneous fistula; he did however endorse gradually worsening dysphagia with globus sensation. Subsequent Otolaryngology consultation, including computerized tomography revealed a 5.8 cm anterior-posterior diameter, by 5.1 transverse diameter, by 4.4 cm cranial-caudal diameter, homogeneous, hypo-dense round/oval base of the tongue lesion, slightly to the right of mid line, displacing the epiglottic vallecula posteriorly, and intimately associated with the hyoid bone, consistent with a large TDC. No internal nodularity, or suspicious enhancement was noted, which was resected along with central hyoid bone using a combined trans-oral robotic surgery (TORS)/Sistrunk procedure. Surgical specimen histopathology revealed a sub-acutely inflamed cyst lined by respiratory and squamous epithelium abutting resected hyoid bone, histologically consistent with a TDC. HSAT after TDC surgical resection revealed an AHI of 10.5 events per hour which was a significant drop from 51.1 events per hour noted before the surgery.