A 67-year-old man under treatment with antihypertensives and anticoagulants due to cardiac arrhythmia was referred to a dental private practice complaining of a mandibular pain in the fourth quadrant. He had experienced multiple abscesses associated with a fixed dental prosthesis since 2018. Radiographic examination showed the inclusion of 4.5 and a complex endo-periodontal radiolucent lesion on 4.6, with double root fracture and hypercementosis. CT scan revealed an extensive circumferential alveolar bone loss. Under local anesthesia, tooth 4.5 and 4.6 were removed and the alveolar lesion was curetted. The bone defect was filled with platelet-rich plasma and a collagen membrane (Creos Xenoprotec®), fixed with titanium micro-screws (Bioner®). On a follow-up examination, an irregular radiolucency was observed in the surgically treated area two years before. Under local anesthesia, a cystic lesion was excised and submitted for histopathologic analysis with the presumptive diagnosis of residual cyst. The tissue specimen consisted of a dark brown and irregularly-shaped fragment of soft tissue, measuring 1.2 × 1 × 1 cm and brownish cut surface. Microscopic examination showed a thick fibrocollagenous connective tissue wall with different densities and a chronic inflammatory infiltrate. The epithelial lining consisted of a non-keratinised and hyperplastic stratified epithelium with inflammatory exocytosis, that presented large areas of ciliated pseudostratified epithelium with papillary foci. Epithelial nests were observed in the parietal connective tissue, showing the transition between non-keratinised stratified epithelium and ciliated pseudostratified epithelium. Positivity for CK19 (RCK108, ThermoFisher, Thermo Fisher Scientific, Waltham, MA ®) and PAS (+) mucous cells confirmed the respiratory profile of the epithelium. Based on these data, the final diagnosis of the lesion was surgical ciliated cyst of the mandible. At a one-year of follow-up there was no evidence of recurrence or complications.