Unexpected failure of videolaryngoscopy due to lingual tonsillar hypertrophy in a patient with Crouzon syndrome and ankylosing spondylitis: a case report

Background: Crouzon syndrome and ankylosing spondylitis are independently associated with difficult airway management because of craniofacial abnormalities and severe restriction of cervical spine mobility. Lingual tonsillar hypertrophy is an underrecognized supraglottic pathology that may cause unexpected failure of tracheal intubation. Case: A 54-year-old man with Crouzon syndrome and ankylosing spondylitis underwent thoracic spinal surgery. Preoperative airway assessment revealed markedly limited neck extension and a Mallampati class III airway; videolaryngoscopy was selected for tracheal intubation. However, no laryngeal structures were visualized (Cormack-Lehane grade IV), and further attempts were abandoned. After reversal of neuromuscular blockade with sugammadex, the patient was awakened and underwent awake fiberoptic intubation. Fiberoptic examination revealed severe lingual tonsillar hypertrophy with atypical supraglottic soft tissue structures obscuring the epiglottis and laryngeal inlet. Despite technical difficulties with airway blocks, incremental topical anesthesia enabled successful awake fiberoptic intubation while maintaining spontaneous ventilation. The surgery and extubation over an airway exchange catheter were completed uneventfully. Conclusion: Videolaryngoscopy may fail in patients with combined craniofacial abnormalities and severe cervical spine rigidity, particularly in the presence of occult supraglottic pathology such as lingual tonsillar hypertrophy. Early recognition of failed intubation, timely transition to awake fiberoptic intubation, and careful extubation planning are essential for safe airway management.

Airway management; Ankylosing spondylitis; Crouzon syndrome; Difficult airway; Fiberoptic intubation; Lingual tonsil hypertrophy; Laryngoscopy

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