We report a case of anesthetic management of a 43-year-old patient with Eagle's syndrome (ES) in whom post-extubation acute airway obstruction occurred due to bilateral hypoglossal nerve paralysis. After an accurate examination, elongated bilateral stylohyoid ligament was observed and surgical resection was planned. After completion of the surgery following extubation, significant dysfunction in swallowing, speech function, and tongue motion was observed. The clinical situation was evaluated as bilateral hypoglossal nerve paralysis related to the procedure. The patient was closely observed over 48 h in the intensive care unit. After 2 days, the patient was discharged to a surgical ward. Following clinical assessment, the patient was discharged from hospital for monthly return. At the 6-month follow-up, there were no further episodes of paresthesia and other symptoms. In conclusion, patients with ES represent a real challenge for physicians from diagnosis to treatment, especially regarding perioperative complications, and close collaboration between surgeons and anesthesiologists is of crucial importance.