Median mandibular cyst is defined as an odontogenic cyst in a rare midline location. In spite of this definition, there have been two reports of a peculiar lesion, so-called "ciliated" median mandibular cyst associated with vital teeth, the origin of which cannot be explained in terms of odontogenic epithelium multipotentiality. We describe a thorough profile of an additional example. On the basis of the cone-beam computed tomography images of a wide open cavity in the midline of the lingual bone, the surgical findings of no association with the vital incisor roots, the immunophenotypes of the excretory duct epithelium and the absence of oncogenic mutations, the present "ciliated" median mandibular cyst associated with vital teeth may be an artifact caused by impingement of the lingual bone due to pedicled extension from an extra-glandular salivary duct cyst in the anterior mouth floor. Awareness of this ectopic presentation is of clinical importance for the differential diagnosis of radiolucencies in the median mandible.
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