A 10-year-old female presenting with a discharging sinus in the midline of the neck of one month duration was diagnosed to have a thyroglossal sinus. She underwent sistrunk's operation, intra-operatively a thyroglossal duct cyst was atypically sited in the intrahyoid region. The cyst may be located in the intralingual, suprahyoid, thyrohyoid or suprasternal region. A literature search revealed that this intrahyoid location of a thyroglossal duct cyst is extremely rare and so far only three cases have been reported. A case and review of literature regarding this unusual entity are presented.
Keywords: Hyoid Bone; Thyroglossal Cyst; Thyroglossal Duct Cyst.