Dermoid cysts are uncommon developmental anomalies that rarely occur in the oral cavity. Here, we report a case of a dermoid cyst at the midline of the floor of the mouth and evaluate the density of Langerhans cells in the epithelium lining of this dermoid cyst. A 7-year-old girl came to the dental department of our hospital for treatment of a sublingual swelling detected when she wasw2-years old. Intraoral examination revealed a large soft tissue mass in the midline of the floor of the mouth. There was no palpation pain, the consistency was soft, and the surface mucosa was intact (Figure 1A). The lateral facial view showed an infralingual swelling (Figure 1B) and the occlusal film revealed no sialolith. The clinical impression was a dermoid cyst. Thus, the cystic lesion was removed by surgery under general anesthesia. The surgical specimen measured approximately 2.5 cm 1.5 cm 1.2 cm. On section, it contained cheeselike materials. Histopathological examination showed a cystic lesion lined by orthokeratinized stratified squamous epithelium. The cystic lumen was filled with shreds of keratin and hair-follicle-like structures and sebaceous glands were found in the fibrous cystic wall (Figures 1C and D). Therefore, a dermoid cyst was confirmed.