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Case Reports
. 2022 Oct-Dec;26(4):583-588.
doi: 10.4103/jomfp.jomfp_20_22. Epub 2022 Dec 22.

Unicystic desmoplastic osteo-ameloblastoma of posterior mandible: Report of rarest of the rare case and review of literature

Setu P Shah  1 , Deval Mehta  1 , Sonal Madan  1 , Binita Gandhi  2
Affiliations
Case Reports

Unicystic desmoplastic osteo-ameloblastoma of posterior mandible: Report of rarest of the rare case and review of literature

Setu P Shah et al. J Oral Maxillofac Pathol. 2022 Oct-Dec.

Abstract

Desmoplastic ameloblastoma (DA) is an epithelial odontogenic tumor and a rare variant of ameloblastoma. It was first described by Eversole in 1984. In the World Health Organization (WHO) classification of odontogenic tumors (2005), DA has been considered as a distinct entity from conventional/multicystic ameloblastoma. DA differs strikingly in its clinical, radiological, and histopathological presentation when compared to other variants of ameloblastoma. We report here an extremely rare "Hybrid DA" in a 50-year-old female patient with painless hard swelling involving right posterior mandible with detailed clinical history, an unusual radiographic and histopathological presentation. Histopathology revealed odontogenic epithelium in the form of follicles, ameloblastoma with cystic degeneration, and squamous metaplasia at places and elsewhere there were odosntogenic islands compressed by dense fibrocellular stroma suggestive of desmoplasia along with osseous tissue formation. Also review of the literature and possible explanation of etiopathogenesis of cystic change and osseous tissue formation in DA are discussed.

Keywords: Desmoplastic ameloblastoma; hybrid; osteoplasia; rare; unicystic.

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Conflict of interest statement

There are no conflicts of interest.

Figures

Figure 1
Figure 1
Preoperative clinical views. (a, b) Extra-oral views (c) Intra-oral view
Figure 2
Figure 2
Preoperative radiographs. (a) OPG (b) Sagittal view (c) Axial view (d) Coronal view (e, f) 3D views
Figure 3
Figure 3
Intraoperative photographs. (a) Inferior alveolar and mental neurovascular bundle separated and saved (b) Application of Cornoy's solution (c) Aspirated fluid with specimen
Figure 4
Figure 4
Microscopic features. (a) Odontogenic epithelium lining cystic lumen with dense fibrous connective tissue (scanner view). (b) Degenerated odontogenic epithelium with hyalinization and ossification (low power). (c) Cystic lumen lined by odontogenic epithelium with lining epithelial cells proliferating within connective tissue capsule (high power)
Figure 5
Figure 5
Postoperative follow-up after 6 months. (a) Clinical intra-oral view (b) OPG
See this image and copyright information in PMC

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