Thyroglossal duct cysts are common midline neck swellings that can present at any site along their migratory pathway. They are frequently situated just below the hyoid bone. Extension to the subglottic area is very rare; such an unusual presentation can complicate the diagnosis of a thyroglossal duct cyst. We report the case of a 30-year-old man who presented with a subglottic thyroglossal duct cyst and associated laryngeal symptoms. To the best of our knowledge, only 2 similar cases have been previously reported in the literature, both of which occurred in 2-year-old boys. We believe, therefore, that ours is the first reported case of a subglottic thyroglossal duct cyst in an adult. We discuss the clinical presentation, diagnosis, and treatment of our patient, and we summarize the literature on intralaryngeal thyroglossal duct cysts.