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.
Keywords: choristoma; neuroglial; sistrunk; thyroglossal duct.