Sublingual thyroglossal duct cyst (SLTGDC): An unusual location

•Peculiar location of the thyroglossal cyst.•Bilobed/multilobed nature of the cyst.•Varied surgical approach was taken for the enucleation of cyst.•Absence of tract for the thyroglossal cyst

Thyroglossal duct cyst is the most common cause of midline congenital swelling in the neck. Classically, it presents as an asymptomatic midline swelling below the hyoid bone that moves with deglutination and protrusion of the tongue. Sometimes thyroglossal duct cyst presents atypical posing a diagnostic challenge. A sublingual location of thyroglossal duct cyst is rare, and differs quite remarkably in presentation from the classical thyroglossal duct cyst. We describe here the case of a young boy who presented with episodes of postural dyspnea due to elevation of the tongue which was secondary to huge sublingual swelling. Surgical decompression of the lesion was planned on an emergency basis with feasible preoperative workup. The aim of this case report is to highlight the unique presentation and a varied approach in the management of an unusually located sublingual thyroglossal duct cyst. Sublingual/intralingual thyroglossal duct cyst needs to be analyzed in a larger study population for establishing definitive management protocols.