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Two-year old girl with glial choristoma presented in a thyroglossal duct cyst. BACKGROUND: Neuroglial choristomas are rare entities that are composed of differentiated neuroectodermal cells presenting where they do not belong. CASE: Here in this paper, we represent a two-year old patient with a very rare presentation of neuroglial choristoma which manifested itself within a thyroglossal duct cyst. In this paper we will also discuss pathogenesis, clinical manifestation, differential diagnosis and management of the neuroglial choristomas. CONCLUSION: In conclusion we believe this unique case may aid in understanding the pathophysiology, differential diagnosis, and management of this rare congenital anomaly.