A 17-year-old male patient was referred for the extraction of impacted mesiodens and canine (#23) before orthodontic treatment. In the radiologic examination, an ovoid-shape radiolucent lesion was found with well-defined hyperostotic rim involved the teeth in left maxillary area. After biopsy examination the lesion was diagnosed as glandular odontogenic cyst (GOC) differentially from non-odontogrnic cysts, i.e., nasopalatine duct cyst. In the immunohistochemistry the cyst epithelium was freguently positive for PCNA, clearly positive for amtloblastin, slightly positive for β-catenin, but rarely positive for amelogenin and cytokeratin 7. Mucin and PRP were positive in the secretory cells of the cyst epithelium. On the other hand, TNFα was strongly positive in the macropahges infiltrated into the mucin-filled cyst lumen, and CD68 and CD31 were positive in the mucin-filled macropahges. These findings may indicate that the glandular elements are derived from hamartomatous secretory organ and that this odontogenic cyst is arisen from the embryonal rests of enamel epithelium, which has a potential to be differentiated into not only the enamel organ but also the secretory gland. Taken together, we assumed that the present case of GOC was originated from the odontogenic epithelium of impacted teeth, but showed the cytodifferentiation of glandular cells.