Objective To explore the clinical features, reasons of misdiagnosis and surgical treatment of recurrent lingual thyroglossal duct cyst (LTGDC) in adults.Methods Clinical data of 6 adult patients with recurrent LTGDC enrolled from January 2018 to March 2021 were analyzed retrospectively. All patients underwent ultrasound B examination to determine the location of the thyroid, computed tomography (CT) and magnetic resonance imaging (MRI) scans to evaluate the size, location and property of the mass, as well as the relationship between the mass and hyoid bone. Sistrunk operation were performed for all patients.Results Four patients were misdiagnosed as epiglottic cyst while the other two patients were misdiagnosed as cyst of the lingual root. All patients had previously recieved operation for one to three times. The interval between each surgery and the following recurrence ranged from 3 weeks to 1 year. Thyroglossal duct cyst was confirmed by postoperative pathology in all cases. Postoperative follow-up over one year revealed no recurrence.Conclusion For the recurrent cyst located at the base of the tongue or in the preepiglottic space, LTGDC should be differentiated from the other cysts. Besides laryngoscopy, ultrasound B examination and CT scan should be performed to evaluate the morphology and position of the cyst and its relationship with the hyoid bone. Sistrunk surgery is the effective treatment for LTGDC to avoid the recurrence.