Introduction: The ectopic ciliated cyst is a rare non-odontogenic cyst which occurs as a delayed complication after maxillary sinus radical surgery; this lesion emerges due to the destruction of the sinus mucosa during the surgery and entrapment of the respiratory epithelium in the region. This lesion has been observed in very rare cases following genioplasty and bimaxillary orthognathic surgery.
Case Report: We reported a case of the ectopic ciliated cyst in in the mandible of a 37-year-old Iranian woman following genioplasty and Lefort 1 surgery after 2 years. Its treatment was enucleation.
Conclusion: Long-term follow-up after cosmetic surgery of both jaws is recommended due to the probability of this cyst.
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