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Soluk-Tekkesin M, Bologna-Molina R, Magliocca K, van Heerden W, Robinson L, Bilodeau EA, Hussaini HM, Adisa AO, Tilakaratne WM, Li J, Hunter KD, Gomez RS. Malformations vs. Neoplasia in the Oral Cavity: Special Emphasis on Mixed Odontogenic Tumors. J Oral Pathol Med 2025; 54:76-79. [PMID: 39617625 DOI: 10.1111/jop.13592] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/22/2024] [Revised: 11/02/2024] [Accepted: 11/03/2024] [Indexed: 01/15/2025]
Abstract
BACKGROUND The terminology surrounding developmental lesions in the oral cavity is widely applied, often leading to confusion in differentiating between developmental malformations and neoplasia. Odontogenic tumor classification includes both true neoplasms and malformations which make it very complex and dynamic. METHOD AND CONCLUSION In this brief report, we will first discuss the concepts of malformations and neoplasia and then focusing on their relevance to odontogenic tumors, which impacts their classification and treatment, particularly mixed odontogenic lesions.
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Affiliation(s)
- Merva Soluk-Tekkesin
- Department of Oral Pathology, Faculty of Dentistry, University of Istanbul, Istanbul, Türkiye
| | - Ronell Bologna-Molina
- Diagnostic in Oral Pathology and Oral Medicine Department, School of Dentistry, Universidad de la Republica (UDELAR), Montevideo, Uruguay
| | - Kelly Magliocca
- Department of Pathology and Laboratory Medicine, Emory University, Atlanta, Georgia, USA
| | - Willie van Heerden
- Department of Oral and Maxillofacial Pathology, School of Dentistry, University of Pretoria, Pretoria, South Africa
| | - Liam Robinson
- Department of Oral and Maxillofacial Pathology, School of Dentistry, University of Pretoria, Pretoria, South Africa
| | - Elizabeth Ann Bilodeau
- Oral and Maxillofacial Pathology, UDHS Oral Pathology Laboratory, University of Pittsburgh School of Dental Medicine, Pittsburgh, Pennsylvania, USA
| | | | - Akinyele Olumuyiwa Adisa
- Department of Oral Pathology, Faculty of Dentistry, College of Medicine, University of Ibadan, Ibadan, Nigeria
| | | | - Jiang Li
- Department of Oral Pathology, Shanghai Ninth People's Hospital, Shanghai Jiaotong University School of Medicine, Shanghai, China
| | - Keith David Hunter
- Liverpool Head and Neck Centre, Molecular and Clinical Cancer Medicine, University of Liverpool, Liverpool, UK
| | - Ricardo Santiago Gomez
- Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil
- Faculty of Medical Science of Minas Gerais, Belo Horizonte, Brazil
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2
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Jung Y, Oh KY, Han SS, Lee C. A rare case report of ameloblastic fibrodentinoma with imaging features in a pediatric patient. Imaging Sci Dent 2024; 54:207-210. [PMID: 38948190 PMCID: PMC11211030 DOI: 10.5624/isd.20230247] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/15/2023] [Revised: 02/29/2024] [Accepted: 03/08/2024] [Indexed: 07/02/2024] Open
Abstract
Ameloblastic fibrodentinoma (AFD) is a rare benign odontogenic tumor that resembles an ameloblastic fibroma with dysplastic dentin. This report presents a rare case of mandibular AFD with imaging features in a young patient. Panoramic radiography and computed tomography revealed a well-defined lesion with internal septa and calcified foci, causing inferior displacement of the adjacent molars as well as buccolingual cortical thinning and expansion of the posterior mandible. The lesion was surgically removed via mass excision, and the involved tooth was extracted under general anesthesia. During the 5-year follow-up period, no evidence of recurrence was observed. Radiologic features of AFD typically reveal a moderately to well-defined mixed lesion with varying degrees of radiopacity, reflecting the extent of dentin formation. Radiologists should consider AFD in the differential diagnosis when encountering a multilocular lesion with little dense radiopacity, particularly if it is associated with delayed eruption, impaction, or absence of involved teeth, on radiographic images of young patients.
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Affiliation(s)
- Youjin Jung
- Department of Oral and Maxillofacial Radiology, Yonsei University College of Dentistry, Seoul, Korea
| | - Kyu-Young Oh
- Department of Oral Pathology, College of Dentistry, Dankook University, Cheonan, Korea
| | - Sang-Sun Han
- Department of Oral and Maxillofacial Radiology, Yonsei University College of Dentistry, Seoul, Korea
| | - Chena Lee
- Department of Oral and Maxillofacial Radiology, Yonsei University College of Dentistry, Seoul, Korea
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3
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Singh Sihmar S, Rathi S, Ramalingam K, Ramani P, Sethuraman S. Developing Odontoma in the Mandible of an Eight-Year-Old Boy. Cureus 2024; 16:e56230. [PMID: 38618463 PMCID: PMC11016319 DOI: 10.7759/cureus.56230] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/15/2024] [Indexed: 04/16/2024] Open
Abstract
Benign mixed odontogenic tumors have been repeatedly classified and reclassified over the past few decades. Odontoma is considered a hamartoma due to its slow growth and non-aggressive nature. We present an interesting case of developing odontoma in an eight-year-old boy. His complaint was a slow-growing swelling in the lower right back tooth region. Clinical examination revealed a carious deciduous second molar. The orthopantomogram revealed a well-defined radiolucency around the unerupted mandibular first premolar and impacted mandibular second premolar. Histopathology revealed an odontogenic epithelial lining overlying myxofibrous stroma with inflammatory cells and calcified structures with few odontogenic rests. Special staining methods including Van Gieson and modified Gallego stains led to the final diagnosis of a developing odontoma.
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Affiliation(s)
- Sandeep Singh Sihmar
- Oral Pathology, Daswani Dental College, Kota, IND
- Oral Pathology, Darshan Foundation, Bhiwani, IND
| | - Shalini Rathi
- Oral Medicine and Radiology, Maharaja Ganga Singh Dental College and Research Centre, Sri Ganganagar, IND
- Oral Medicine and Radiology, Darshan Foundation, Bhiwani, IND
| | - Karthikeyan Ramalingam
- Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, IND
| | - Pratibha Ramani
- Oral Pathology and Microbiology, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, IND
| | - Sathya Sethuraman
- Dentistry, Saveetha Dental College and Hospitals, Saveetha Institute of Medical and Technical Sciences, Saveetha University, Chennai, IND
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4
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Jihed S, Slim A, Charfeddine A, Smida A, Abdellatif C, Selmi J. Ameloblastic Fibro-odontoma - A Case Report of Two Uncommon Cases. Ann Maxillofac Surg 2024; 14:116-119. [PMID: 39184429 PMCID: PMC11340845 DOI: 10.4103/ams.ams_7_24] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/17/2024] [Revised: 04/12/2024] [Accepted: 04/24/2024] [Indexed: 08/27/2024] Open
Abstract
Rationale Ameloblastic fibro-odontoma (AFO) is a rare mixed odontogenic tumour with a preferential location in the posterior mandible and with a variety of radiological aspects. We report two clinical cases of AFO in two rare locations and with unusual radiological aspects. Patient Concerns The first patient is a 37-year-old female with an asymptomatic intraoral swelling located in the anterior mandibular. The second patient is a 16-year-old girl with a chief complaint of missing maxillary canine. Diagnosis Both cases were diagnosed with AFO. Treatment For the first patient, a biopsy was performed, and for the second one, the lesion was surgically excised. Outcomes The first patient is under regular surveillance and the other was followed up for one year without any evidence of recurrences. Take-away Lessons Despite many efforts, the nature, histology and therapy of these lesions remain very confusing.
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Affiliation(s)
- Sghaier Jihed
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
| | - Afef Slim
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
| | - Abir Charfeddine
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
| | - Abdelkader Smida
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
| | - Chokri Abdellatif
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
| | - Jamil Selmi
- Department of Oral Medicine and Oral Surgery, Faculty of Dental Medicine of Monastir, Monastir, Tunisia
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5
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Liu Y, Smith MH, Patel PB, Bilodeau EA. Pediatric Odontogenic Tumors. Pediatr Dev Pathol 2023; 26:583-595. [PMID: 38032744 DOI: 10.1177/10935266231200115] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/02/2023]
Abstract
Odontogenic tumors are rare tumors of the jaws that arise from remnants of the tooth forming apparatus. Some odontogenic tumors demonstrate strong predilection for pediatric patients including the unicystic ameloblastoma, adenomatoid odontogenic tumor, ameloblastic fibroma, ameloblastic fibro-odontoma, odontoma, and primordial odontogenic tumor. In this review, we discuss the clinical, radiographic, histopathologic, and molecular characteristics of select odontogenic tumors that demonstrate pediatric predilection and review management.
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Affiliation(s)
- Yingci Liu
- Rutgers School of Dental Medicine, Newark, NJ, USA
| | | | - Paras B Patel
- CMO, Center for Oral Pathology, Dallas, TX, USA
- Oral and Maxillofacial Pathology ProPath, Dallas, TX, USA
| | - Elizabeth Ann Bilodeau
- Oral and Maxillofacial Pathology, UDHS Oral Pathology Laboratory, University of Pittsburgh School of Dental Medicine, Pittsburgh PA, USA
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6
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Darwish NMA, Amer HWAF, Mahrous NNM. Ameloblastic fibro-dentinoma: a rare mixed odontogenic tumor case report with review of literature. J Egypt Natl Canc Inst 2023; 35:34. [PMID: 37899408 DOI: 10.1186/s43046-023-00193-0] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2023] [Accepted: 10/15/2023] [Indexed: 10/31/2023] Open
Abstract
BACKGROUND Ameloblastic fibro-dentinoma is considered a rare, benign, mixed odontogenic tumor that occurs mainly in the posterior mandible in the 1st-2nd decade of life. Although the clinical behavior of Ameloblastic fibro-dentinoma is similar to that of ameloblastic fibroma, there is a debate about whether Ameloblastic fibro-dentinoma is a developing hamartomatous odontoma or a separate neoplastic odontogenic tumor like ameloblastic fibroma. However, it is important to understand the histopathogenesis of this rare tumor. CASE PRESENTATION A case report presenting an 11-year-old male child with a swelling in the posterior mandible. Radiographic examination revealed a multilocular lesion with mixed radiodensity related to the impacted lower left second premolar tooth. Incisional biopsy was done, and microscopic examination revealed cords and nests of odontogenic follicles lined by ameloblast-like cells and central stellate reticulum-like cells in the primitive ecto-mesenchymal stroma with areas of dentinoid material and osteodentin. The diagnosis was ameloblastic fibro-dentinoma. Surgical excision of the lesion was done, and the patient was followed up for 1 year without evidence of recurrence. CONCLUSION Reporting such a rare entity clarifies the debate about its nature and the importance of early diagnosis of lesions that are associated with unerupted teeth showing how it is effective in early management and prognosis.
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Gomes IP, Bastos VC, Guimarães LM, Gomes CC. The molecular basis of odontogenic cysts and tumours. J Oral Pathol Med 2023; 52:351-356. [PMID: 36629457 DOI: 10.1111/jop.13401] [Citation(s) in RCA: 13] [Impact Index Per Article: 6.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/03/2022] [Accepted: 12/19/2022] [Indexed: 01/12/2023]
Abstract
The advances in molecular technologies have allowed a better understanding of the molecular basis of odontogenic cysts and tumours. PTCH1 mutations have been reported in a high proportion of odontogenic keratocyst. BRAF p.V600E are recurrent in ameloblastoma and KRAS p.G12V/R in adenomatoid odontogenic tumour, dysregulating the MAPK/ERK pathway. Notably, BRAF p.V600E is also detected in ameloblastic carcinoma, but at a lower frequency than in its benign counterpart ameloblastoma. Recently, adenoid ameloblastoma has been shown to be BRAF wild-type and to harbour CTNNB1 (β-catenin gene) mutations, further suggesting that it is not an ameloblastoma subtype. CTNNB1 mutations also occur in other ghost-cell-containing tumours, including calcifying odontogenic cysts, dentinogenic ghost cell tumours and odontogenic carcinoma with dentinoid, but the link between CTNNB1 mutations and ghost cell formation in these lesions remains unclear. Regarding mixed tumours, BRAF p.V600E has been reported in a subset of ameloblastic fibromas, ameloblastic-fibrodentinomas and fibro-odontomas, in addition to ameloblastic fibrosarcoma. Such mutation-positivity in a subset of samples can be helpful in differentiating some of these lesions from odontoma, which is BRAF-wild-type. Recently, FOS rearrangements have been reported in cementoblastoma, supporting its relationship with osteoblastoma. Collectively, the identification of recurrent mutations in these aforementioned lesions has helped to clarify their molecular basis and to better understand the interrelationships between some tumours, but none of these genetic abnormalities is diagnostic. Since the functional effect of pathogenic mutations is context and tissue-dependent, a clear role for the reported mutations in odontogenic cysts and tumours in their pathogenesis remains to be elucidated.
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Affiliation(s)
- Isadora Pereira Gomes
- Department of Pathology, Biological Science Institute (ICB), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Minas Gerais, Brazil
| | - Victor Coutinho Bastos
- Department of Pathology, Biological Science Institute (ICB), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Minas Gerais, Brazil
| | - Letícia Martins Guimarães
- Department of Pathology, Biological Science Institute (ICB), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Minas Gerais, Brazil
| | - Carolina Cavaliéri Gomes
- Department of Pathology, Biological Science Institute (ICB), Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Minas Gerais, Brazil
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8
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MR appearance of a rare ameloblastic fibroma with formation of dental hard tissues with histopathologic correlation: a case report. Oral Radiol 2023; 39:220-224. [PMID: 36002688 DOI: 10.1007/s11282-022-00649-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/21/2022] [Accepted: 08/10/2022] [Indexed: 01/07/2023]
Abstract
An ameloblastic fibroma with formation of dental hard tissues, which the classical name is ameloblastic fibro-odontoma (AFO), is a rare type of mixed odontogenic tumor. An 8-year-old boy was diagnosed with AFO, with an inhomogeneous high signal within the lesion shown by T2-weighted magnetic resonance imaging (MRI). Computed tomography (CT) imaging revealed a unilocular low CT value area of 24 × 19 × 26 mm with buccolingual bony expansion and cortical bone thinning on the left side of the mandible including the crown of the mandibular left second molar. In addition, multiple calcified bodies were detected within the lesion, one of which had a CT value of approximately 2200 HU, equivalent to that of enamel. MRI indicated the lesion to be sized 24 × 19 × 25 mm along with buccolingual bony expansion in the left side of the mandible. Additionally, the lesion showed an internal inhomogeneous high signal, while a portion had an especially high signal in T2-weighted images. That particularly high signal area coincided with the nodular growth area of mucus-rich mesenchymal components without the epithelial component in histopathology findings. The particularly high signal revealed by T2-weighted imaging could be attributed to the mucus-rich component. MRI was found useful for revealing differences in the internal histopathological properties of an AFO in our patient.
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9
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Ameloblastic Fibro-Odontoma in a Free-Ranging Rocky Mountain Bighorn Sheep (Ovis canadensis canadensis), Alberta, Canada. J Wildl Dis 2022; 58:689-691. [PMID: 35512297 DOI: 10.7589/jwd-d-21-00157] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/01/2021] [Accepted: 01/18/2022] [Indexed: 11/20/2022]
Abstract
An adult female Rocky Mountain bighorn sheep (Ovis canadensis canadensis) was euthanized because of a large mandibular mass. Histopathology revealed an ameloblastic fibro-odontoma, a rare odontogenic neoplasia.
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10
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Sanjai K, Reddy LP, Roopavathi K, Muniswamy HK. Ameloblastic Fibro-Odontoma: A Journey of Progression? J Oral Maxillofac Pathol 2022; 26:S40-S45. [PMID: 35450248 PMCID: PMC9017823 DOI: 10.4103/jomfp.jomfp_184_21] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/11/2021] [Revised: 10/19/2021] [Accepted: 10/29/2021] [Indexed: 11/11/2022] Open
Abstract
A 19-year-old male patient reported to dental OPD of our institution with a swelling in the posterior part of the mandible on the left side. The patient gave a history of gradual increase in the size of swelling for 7 years. The patient also had a radiograph and histopathology slides from his previous dental visit at another facility. The radiograph revealed a well-circumscribed radiolucency with an impacted tooth (38). Histopathology slides showed features of an ameloblastic fibroma (AF). The patient had deferred the treatment for 5 years since he was young and reported to our OPD due to increase in the size of the swelling over the past few weeks. The present radiographs revealed radiolucency with radiopacity. Excisional biopsy was performed and the histopathological examination revealed an Ameloblastic fibro odontoma. This case report is to document and highlight the possible progression of AF to Ameloblastic fibro-odontoma.
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Affiliation(s)
- Karpagaselvi Sanjai
- Department of Oral Pathology, Vydehi Institute of Dental Sciences and Research Centre, Bengaluru, Karnataka, India
| | - Lokesh Papiah Reddy
- Department of Oral Pathology, Vydehi Institute of Dental Sciences and Research Centre, Bengaluru, Karnataka, India
| | - Keshavaiah Roopavathi
- Department of Oral Pathology, Vydehi Institute of Dental Sciences and Research Centre, Bengaluru, Karnataka, India
| | - Harish Kumar Muniswamy
- Department of Oral Pathology, Vydehi Institute of Dental Sciences and Research Centre, Bengaluru, Karnataka, India
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11
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Sánchez-Romero C, Paes de Almeida O, Bologna-Molina R. Mixed odontogenic tumors: A review of the clinicopathological and molecular features and changes in the WHO classification. World J Clin Oncol 2021; 12:1227-1243. [PMID: 35070741 PMCID: PMC8716991 DOI: 10.5306/wjco.v12.i12.1227] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/19/2021] [Revised: 05/25/2021] [Accepted: 11/25/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Ameloblastic fibromas and ameloblastic fibrosarcomas are rare odontogenic tumors, and controversy exists in the classification of cases presenting hard-tissue production: Ameloblastic fibrodentinoma (AFD) and ameloblastic fibro-odontoma (AFO). These cases are currently considered “developing odontomas” (hamartomatous lesions).
AIM To analyze the clinicopathologic features of these lesions and discuss the changes in the 2017 World Health Organization classification.
METHODS An electronic literature search was performed in the PubMed/MEDLINE database. An electronic search of the English language literature was performed and last updated in September 2020 in the PubMed/MEDLINE database using the following terms: “ameloblastic fibroma”, “ameloblastic fibrodentinoma”, “ameloblastic fibro-odontoma”, “ameloblastic sarcoma”, “ameloblastic fibrosarcoma”, “ameloblastic fibrodentinosarcoma”, “ameloblastic fibroodontosarcoma” and “odontogenic carcinosarcoma”. The inclusion criteria were odontogenic tumor series, case reports and systematic reviews that provided sufficient clinical, radiological and microscopic documentation to confirm the diagnosis.
RESULTS The database search strategy resulted in 947 papers. Articles focusing on other topics, articles that were not in English, duplicate articles, and articles without fulfilling the inclusion criteria were excluded. Finally, 96 publications were included in this review to describe and discuss the main features of the searched entities. Several aspects of AFO and AFD, such as biological behavior, age of occurrence, amount of hard tissue, and potential for malignant transformation into odontogenic sarcomas, support the neoplastic nature in most of the reported cases. Considering the clinical, radiographic, histopathological and molecular characteristics of odontogenic lesions with hard tissue production, we suggest that these types of lesions should continue to be recognized as odontogenic tumors by maintaining the classically used terms.
CONCLUSION This recommendation will be relevant for future clinical, microscopic, and molecular studies to better understand the biology of these interesting odontogenic tumors.
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Affiliation(s)
- Celeste Sánchez-Romero
- Department of Oral Pathology, Faculty of Dentistry, University Juarez of the Durango State, Durango 33106, Mexico
| | - Oslei Paes de Almeida
- Department of Diagnosis, Faculty of Piracicaba, State University of Campinas, São Paulo Brazil, Piracicaba 13414-903, Sao Paulo, Brazil
| | - Ronell Bologna-Molina
- Department of Oral Pathology, Faculty of Dentistry, University Juarez of the Durango State, Durango 33106, Mexico
- Department of Oral and Maxillofacial Pathology, Universidad de la República UDELAR (URUGUAY), Montevideo 16400, Uruguay
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12
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Omar N, Ullah A, Ghleilib I, Patel N, Abdelsayed RA. A Locally Aggressive Ameloblastic Fibro-Odontoma: A Case Report and Literature Review. Cureus 2021; 13:e20366. [PMID: 35036200 PMCID: PMC8752350 DOI: 10.7759/cureus.20366] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 12/12/2021] [Indexed: 11/29/2022] Open
Abstract
Ameloblastic fibro-odontoma (AFO) is a relatively rare, benign noninvasive mixed odontogenic neoplasm derived from epithelial and ectomesenchymal elements of the dental tissues. It usually presents with a mean age of 11.5 years and in the posterior segment of the mandible. It is extremely rare in the posterior maxilla. Although the latest WHO edition classified AFO as developing odontoma, here we present a locally aggressive AFO in a 21-year-old male involving the posterior maxilla and sinus with bone destruction. The patient presents with a two-year history of slowly progressive left facial swelling with malodorous drainage. The CT scan revealed a 5.5 x 4.3 cm well-circumscribed expansile mass with mixed attenuation and peripheral calcification occupying the left maxilla and sinus with bone destruction of the hard palate and orbital rim. According to the literature, most of the AFO cases were treated adequately through a conservative approach with just enucleation or surgical curettage. To our knowledge, our case is the first case treated aggressively with left maxillectomy, palatectomy, and reconstruction surgery because of its radiologic findings, which suggested a locally invasive neoplasm. Histologically, the specimen showed a mixture of proliferative epithelial, mesenchymal tissue elements, and variable amounts of mineralized deposits consisting of enamel matrix and dentinoid deposits, and the final diagnosis was AFO. In conclusion, we present a rare case of AFO with an unusual aggressive presentation, age group, and site involved. The radiographic, histopathologic features, and therapeutic approaches of this unusual locally aggressive tumor are presented with the review of relevant literature.
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13
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Nandini DB, Reddy PB, Singh WR, Singh KS. Ameloblastic fibro-odontoma or complex odontoma masquerading as gingival enlargement. J Indian Soc Periodontol 2021; 25:438-442. [PMID: 34667389 PMCID: PMC8452172 DOI: 10.4103/jisp.jisp_778_20] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/18/2020] [Revised: 02/09/2021] [Accepted: 03/08/2021] [Indexed: 11/23/2022] Open
Abstract
Ameloblastic fibro-odontoma is a rare tumor affecting the pediatric population and young adults. The World Health Organization (WHO) in 2005 defined it as “A neoplasm composed of proliferating odontogenic epithelium in a cellular ectomesenchymal tissue with varying degrees of inductive changes and dental hard tissue formation.” There exists a controversy on its histogenesis designating it as a hamartoma (developing complex odontoma [CO]) or a true neoplasm since both the lesions appear similar histologically. Recently, the WHO in 2017 has clubbed both these lesions as the same entity. Most cases are reported in males and in mandible, while cases in maxilla are scarce. This article describes a recurrence of a previously reported case of ameloblastic fibroma which showed maturation into AFO or CO in a girl aged 6 years in the posterior maxilla. This case is reported due to its rarity and a brief review with differential diagnosis is also discussed.
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Affiliation(s)
| | - Praveen Bokka Reddy
- Department of Oral and Maxillofacial Surgery, Dental College, Regional Institute of Medical Sciences, Imphal, Manipur, India
| | - Waikhom Robindro Singh
- Department of Oral and Maxillofacial Surgery, Dental College, Regional Institute of Medical Sciences, Imphal, Manipur, India
| | - Koijam Sashikumar Singh
- Department of Oral Medicine and Radiology, Dental College, Regional Institute of Medical Sciences, Imphal, Manipur, India
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14
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Holmes KR, Holmes RD, Martin M, Murray N. Practical Approach to Radiopaque Jaw Lesions. Radiographics 2021; 41:1164-1185. [PMID: 34086497 DOI: 10.1148/rg.2021200187] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Radiopaque lesions of the jaw are myriad in type and occasionally protean in appearance. In turn, the radiologic analysis of these lesions requires a systematic approach and a broad consideration of clinical and imaging characteristics to enable reliable radiologic diagnosis. Initially categorizing lesions by attenuation pattern provides a practical framework for organizing radiopaque jaw lesions that also reflects important tissue characteristics. Specifically, the appearance of radiopaque lesions can be described as (a) densely sclerotic, (b) ground glass, or (c) mixed lytic-sclerotic, with each category representing a distinct although occasionally overlapping differential diagnosis. After characterizing attenuation pattern, the appreciation of other radiologic features, such as margin characteristics or relationship to teeth, as well as clinical features including demographics and symptoms, can aid in further narrowing the differential diagnosis and lend confidence to clinical decision making. The authors review the potential causes of a radiopaque jaw lesion, including pertinent clinical and radiologic features, and outline a simplified approach to its radiologic diagnosis, with a focus on cross-sectional CT. An invited commentary by Buch is available online. ©RSNA, 2021.
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Affiliation(s)
- Kenneth R Holmes
- From the Departments of Medicine (K.R.H.) and Radiology (R.D.H.), University of British Columbia, 2775 Laurel St, 11th Floor, Vancouver, BC, Canada V5Z 1M9; BC Cancer Agency, Vancouver, British Columbia, Canada (M.M.); and Vancouver General Hospital, Vancouver, British Columbia, Canada (N.M.)
| | - R Davis Holmes
- From the Departments of Medicine (K.R.H.) and Radiology (R.D.H.), University of British Columbia, 2775 Laurel St, 11th Floor, Vancouver, BC, Canada V5Z 1M9; BC Cancer Agency, Vancouver, British Columbia, Canada (M.M.); and Vancouver General Hospital, Vancouver, British Columbia, Canada (N.M.)
| | - Montgomery Martin
- From the Departments of Medicine (K.R.H.) and Radiology (R.D.H.), University of British Columbia, 2775 Laurel St, 11th Floor, Vancouver, BC, Canada V5Z 1M9; BC Cancer Agency, Vancouver, British Columbia, Canada (M.M.); and Vancouver General Hospital, Vancouver, British Columbia, Canada (N.M.)
| | - Nicolas Murray
- From the Departments of Medicine (K.R.H.) and Radiology (R.D.H.), University of British Columbia, 2775 Laurel St, 11th Floor, Vancouver, BC, Canada V5Z 1M9; BC Cancer Agency, Vancouver, British Columbia, Canada (M.M.); and Vancouver General Hospital, Vancouver, British Columbia, Canada (N.M.)
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15
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Soluk-Tekkesin M, Vered M. Ameloblastic Fibro-Odontoma: At the Crossroad Between "Developing Odontoma" and True Odontogenic Tumour. Head Neck Pathol 2021; 15:1202-1211. [PMID: 33991318 PMCID: PMC8633217 DOI: 10.1007/s12105-021-01332-6] [Citation(s) in RCA: 12] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/16/2021] [Accepted: 05/05/2021] [Indexed: 11/25/2022]
Abstract
Ameloblastic fibro-odontoma (AFO) is a controversial, rare benign mixed odontogenic tumour that was re-defined as "developing odontoma" in the 2017 WHO classification arguing that once dental hard tissues form, it is programmed to transform into odontoma. However, AFO still remains unclear in terms of its nature. We aimed to analyze a large series of AFOs and compare it to a large series of odontomas (ODs) in an attempt to set cut-off diagnostic parameters between these entities and discuss latest updates on AFO histopathologic, clinical and molecular features. A total of 23 well-documented AFOs were analyzed versus 310 ODs focusing on the age of the patients and size of the lesions. For AFO, mean age was 9.4 ± 3.9 years (range 3-16 years) and mean size (greatest diameter) was 2.9 ± 1.5 cm (range 0.8-5.5 cm). For OD-mean age was 26.5 ± 15.6 years (range 3-81 years), mean size 1.9 ± 0.9 cm (range 1-5 cm). Receiver operating curve (ROC) showed that a cut-off age of 13.5 years and below [area under the curve (AUC) 0.902, 95%CI 0.859-0.945; p < 001; sensitivity 80%, specificity 87%] and a cut-off size of 2.1 cm and above are likely to be associated with AFO (AUC 0.7, 95%CI 0.574-0.827; p = 0.001; sensitivity 57%, specificity 77%). Thus, the combination of age and lesion size may be used to distinguish between lesions of a true neoplastic nature (i.e., AFO) and hamartomatous formation (i.e., OD). Further molecular and genetic specifications are needed to provide a better understanding on the pathogenesis of AFO in support of our suggestion and aid in an accurate classification of AFO.
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Affiliation(s)
- Merva Soluk-Tekkesin
- Department of Tumour Pathology, Institute of Oncology, Istanbul University, Istanbul, Turkey
| | - Marilena Vered
- Department of Oral Pathology, Oral Medicine and Maxillofacial Imaging, School of Dental Medicine, Tel Aviv University, Tel Aviv, Israel ,Institute of Pathology, Sheba Medical Center, Tel Hashomer, Ramat Gan, Israel
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16
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Onda T, Yamazaki A, Hayashi K, Iwasaki T, Hashimoto K, Takano M. Ameloblastic Fibroma in Mandibular Anterior Tooth Region: A Case Report. THE BULLETIN OF TOKYO DENTAL COLLEGE 2021; 62:49-54. [PMID: 33583881 DOI: 10.2209/tdcpublication.2020-0031] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
An ameloblastic fibroma (AF) is a proliferative mixed tumor which includes components of both odontogenic epithelium and mesenchymal tissue. It is a relatively rare neoplasm, accounting for approximately only 1.5-4.5% of odontogenic tumors. This case report describes an AF that occurred in the mandibular anterior tooth region in a 9-year-old girl who presented with the chief complaint of swelling in the left mandibular anterior tooth region. Intraoral examination revealed a swelling along the labial gingiva, extending from the left mandibular lateral incisor region to the left mandibular canine. Upon palpation, the swelling appeared to comprise a hard tissue. Computed tomography revealed a supernumerary impacted tooth; soft tissue density in the bone surrounding the region extending from the left mandibular lateral incisor to the left mandibular canine; labial bone expansion; and thinning of the labial cortical bone. A biopsy was performed under local anesthesia and the lesion subsequently diagnosed as an AF. Tumor resection and extraction of the supernumerary impacted tooth were carried out under general anesthesia. At 2 years postoperatively the prognosis is good. Although relapse with an AF is rarer than that with an ameloblastoma, strict follow-up is required, as malignant transformation to an ameloblastic fibrosarcoma has been reported in relapsed cases.
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Affiliation(s)
- Takeshi Onda
- Department of Oral and Maxillofacial Surgery, Tokyo Dental College
| | - Azusa Yamazaki
- Department of Oral and Maxillofacial Surgery, Tokyo Dental College
| | | | - Takahiro Iwasaki
- Department of Oral and Maxillofacial Surgery, Tokyo Dental College
| | - Kazuhiko Hashimoto
- Division of Surgical Pathology, Clinical Laboratory, Ichikawa General Hospital, Tokyo Dental College
| | - Masayuki Takano
- Department of Oral and Maxillofacial Surgery, Tokyo Dental College
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17
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Oh KY, Cho SD, Yoon HJ, Lee JI, Hong SD. Discrepancy between immunohistochemistry and sequencing for BRAF V600E in odontogenic tumours: Comparative analysis of two VE1 antibodies. J Oral Pathol Med 2020; 50:85-91. [PMID: 32939809 DOI: 10.1111/jop.13108] [Citation(s) in RCA: 15] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/25/2020] [Revised: 08/14/2020] [Accepted: 09/06/2020] [Indexed: 11/29/2022]
Abstract
BACKGROUND Although immunohistochemistry (IHC) along with molecular tests has been investigated in ameloblastoma for BRAF V600E detection, VE1 IHC has not been studied in odontogenic carcinomas (OCs) and benign mixed epithelial and mesenchymal odontogenic tumours (BMOTs). Here, we performed BRAF V600E mutation analysis, examined the expression pattern of VE1 IHC, and comparatively evaluated the performance of two VE1 antibodies in ameloblastomas, OCs and BMOTs. METHODS BRAF V600E detection was performed using Sanger sequencing in a total of 47 odontogenic tumours: 28 ameloblastomas, 6 OCs and 13 BMOTs. VE1 IHC was conducted using two different antibodies (IHC-A and IHC-V), and their performance was analysed by calculating the sensitivity and specificity compared with sequencing. RESULTS BRAF V600E mutations were identified in 24/28 (85.7%) ameloblastomas, 2/5 (40.0%) ameloblastic carcinomas (ACs), 3/7 (42.9%) ameloblastic fibromas and 1/2 (50.0%) ameloblastic fibro-odontomas. In the presence of the mutation, VE1 showed diffuse cytoplasmic staining in ameloblastomas and ACs, whereas all BMOTs were negative for VE1. IHC-A and IHC-V yielded a sensitivity of 76.7% and 60.0%, respectively, although both antibodies showed 100% specificity. CONCLUSION OCs and BMOTs have BRAF V600E mutations in common at lower frequencies than ameloblastoma. Diffuse VE1 cytoplasmic staining in AC suggests the utility of MAPK-targeted therapy as selectively applied in ameloblastoma, and consistent VE1 false-negative expression in BMOTs requires further investigation. Considering the high specificity but low sensitivity of VE1 IHC, molecular tests should be performed to determine the presence of BRAF V600E mutations in odontogenic tumours.
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Affiliation(s)
- Kyu-Young Oh
- Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University, Seoul, Korea
| | - Sung-Dae Cho
- Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University, Seoul, Korea
| | - Hye-Jung Yoon
- Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University, Seoul, Korea
| | - Jae-Il Lee
- Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University, Seoul, Korea
| | - Seong-Doo Hong
- Department of Oral Pathology, School of Dentistry and Dental Research Institute, Seoul National University, Seoul, Korea
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18
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Coura BP, Bernardes VF, de Sousa SF, Diniz MG, Moreira RG, de Andrade BAB, Romañach MJ, Pontes HAR, Gomez RS, Odell EW, Gomes CC. Targeted Next-Generation Sequencing and Allele-Specific Quantitative PCR of Laser Capture Microdissected Samples Uncover Molecular Differences in Mixed Odontogenic Tumors. J Mol Diagn 2020; 22:1393-1399. [PMID: 32966885 DOI: 10.1016/j.jmoldx.2020.08.005] [Citation(s) in RCA: 23] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/03/2020] [Revised: 08/13/2020] [Accepted: 08/27/2020] [Indexed: 02/07/2023] Open
Abstract
The molecular pathogenesis of mixed odontogenic tumors has not been established, and understanding their genetic basis could refine their classification and help define molecular markers for diagnostic purposes. Potentially pathogenic mutations in the component tissues of 28 cases of mixed odontogenic tumors were assessed. Laser capture microdissected tissue from 10 ameloblastic fibromas (AF), 4 ameloblastic fibrodentinomas (AFD), 6 ameloblastic fibro-odontomas (AFO), 3 ameloblastic fibrosarcomas (AFS), and 5 odontomas (OD) were screened by next-generation sequencing and results confirmed by TaqMan allele-specific quantitative PCR. BRAF p.V600E mutation in the mesenchymal component was shown in 4 of 10 AF (40%), 2 of 4 AFD (50%), 2 of 6 AFO (33%), and 2 of 3 AFS (67%), whereas all 5 OD were wild type for BRAF p.V600E. Mutation in the epithelial component was only observed in one AF and one AFO. One AFS contained an area of benign AF, and the mesenchymal component of both (AFS and AF) contained BRAF p.V600E, supporting the concept of malignant progression from a benign AF precursor. KDR, TP53, KIT, and PIK3CA single-nucleotide polymorphisms are reported. In conclusion, AF, AFD, AFO, and AFS show BRAF p.V600E in their mesenchymal component, unlike OD, which are BRAF wild type, suggesting that at least a subset of AF, AFD, and AFO are molecularly distinct from OD, and may represent distinct entities and be neoplastic.
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Affiliation(s)
- Bruna Pizziolo Coura
- Department of Pathology, Biological Sciences Institute, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | - Vanessa Fátima Bernardes
- Department of Pathology, Biological Sciences Institute, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | - Sílvia Ferreira de Sousa
- Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | - Marina Gonçalves Diniz
- Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | - Rennan Garcias Moreira
- Center of Multi-Users Laboratories-Genomics Laboratory, Biological Sciences Institute, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | | | - Mario José Romañach
- Department of Oral Diagnosis and Pathology, Dental School, Universidade Federal do Rio de Janeiro (UFRJ), Rio de Janeiro, Brazil
| | - Helder Antônio Rebelo Pontes
- Service of Oral Pathology, João de Barros Barreto University Hospital, Universidade Federal do Pará (UFPA), Belém, Brazil
| | - Ricardo Santiago Gomez
- Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil
| | - Edward William Odell
- Head and Neck Pathology, King's College London, Guy's Hospital, London, United Kingdom.
| | - Carolina Cavalieri Gomes
- Department of Pathology, Biological Sciences Institute, Universidade Federal de Minas Gerais (UFMG), Belo Horizonte, Brazil.
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19
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Omami G. Mandibular radiolucency with snowflakelike calcifications. J Am Dent Assoc 2020; 152:240-244. [PMID: 32631555 DOI: 10.1016/j.adaj.2020.03.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2020] [Revised: 02/23/2020] [Accepted: 03/13/2020] [Indexed: 11/27/2022]
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20
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Cossiez M, Del Pin D. A rare case of maxillary ameloblastic fibro-odontoma and a review of the literature. JOURNAL OF ORAL MEDICINE AND ORAL SURGERY 2020. [DOI: 10.1051/mbcb/2020010] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022] Open
Abstract
Introduction: Ameloblastic fibro-odontoma (AFO) is a benign complex odontogenic tumor in the mandible in children and adolescents. Observation: A 9-year-old boy was referred to the dental department for the delayed eruption of tooth 26. He was asymptomatic, and on clinical examination, we found that tooth 26 was absent with tumefaction instead of the tooth. Cone beam computed tomography revealed an opaque mass in the root of 26. The management was surgical, and anatomopathological examination facilitated the diagnosis of AFO. Commentary: The upper maxillary localization of AFO is rare and is not often a practitioner's first thought. Surgical excision of the tumor is essential based on the histological diagnosis. The patient must be followed up after 3 months and regularly until the eruption of the affected tooth. Conclusion: AFO is one of the differential diagnoses to be considered when a young patient presents with firm swelling with delayed eruption of the opposing tooth, especially since it has good prognosis and is easy to manage.
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21
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Developing odontoma with an atypical radiological appearance: A case report. ORAL AND MAXILLOFACIAL SURGERY CASES 2020. [DOI: 10.1016/j.omsc.2019.100138] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/18/2022] Open
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22
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Althubaiti S, Kafkas Ş, Abdelhakim M, Hoehndorf R. Combining lexical and context features for automatic ontology extension. J Biomed Semantics 2020; 11:1. [PMID: 31931870 PMCID: PMC6958746 DOI: 10.1186/s13326-019-0218-0] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/02/2018] [Accepted: 12/24/2019] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Ontologies are widely used across biology and biomedicine for the annotation of databases. Ontology development is often a manual, time-consuming, and expensive process. Automatic or semi-automatic identification of classes that can be added to an ontology can make ontology development more efficient. RESULTS We developed a method that uses machine learning and word embeddings to identify words and phrases that are used to refer to an ontology class in biomedical Europe PMC full-text articles. Once labels and synonyms of a class are known, we use machine learning to identify the super-classes of a class. For this purpose, we identify lexical term variants, use word embeddings to capture context information, and rely on automated reasoning over ontologies to generate features, and we use an artificial neural network as classifier. We demonstrate the utility of our approach in identifying terms that refer to diseases in the Human Disease Ontology and to distinguish between different types of diseases. CONCLUSIONS Our method is capable of discovering labels that refer to a class in an ontology but are not present in an ontology, and it can identify whether a class should be a subclass of some high-level ontology classes. Our approach can therefore be used for the semi-automatic extension and quality control of ontologies. The algorithm, corpora and evaluation datasets are available at https://github.com/bio-ontology-research-group/ontology-extension.
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Affiliation(s)
- Sara Althubaiti
- Computational Bioscience Research Center, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia.,Computer, Electrical and Mathematical Sciences and Engineering Division, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia
| | - Şenay Kafkas
- Computational Bioscience Research Center, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia.,Computer, Electrical and Mathematical Sciences and Engineering Division, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia
| | - Marwa Abdelhakim
- Computational Bioscience Research Center, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia.,Computer, Electrical and Mathematical Sciences and Engineering Division, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia
| | - Robert Hoehndorf
- Computational Bioscience Research Center, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia. .,Computer, Electrical and Mathematical Sciences and Engineering Division, King Abdullah University of Science and Technology, Thuwal, 23955-6900, Saudi Arabia.
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23
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Morice A, Neiva C, Fabre M, Spina P, Jouenne F, Galliani E, Vazquez MP, Picard A. Conservative management is effective in unicystic ameloblastoma occurring from the neonatal period: A case report and a literature review. Oral Surg Oral Med Oral Pathol Oral Radiol 2019; 129:e234-e242. [PMID: 31562035 DOI: 10.1016/j.oooo.2019.08.009] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/14/2019] [Revised: 07/28/2019] [Accepted: 08/26/2019] [Indexed: 12/28/2022]
Abstract
Unicystic ameloblastoma (UA), a benign odontogenic tumor of the jaw, represents less than a third of all ameloblastomas and seems less aggressive than other types of ameloblastoma. We present here the first case of UA that developed prenatally and was successfully managed in the early neonatal period with marsupialization and curettage performed carefully to avoid injury to the tooth germ. BRAF and SMO mutations were not detected. After 2 years of follow-up, complete reossification and normal eruption of deciduous teeth were noted, and there was no recurrence of UA. We recommend conservative treatment of UA in the pediatric population to avoid loss of and/or injury to the tooth germ, provided close follow-up is carried out all through the individual's growth for early detection of potential recurrences, growth impairments, or tooth eruption disorders. The intratumoral somatic mutational status of BRAF, SMO, RAS family, and FGFR2 may help determine personalized targeted treatment, particularly in case of recurrence.
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Affiliation(s)
- Anne Morice
- Department of Maxillofacial and Plastic Surgery, Rare Diseases Reference Center Coordinator for Clefts and Facial Malformations, Hôpital Universitaire Necker-Enfants Malades, Paris, France; Université Paris Descartes-Sorbonne Paris Cité, Paris, France.
| | - Cecilia Neiva
- Department of Maxillofacial and Plastic Surgery, Rare Diseases Reference Center Coordinator for Clefts and Facial Malformations, Hôpital Universitaire Necker-Enfants Malades, Paris, France
| | - Monique Fabre
- Department of Pathology, Hôpital Universitaire Necker-Enfants Malades, Assistance Publique-Hôpitaux de Paris, and Université Paris Descartes, Paris, France
| | - Paolo Spina
- Cantonal Institute of Pathology, Locarno, Switzerland; Department of Health Sciences, University of Eastern Piedmont, Novara, Italy
| | - Fanélie Jouenne
- Genomic of Solid Tumors Department, Hôpital Saint-Louis, Assistance Publique-Hôpitaux de Paris; Université Paris-Diderot, Sorbonne Paris Cité, Paris, France
| | - Eva Galliani
- Department of Maxillofacial and Plastic Surgery, Rare Diseases Reference Center Coordinator for Clefts and Facial Malformations, Hôpital Universitaire Necker-Enfants Malades, Paris, France
| | - Marie-Paule Vazquez
- Department of Maxillofacial and Plastic Surgery, Rare Diseases Reference Center Coordinator for Clefts and Facial Malformations, Hôpital Universitaire Necker-Enfants Malades, Paris, France; Université Paris Descartes-Sorbonne Paris Cité, Paris, France
| | - Arnaud Picard
- Department of Maxillofacial and Plastic Surgery, Rare Diseases Reference Center Coordinator for Clefts and Facial Malformations, Hôpital Universitaire Necker-Enfants Malades, Paris, France; Université Paris Descartes-Sorbonne Paris Cité, Paris, France
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24
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Chrcanovic B, Gomez R. Gorham–Stout disease with involvement of the jaws: a systematic review. Int J Oral Maxillofac Surg 2019; 48:1015-1021. [PMID: 30898430 DOI: 10.1016/j.ijom.2019.03.002] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/02/2019] [Accepted: 03/05/2019] [Indexed: 01/21/2023]
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25
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Chrcanovic B, Gomez R. Idiopathic bone cavity of the jaws: an updated analysis of the cases reported in the literature. Int J Oral Maxillofac Surg 2019; 48:886-894. [PMID: 30792085 DOI: 10.1016/j.ijom.2019.02.001] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/19/2018] [Revised: 12/19/2018] [Accepted: 02/04/2019] [Indexed: 10/27/2022]
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26
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Altamirano J, Salas M, Contreras A, Martínez F, Slater J, Barrera A, Cifuentes J. Ameloblastic fibroodontoma, literature review apropos of a case. Int J Oral Maxillofac Surg 2019. [DOI: 10.1016/j.ijom.2019.03.264] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/28/2022]
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27
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Pereira NB, de Souza JC, Bastos VC, Fonseca FP, de Avelar GF, Castro WH, Dias AAM, Mosqueda‐Taylor A, Gomez RS, Gomes CC. Patient‐derived xenografts of a case of ameloblastic fibrodentinoma. Oral Dis 2019; 25:1229-1233. [DOI: 10.1111/odi.13056] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/15/2018] [Revised: 01/04/2019] [Accepted: 01/25/2019] [Indexed: 12/26/2022]
Affiliation(s)
- Núbia B. Pereira
- Departament of Pathology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Juliana C. de Souza
- Departament of Pathology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Victor C. Bastos
- Departament of Pathology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Felipe P. Fonseca
- Departament of Oral Surgery and Pathology, School of Dentistry Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Gleide F. de Avelar
- Departament of Morphology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Wagner H. Castro
- Departament of Oral Surgery and Pathology, School of Dentistry Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Adriana A. M. Dias
- Departament of General Biology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | | | - Ricardo S. Gomez
- Departament of Oral Surgery and Pathology, School of Dentistry Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
| | - Carolina C. Gomes
- Departament of Pathology, Biological Science Institute Universidade Federal de Minas Gerais (UFMG) Belo Horizonte Brazil
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28
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Chrcanovic B, Gomez R. Ameloblastic fibrodentinosarcoma and ameloblastic fibro-odontosarcoma: A systematic review. JOURNAL OF STOMATOLOGY, ORAL AND MAXILLOFACIAL SURGERY 2018; 119:401-406. [PMID: 29574112 DOI: 10.1016/j.jormas.2018.03.007] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Received: 10/22/2017] [Revised: 01/15/2018] [Accepted: 03/18/2018] [Indexed: 11/25/2022]
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29
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Kale SG, Shetty A, Balakrishnan J, Purvey P. Ameloblastic Fibro-odontoma with a Predominant Radiopaque Component. Ann Maxillofac Surg 2017; 7:304-307. [PMID: 29264304 PMCID: PMC5717913 DOI: 10.4103/ams.ams_84_17] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Ameloblastic fibro-odontoma (AFO) is a rare odontogenic tumor. Initially believed to be a lesion similar to ameloblastic fibroma (AF), it is now considered as a separate entity in the WHO odontogenic tumor classification. Commonly associated with a painless swelling and an associated absence of eruption of a tooth, AFO presents as a mixed radiopaque and radiolucent lesion in the younger population with a predilection for the posterior region. Histologically, it shows the characteristics of an immature complex odontoma with irregularly arranged enamel, dentinoid, cementoid-like structures, and ectomesenchymal tissue. The following case report describes a case of AFO with a predominantly radiopaque component and briefly discusses the available literature pertaining to this rare entity.
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Affiliation(s)
- Saurabh Gajanan Kale
- Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Bengaluru, Karnataka, India
| | - Akshay Shetty
- Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Bengaluru, Karnataka, India
| | - Jintu Balakrishnan
- Department of Oral and Maxillofacial Pathology, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Bengaluru, Karnataka, India
| | - Praveen Purvey
- Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Bengaluru, Karnataka, India
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30
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Wright JM, Soluk Tekkesin M. Odontogenic tumors: where are we in 2017 ? J Istanb Univ Fac Dent 2017; 51:S10-S30. [PMID: 29354306 PMCID: PMC5750825 DOI: 10.17096/jiufd.52886] [Citation(s) in RCA: 36] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/05/2017] [Accepted: 08/04/2017] [Indexed: 11/30/2022] Open
Abstract
Odontogenic tumors are a heterogeneous group of
lesions of diverse clinical behavior and histopathologic
types, ranging from hamartomatous lesions to malignancy.
Because odontogenic tumors arise from the tissues which
make our teeth, they are unique to the jaws, and by extension
almost unique to dentistry. Odontogenic tumors, as in normal
odontogenesis, are capable of inductive interactions between
odontogenic ectomesenchyme and epithelium, and the
classification of odontogenic tumors is essentially based
on this interaction. The last update of these tumors was
published in early 2017. According to this classification,
benign odontogenic tumors are classified as follows:
Epithelial, mesenchymal (ectomesenchymal), or mixed
depending on which component of the tooth germ gives
rise to the neoplasm. Malignant odontogenic tumors are
quite rare and named similarly according to whether the
epithelial or mesenchymal or both components is malignant.
The goal of this review is to discuss the updated changes to
odontogenic tumors and to review the more common types
with clinical and radiological illustrations.
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Affiliation(s)
- John M Wright
- Department of Diagnostic Sciences, School of Dentistry, Texas A&M University, Dallas, TX USA
| | - Merva Soluk Tekkesin
- Department of Tumor Pathology, Institute of Oncology, Istanbul University, Istanbul Turkey
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31
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Chrcanovic BR, Brennan PA, Rahimi S, Gomez RS. Ameloblastic fibroma and ameloblastic fibrosarcoma: A systematic review. J Oral Pathol Med 2017; 47:315-325. [PMID: 28776760 DOI: 10.1111/jop.12622] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/01/2017] [Indexed: 11/29/2022]
Abstract
PURPOSE To integrate the available data published to date on ameloblastic fibromas (AF) and ameloblastic fibrosarcomas (AFS) into a comprehensive analysis of their clinical/radiological features. METHODS An electronic search was undertaken in July 2017. Eligibility criteria included publications having enough clinical, radiological and histological information to confirm a definite diagnosis. RESULTS A total of 244 publications (279 central AF tumours, 10 peripheral AF, 103 AFS) were included. AF and AFS differed significantly with regard to the occurrence of patients' mean age, bone expansion, cortical bone perforation and lesion size. Recurrence rates were as follows: central AF (19.2%), peripheral AF (12.5%), AFS (all lesions, 35%), primary (de novo) AFS (28.8%) and secondary AFS (occurring after an AF, 50%). Larger lesions and older patients were more often treated by surgical resections for central AF. Segmental resection resulted in the lowest rate of recurrence for most of the lesion types. AFS treated by segmental resection had a 70.5% lower probability to recur (OR 0.295; P = .049) than marginal resection; 21.3% of the AFS patients died due to complications related to the lesion. CONCLUSIONS Very long follow-up is recommended for AF lesions, due to the risk of recurrence and malignant change into AFS. Segmental resection is the most recommended therapy for AFS.
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Affiliation(s)
| | - Peter A Brennan
- Department of Oral and Maxillofacial Surgery, Queen Alexandra Hospital, Portsmouth, UK
| | - Siavash Rahimi
- Department of Histopathology, Queen Alexandra Hospital, Portsmouth, UK.,School of Pharmacy and Biomedical Sciences, University of Portsmouth, Portsmouth, UK
| | - Ricardo Santiago Gomez
- Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil
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Chrcanovic BR, Gomez RS. Glandular odontogenic cyst: An updated analysis of 169 cases reported in the literature. Oral Dis 2017; 24:717-724. [PMID: 28744957 DOI: 10.1111/odi.12719] [Citation(s) in RCA: 30] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/26/2017] [Revised: 06/27/2017] [Accepted: 07/17/2017] [Indexed: 11/29/2022]
Affiliation(s)
- B R Chrcanovic
- Department of Prosthodontics; Faculty of Odontology; Malmö University; Malmö Sweden
| | - R S Gomez
- Department of Oral Surgery and Pathology; School of Dentistry; Universidade Federal de Minas Gerais; Belo Horizonte Brazil
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New tumour entities in the 4th edition of the World Health Organization Classification of Head and Neck tumours: odontogenic and maxillofacial bone tumours. Virchows Arch 2017; 472:331-339. [PMID: 28674741 PMCID: PMC5886999 DOI: 10.1007/s00428-017-2182-3] [Citation(s) in RCA: 183] [Impact Index Per Article: 22.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/17/2017] [Revised: 06/14/2017] [Accepted: 06/19/2017] [Indexed: 02/06/2023]
Abstract
The latest (4th) edition of the World Health Organization Classification of Head and Neck tumours has recently been published with a number of significant changes across all tumour sites. In particular, there has been a major attempt to simplify classifications and to use defining criteria which can be used globally in all situations, avoiding wherever possible the use of complex molecular techniques which may not be affordable or widely available. This review summarises the changes in Chapter 8: Odontogenic and maxillofacial bone lesions. The most significant change is the re-introduction of the classification of the odontogenic cysts, restoring this books status as the only text which classifies and defines the full range of lesions of the odontogenic tissues. The consensus group considered carefully the terminology of lesions and were concerned to ensure that the names used properly reflected the best evidence regarding the true nature of specific entities. For this reason, this new edition restores the odontogenic keratocyst and calcifying odontogenic cyst to the classification of odontogenic cysts and rejects the previous terminology (keratocystic odontogenic tumour and calcifying cystic odontogenic tumour) which were intended to suggest that they are true neoplasms. New entities which have been introduced include the sclerosing odontogenic carcinoma and primordial odontogenic tumour. In addition, some previously poorly defined lesions have been removed, including the ameloblastic fibrodentinoma, ameloblastic fibro-odontoma, which are probably developing odontomas, and the odontoameloblastoma, which is not regarded as an entity. Finally, the terminology “cemento” has been restored to cemento-ossifying fibroma and cemento-osseous dysplasias, to properly reflect that they are of odontogenic origin and are found in the tooth-bearing areas of the jaws.
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Bavle RM, Muniswammappa S, Venugopal R, R AS. Ameloblastic Fibrodentinoma: A Case with Varied Patterns of Dysplastic Dentin. Cureus 2017; 9:e1349. [PMID: 28721317 PMCID: PMC5510976 DOI: 10.7759/cureus.1349] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/27/2022] Open
Abstract
Ameloblastic fibrodentinoma is a benign odontogenic tumor belonging to the category of "odontogenic epithelium with odontogenic ectomesenchyme" along with recognition of induction in the form of dentin in atypical or dysplastic forms. The biological behaviour of ameloblastic fibrodentinoma is not very different from ameloblastic fibroma; hence, it is treated similarly by conservative procedures. It is important to understand the histopathogenesis of these rare tumors. Though rare, they are an independent entity awaiting recognition. Here, we report a case of amelobalstic fibrodentinoma in a 14-year-old female patient.
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Affiliation(s)
- Radhika M Bavle
- Department of Oral and Maxillofacial Pathology, Krishnadevaraya College of Dental Sciences and Hospital
| | - Sudhakara Muniswammappa
- Department of Oral and Maxillofacial Pathology, Krishnadevaraya College of Dental Sciences and Hospital
| | - Reshma Venugopal
- Department of Oral and Maxillofacial Pathology, Krishnadevaraya College of Dental Sciences and Hospital
| | - Amulya S R
- Department of Oral and Maxillofacial Pathology, Krishnadevaraya College of Dental Sciences and Hospital
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