Document Type : Original Article
Authors
- Saede Atarbashi Moghadam 1
- Termeh Sarrafan Sadeghi 2
- Shokoufeh Shahrabi-Farahani 3
- Leyla Roghanizadeh 4
- Sanaz Gholami Toghchi 1
1 Dept. of Oral and Maxillofacial Pathology, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
2 Dental Student, Student Research Center, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
3 Division of Oral and Maxillofacial Pathology, Dept. of Diagnostic Sciences and Oral Medicine, University of Tennessee Health Science Center, College of Dentistry, Memphis, TN, USA.
4 Iranian Center for Endodontic Research, Research Institute for Dental Sciences, Shahid Beheshti University of Medical Sciences, 1983963113, Tehran, Iran.
Abstract
Background: Hybrid odontogenic lesions (HOLs) show combined microscopic features of two or more recognized odontogenic cysts and neoplasms, occurring in the same primary location. These lesions are uncommon and there is limited information on the clinical and microscopic features of such lesions.
Purpose: We aimed to assess the frequency and types of HOLs admitted to a main oral pathology center in Iran in 30 years.
Materials and Method: In this retrospective observational study, the archives of the Oral and Maxillofacial Pathology Department of Shahid Beheshti University of Medical Sciences from 1993 to 2022 were reviewed, and cases diagnosed with odontogenic lesions were selected. All microscopic slides were screened and cases of the HOLs were extracted.
Results: Over 30 years, a total of 1767 cases (composed of 1264 cysts and 503 tumors) were found to be odontogenic lesions, of which 19 cases (1.07%) were classified as HOLs. The mean±SD and median age of patients were 22.57±13.19 and 15 years, respectively. The most common HOL was dentigerous cyst/odontoma (42.10%) followed by calcifying odontogenic cyst/odontoma (10.52%) and central odontogenic fibroma/central giant cell granuloma (10.52%). About 68.42% of the lesions were associated with impacted teeth. Radiographically, most of the HOLs had a mixed internal structure (68.42%) and were unilocular (73.68%). Most of the lesions showed painless expansion (63.15%). All cases were managed with surgical treatment alone, most of which had conservative surgery (enucleation of the lesion) (88.88%).
Conclusion: HOLs are rare and show a wide variety of histopathologic features. HOLs generally showed the highest frequency in the second decade of life. Awareness of these microscopic patterns can lead to proper diagnosis and management.
Highlights
Saede Atarbashi-Moghadam (Google Scholar)
Keywords
- Pontes FSC, Mosqueda-Taylor A, de*Souza LL, de*Paula LP, Batista LAL, Rodrigues-Fernandes CI, et al. Hybrid odontogenic lesions: A systematic review of 203 cases reported in the literature. J Oral Pathol Med. 2022; 51: 5-12.
- Ide F, Horie N, Shimoyama T, Sakashita H, Kusama K. So-called hybrid odontogenic tumors: do they really exist? Oral Med Patho. 2001; 6: 13-21.
- Akbarizadeh F, Garmabi J, Paknahad M. Concurrent Odontogenic Keratocyst and Odontoma: Report of an Unusual and Rare Entity. J Dent (Shiraz). 2023; 24: 438-443.
- Yoon JH, Kim HJ, Yook JI, Cha IH, Ellis GL, Kim J. Hybrid odontogenic tumor of calcifying odontogenic cyst and ameloblastic fibroma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004; 98: 80-84.
- Zhang W, Chen Y, Geng N, Bao D, Yang M. A case report of a hybrid odontogenic tumour: ameloblastoma and adenomatoid odontogenic tumour in calcifying cystic odontogenic tumour. Oral Oncology Extra. 2006; 42: 287-290.
- Ponniah I, Murali*Gopika*Manoharan GV, Suresh*Kumar P, Karthikeyan K. How to name it: a rare case of odontogenic cyst. J Oral Pathol Med. 2007; 36: 563-569.
- Neuman AN, Montague L, Cohen D, Islam N, Bhattacharyya I. Report of two cases of combined odontogenic tumors: ameloblastoma with odontogenic keratocyst and ameloblastic fibroma with calcifying odontogenic cyst. Head Neck Pathol. 2015; 9: 417-420.
- Tippu SR, Rahman F, Sharma N, Srivastava S. Collision tumor of the palate: A rare case report. Contemp Clin Dent. 2014; 5: 102-105.
- Siar CH, Ng KH. The combined epithelial odontogenic tumour in Malaysians. Br J Oral Maxillofac Surg. 1991; 29: 106-109.
- Rojo R, Prados-Frutos JC, Gutierrez Lázaro I, Herguedas Alonso JA. Calcifying odontogenic cysts. J Stomatol Oral Maxillofac Surg. 2017; 118: 122-124.
- Vered M, Wright JM. Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Odontogenic and Maxillofacial Bone Tumours. Head Neck Pathol. 2022; 16: 63-75.
- Yamazaki M, Maruyama S, Abé T, Babkair H, Fujita H, Takagi R, et al. Hybrid ameloblastoma and adenomatoid odontogenic tumor: report of a case and review of hybrid variations in the literature. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014; 118: e12-e18.
- Akshatha BK, Manjunath GS, Soundarya N. Calcifying odontogenic cyst associated with compound odontoma - A rare entity. J Oral Maxillofac Pathol. 2023; 27(Suppl 1): s69-s74.
- Vizuete-Bolaños MX, Salgado-Chavarria F, Ramírez-Martínez CM, Ramos-Nieto JJ, Vazquez-Dávalos NM. Compound odontoma associated with a calcifying odontogenic cyst. Case report and systematic review. J Stomatol Oral Maxillofac Surg. 2022; 123: e97-e105.
- Mahdavi N, Kardooni Khoozestani N, Hasheminasab M, Soltani N. Hybrid Odontogenic Tumor of Calcifying Odontogenic Cyst and Ameloblastic Fibroma: a Case Report and Review of Literature. J Dent (Shiraz). 2020; 21: 153-157.
- Fujii S, Nagata K, Matsumoto S, Kohashi KI, Kikuchi A, Oda Y, et al. Wnt/β-catenin signaling, which is activated in odontomas, reduces Sema3A expression to regulate odontogenic epithelial cell proliferation and tooth germ development. Sci Rep. 2019; 9: 4257.
- Upadhyaya JD, Cohen DM, Islam MN, Bhattacharyya I. Hybrid Central Odontogenic Fibroma with Giant Cell Granuloma like Lesion: A Report of Three Additional Cases and Review of the Literature. Head Neck Pathol. 2018; 12: 166-174.
- Allen CM, Hammond HL, Stimson PG. Central odontogenic fibroma, WHO type. A report of three cases with an unusual associated giant cell reaction. Oral Surg Oral Med Oral Pathol. 1992; 73: 62-66.
- Odell EW, Lombardi T, Barrett AW, Morgan PR, Speight PM. Hybrid central giant cell granuloma and central odontogenic fibroma-like lesions of the jaws. Histopathology. 1997; 30: 165-171.
- Mosqueda*Taylor A, Bermúdez*Flores V, Díaz*Franco MA. Combined central odontogenic fibroma and giant cell granuloma-like lesion of the mandible: report of a case and review of the literature. J Oral Maxillofac Surg. 1999; 57: 1258-1262.
- Tosios KI, Gopalakrishnan R, Koutlas IG. So-called hybrid central odontogenic fibroma/central giant cell lesion of the jaws. A report on seven additional cases, including an example in a patient with cherubism, and hypotheses on the pathogenesis. Head Neck Pathol. 2008; 2: 333-338.
- Sargolzaei S, Atarbashi-Moghadam S, Roohi A. Mandibular mural ameloblastoma with unusual histopathologic features: a rare challenging case. J Dent (Shiraz). 2019; 20: 304-307.
- Kumar VM, Chakravarthy A, Sathyanarayanan R, Raghu K, Reddy CD. Hybrid ameloblastoma arising from a treated odontogenic keratocyst of the mandible: a case report with literature review. Indian J Otolaryngol Head Neck Surg. 2022; 74(Suppl 3): 6180-6188.
- Siar CH, Ng KH. 'Combined ameloblastoma and odontogenic keratocyst' or 'keratinising ameloblastoma'. Br J Oral Maxillofac Surg. 1993; 31: 183-186.
- Whitt JC, Dunlap CL, Sheets JL, Thompson ML. Keratoameloblastoma: a tumor sui generis or a chimera? Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007; 104: 368-376.
- Jain KS, Bodhankar K, Desai RS, Bansal S, Shirsat P, Prasad P, et al. Absence of BRAFV600E immunohistochemical expression in sporadic odontogenic keratocyst, syndromic odontogenic keratocyst and orthokeratinized odontogenic cyst. J Oral Pathol Med. 2020; 49: 1061-1067.
- Kurppa KJ, Catón J, Morgan PR, Ristimäki A, Ruhin B, Kellokoski J, et al. High frequency of BRAF V600E mutations in ameloblastoma. J Pathol. 2014; 232: 492-498.
- Cousin T, Bobek S, Oda D. Glandular odontogenic cyst associated with ameloblastoma: Case report and review of the literature. J Clin Exp Dent. 2017; 9: e832-e836.
- Gadewar DR, Srikant N. Adenomatoid odontogenic tumour: tumour or a cyst, a histopathological support for the controversy. Int J Pediatr Otorhinolaryngol. 2010; 74: 333-337.
- Grover S, Rahim AM, Parakkat NK, Kapoor S, Mittal K, Sharma B, et al. Cystic Adenomatoid Odontogenic Tumor. Case Rep Dent. 2015; 2015: 503059.
- Kurra S, Gunupati S, Prasad PR, Raju YS, Reddy BV. An Adenomatoid Odontogenic Cyst (AOC) with an Assorted Histoarchitecture: A Unique Entity. J Clin Diagn Res. 2013; 7: 1232-1235.
- Gomez RS, Castro WH, Gomes CC, Loyola AM. Adenomatoid odontogenic tumor associated with odontoma: a case report and critical review of the literature. Head Face Med. 2013; 9: 20.
- Lang MJ, Wang YP, Lin HP, Chen HM, Kuo YS. Adenomatoid odontogenic tumor: Report of a posterior mandibular case with the presence of ghost cells. J Dent Sci. 2015; 10: 216-222.
- Aithal D, Reddy BS, Mahajan S, Boaz K, Kamboj M. Ameloblastomatous calcifying odontogenic cyst: a rare histologic variant. J Oral Pathol Med. 2003; 32: 376-378.
- Atarbashi-Moghadam S, Ghomayshi M, Sijanivandi S. Unusual microscopic changes of Ameloblastic Fibroma and Ameloblastic Fibro-odontoma: A systematic review. J Clin Exp Dent. 2019; 11: e476-e481.
- Usubütün A, Atayar C, Basal N, Araz K. Cystic ameloblastic fibroma. Br J Oral Maxillofac Surg. 2002; 40: 512-514.
- Economopoulou P, Sotiriadou S. An unusual tumor of the mandible with features of unicystic ameloblastoma and ameloblastic fibroma. J Oral Maxillofac Surg. 1998; 56: 1196-1200.
- Chatterjee S, Devi A, Kamboj M, Narwal A. Localization of beta catenin across the domain of odontogenic lesions: A systematic review. J Oral Pathol Med. 2023; 52: 904-910.
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