Malignant Odontogenic Tumours: A Systematic Review of Cases Reported in Literature

Background: Malignant odontogenic tumours (MOTs) arise either de novo from the tooth forming tissues, their developmental residues or from existing odontogenic epithelial or mesenchymal neoplasms in the jaws. Their management requires extensive surgery due to their infiltrative nature and risk of metastasis. There is a need to understand the clinical and pathological features of MOTs to inform both treatment algorithms and prognostication. This is an area of diagnostic pathology which presents substantial difficulties in diagnosis, compounded by inconsistent use of terminology. Thus, this systematic review aimed to describe the clinical and pathological features of MOTs with a view to consolidating the literature and defining problematic areas in diagnosis and classification.Methods: An electronic database search was conducted in Web of Science, PubMed/Medline, and Embase. Additionally, the grey literature and reference lists of selected papers searched for completeness. Nine hundred and sixty articles were initially identified. Following removal of duplicates and application of inclusion/exclusion criteria, 312 articles were included for qualitative analysis.Results: The 312 articles encompassed a total of 507 patients with most lesions located within the mandible (74.3%). The most common first histological diagnosis was ameloblastic carcinoma (25.7% of all diagnoses), but there is considerable variation in how and when various diagnostic terms are used, and several misdiagnoses were reported. An initial benign diagnosis was made in 24.7% of patients, followed by a later malignant diagnosis and in this sub-group, the most common benign first diagnosis was ameloblastoma (42.4%). Cervical lymph nodes were the most common site of metastasis (9.3% of patients). With respect to distant metastasis (DM), the lungs were the most common organ affected (11.2% of DM patients) with metastasising ameloblastoma the most commonly reported tumour which metastasised to the lungs. Overall, 26.8% of patients developed recurrence.Conclusion: Overall, the quality of the literature on MOTs is poor. This review of the literature has highlighted variations in diagnostic terms and criteria which has resulted in areas of confusion with potential for misdiagnosis. This consolidation of primary data has identified key areas for targeted research including further discussion on the malignant potential of ameloblastoma.

Erupting Compound Odontome - A Case Report

Odontomas are asymptomatic, benign odontogenic tumours comprised of dental tissue. Most of the cases usually get detected on routine radiographic evaluation. Odontoma can be classified according to morphology in two different variants, compound odontoma showing anatomic similarity or miniature version of the tooth and complex odontoma having irregular masses with different type of dental tissues. Early detection of such type of silent lesion is mandatory to rule out certain silent but aggressive pathologies. Here, we present a case of erupting compound odontoma of maxillary area in 15-year-old male patient. Development of tooth is a result of complicated interaction between ectodermal and ecto mesenchymal tissue. Sometimes the remnants of such tissues are left behind in the jaw after development and eruption of tooth, which can become a source of development of odontogenic lesions later on.1 As revealed by World Health Organization (WHO), Odontogenic tumours can be classified in three different categories according to their histopathological form.2 1. Odontogenic tumours which develop from odontogenic epithelium without involvement of odontogenic ectomesenchyme, 2. Odontogenic tumours which develop from odontogenic epithelium with involvement of odontogenic ectomesenchyme, with or without formation of dental hard tissue and 3. Odontogenic tumours which develop from odontogenic ectomesenchyme with or without involvement of odontogenic epithelium.2 For such lesions “Odontoma” word was initially given by Paul Broca in 1866, who revealed the term as tumour occurred by the overgrowth of entire dental tissue. The growth of absolutely differentiated epithelial cells as functional ameloblast and mesenchymal cells as functional odontoblast can give rise to a different developmental anomaly coined as Odontomas.3 According to their behaviour and composition, odontomas can be considered as malformations or hamartomas rather than a true neoplasm. 4,5,6 In 2005, World Health Organization (WHO) classified two variants of odontomas, 1. Compound odontomas, which typically appears as unilocular lesions containing multiple radiopaque, tiny tooth like structures commonly known as denticles; and 2. Complex odontomas, which is comprised of an irregular mass of soft and hard dental tissues.

Clinico-pathological study of malignant odontogenic tumours from a national referral centre

Background Malignant odontogenic tumours are extremely rare tumours occurring within the jaws. Our study was performed to determine the demographic and clinico-pathological features of malignant odontogenic tumours amongst a multi-ethnic Asian population. Methods This was a retrospective cross-sectional study of malignant odontogenic tumours diagnosed at the Institute for Medical Research, Malaysia, from 2009 to 2019. All cases were independently reviewed and reclassified following the criteria set out in the latest edition of the World Health Organization 2017 reference text. Demographic and clinico-pathological data were recorded for each case. Results Twenty-four cases of malignant odontogenic tumours were identified. The patients’ age ranged from 16 to 79 years with the mean age at diagnosis being 50.8 years (SD = 16.18). There was a male predominance (66.7%) in this cohort of patients. The ethnic distribution appeared to reflect the Malaysian population with most cases seen amongst the Malay ethnic group (66.7%). Ameloblastic carcinoma was the most frequently diagnosed malignant odontogenic tumour (45.8%) and was also predominantly seen in males (90.9%). All patients with clear cell odontogenic carcinoma were females. There was no obvious sex predilection in primary odontogenic carcinoma not otherwise specified (NOS). The mandible (79.2%) was more frequently involved compared to the maxilla. Conclusions Diagnosis and management of malignant odontogenic tumours are challenging due to the rarity of these tumours. Our study has elucidated the clinico-pathological features of malignant odontogenic tumours seen in a multi-ethnic Asian population.

Adenomatoid Odontogenic Tumour - The Master of Disguise

Adenomatoid odontogenic tumour, truly coined as one of the masters of disguise of orofacial pathologies, was first reported in the literature by Steen Lands.1 Philipson and Brin used the terminology adenomatoid odontogenic tumour for this pathology with its commonly accepted abbreviation AOT.2 Later on, adenomatoid odontogenic tumour (AOT) name was accepted by the World Health Organization (WHO) in 1971. In 2005, WHO revealed the histological variants of the adenomatoid odontogenic tumour and classified it as a tumour comprised of odontogenic epithelium showing various patterns in histopathologic view within a mature connective tissue stroma.3 It is seldom noticed neoplasm which comprises only 3 % of all the odontogenic tumours. It was commonly found in the maxilla with female predilection and mostly in association with impacted canines.4-6 Adenomatoid odontogenic tumour is an odontogenic epithelial tumour usually seen in females in their second decade of life. The tumour is slow growing in nature which eventually results in painless expansion of jaw. The maxilla is commonly affected than mandible. Being benign in nature, most of the AOT cases usually got treated with conservative surgical enucleation but the greater size of tumour can leave behind an oro-facial defect. To prevent such type of incident, it is important to diagnose them early and treat accordingly. Herewith, we are presenting a case report of adenomatoid odontogenic tumour of mandible in a male patient.

Calcifying odontogenic cysts: A 20-year retrospective clinical and radiological review

Objective: Calcifying odontogenic cysts (COCs) exhibit diverse clinical behaviours and may be associated with other benign odontogenic tumours. In this study, the clinical and radiological features of COCs were analysed according to subtypes based on the classification by Praetorius et al. Emphasis was placed on cases exhibiting atypical or aggressive radiological appearances. This information may assist the clinician to better understand the radiological spectrum of COCs. Methods: Histologically confirmed cases of COCs were retrospectively reviewed in a 20-year period from three tertiary institutions. The following clinical information was reviewed: patient demographics, main complaint, clinical duration, anatomical site and detailed radiological features. Results: Twenty-seven cases of COCs were included in the study. Asymptomatic swelling was the main clinical presentation with infrequent reports of associated pain. COCs had an anterior mandibular predilection. Well-demarcated borders were seen in all cases with isolated cases showing focal areas with loss of demarcation. Unilocular lesions were more common than multilocular variants. Internal calcifications were frequent and six cases presented with associated odontomas. Maxillary COCs resulted in the displacement of the maxillary sinus and/or nasal cavity walls. Radiological signs of aggression, including cortical destruction, were noted in a few cases. Conclusion: Given the fact that COCs can present with a spectrum of clinical behaviours and radiological presentations, the academic debate regarding the cystic versus neoplastic nature of the entity is justifiable. The cases in the current sample presented with diverse presentations, ranging from indolent to lesions with significant growth and aggression.

Contribution of three-dimensional images in the planning of cementoblastoma resection

Cementoblastomas are rare benign tumours that represent less than 1% of all odontogenic tumours. Complete resection is mandatory to avoid recurrence. This case report describes the contribution of three-dimensional imaging and three-dimensional printing in the pre-operative surgical planning of a large cementoblastoma that not only caused substantial compression on the inferior alveolar and mental nerves, but also caused thinning and partial erosion of the lingual and vestibular cortical bone, thus increasing the risk of pre-operative mandibular fracture.

Central odontogenic fibroma of simple type: An original observation

Central odontogenic fibroma is an uncommon, benign, slow-growing intraosseous mesenchymal odontogenic tumour. It presents a diagnostic dilemma to the clinician and the pathologist because its clinical and radiological features resemble other odontogenic and/or non-odontogenic tumours, and the differential diagnosis is based on histological examination. In this report, we describe our experience with a case of a 23-year-old female patient with central odontogenic fibroma of the mandible that was diagnosed as ‘simple type’. Highlighting a subtype that was dropped from the last World Health Organization classification of head and neck tumours is important to accumulate more information about this lesion and to show its different features. Despite its rarity, central odontogenic fibroma should be included in the differential diagnosis of intrabony tumours of the jaws. These findings can better educate oral and maxillofacial surgeons about the unusual nature of this lesion, help establish a correct diagnosis and give the appropriate therapeutic management.

Benign Odontogenic Tumours

Odontogenic tumours of the maxillofacial region presents with a large number of histologic patterns and are derived from the primordial tooth forming tissues. They can occur most commonly in mandible and maxilla. The recent WHO classification helps in developing the appropriate treatment plan and categorizing the tumours. The present chapter deals with various benign odontogenic tumours with their clinical presentation and management in detail, based on clinical scenarios.

Queratocisto odontogênico em mandíbula: relato de caso

Introdução: O queratocisto odontogênico é por definição um cisto de desenvolvimento que apresenta caráter agressivo afetando o complexo bucomaxilofacial. Tendo em vista que muitas vezes são assintomáticos, o seu diagnóstico normalmente é obtido através de exames radiográficos de rotina. Objetivo: apresentar um caso clínico referente ao diagnóstico e tratamento de um queratocisto odontogênico em região posterior de mandíbula. Caso clínico: Paciente do sexo feminino, normosistemica, feoderma. Observou-se aumento de volume extraoral, lado direito da face em região de ângulo mandibular. Ao exame radiografico periapical, observou-se uma imagem radiolúcida, bem delimitada, unilocular, associada ao dente 47. Ao exame físico intraoral, observou-se abaulamento ósseo na região do referido dente. Foi então solicitada uma tomografia computadorizada, na qual foi detectada extensa lesão óssea expansiva em ramo mandibular direito. Realizou-se uma punção aspirativa local, detectando presença de conteúdo líquido. Primeiramente foi realizada uma biópsia incisional com instalação de dispositivo descompressivo. O laudo histopatológico foi de cisto odontogênico inflamatório. O dispositivo de descompressão permaneceu por 9 meses, apresentando uma diminuição significativa da lesão. Foi realizada então, a enucleação da lesão remanescente, associado a exodontia dos dentes 47 e 48 e enviado material para histopatológico, que dessa vez, confirmou diagnóstico de queratocisto odontogênico. Ao exame de imagem pós-operatório de 6 meses, observou-se formação óssea completa sem sinais de recidiva. Conclusão: O tratamento proposto se mostrou eficaz no tratamento do queratocisto odontogênico, o acompanhamento com exames de imagem é de extrema importância, tendo em vista a possibilidade de recidiva. Descritores: Recidiva; Cistos Odontogênicos; Patologia Bucal. Referências Freitas DA, Veloso DA, Santos ALD, Freitas VA. Ceratocistoodontogênico maxilar: relato de caso clínico. RGO Rev Gauch Odontol. 2015;63(4):484-88. Antunes AA, Avelar RL, Santos TS, Andrade ESS, Dourado E. Tumor odontogênico ceratocístico: análise de 69 casos/ Keratocystic odontogenic tumor: analysisof 69 cases. Rev bras cir cabeça pescoço. 2007;36(2):80-2. Moura BS, Cavalcante MA, Hespanhol W. Tumor odontogênico ceratocístico. Rev Col Bras Cir. 2016;43(6):466-71. Wright JM, Vered M. Update from the 4th Edition of the World Health Organization Classification of Head and Neck Tumours: Odontogenic and Maxillofacial Bone Tumors. Head Neck Pathol. 2017;11(1):68-77. Neville BW, Allen CM, Damm DD, Chi A. oral and maxillofacial pathology, 4. ed. Rio de Janeiro: Elsevier; 2016. Aciole GTS, Santos MAM, Aciole JMS, Ribeiro Neto N, Pinheiro, ALB. Tumor odontogênicoqueratocistorecidivante: tratamento cirúrgico conservador ou radical? Relato de caso clínico. Rev cir traumatol buco-maxilo-fac. 2010;10(1):43-8. Hupp JR. Cirurgia oral e maxilofacial contemporânea. 6. ed. Rio de Janeiro: Elsevier, 2015. Marques JAF, Neves, JL, Alencar, DA, Lemos IM, Marques LC. Ceratocisto Odontogênico: relato de caso. Sitientibus. 2006;34(1):59-69. Conceição ACA, Santos AM, Santos GP, Almeida AJ, Dias AMN, Mainenti P. Tumor odontogênico queratocístico: atualidades. RIEE. 2012;4(1): 29-35. Pereira CCS, Carvalho ACG de S, Jardim ECG, Shinohara EH, Garcia Júnior IR. Tumor Odontogênico Queratocístico e considerações diagnósticas. RBCS. 2012;10(32):73-9. Balmick S, Hespanhol W, Cavalcante MAA, Gandelmann IHA. Recidiva do Tumor Odontogênico Ceratocístico: Análise retrospectiva de 10 anos. Rev cir traumatol buco-maxilo-fac. 2011;11(1):85-91. Borghesi A, Nardi C, Giannitto C, Tironi A, Maroldi R, Di Bartolomeo F, Preda L. Odontogenic keratocyst: imaging features of a benign lesion with an aggressive behaviour. Insights Imaging. 2018;9(5):883-97. Johnson NR, Batstone MD, Savage NW. Management and recurrence of keratocystic odontogenic tumor: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;116(4):e271-76. Araújo S, Oliveira LKR, Pigatti FM, Mayrink G. Queratocisto odontogênico em região anterior da maxila: relato de caso. HU rev. 2019;45(1):82-6. Oliveira Júnior HCC, Chaves Netto HDM, Rodrigues MTV, Pinto JMV, Nóia CF. Descompressão cirúrgica no tratamento de lesões císticas da cavidade oral. Rev cir traumatol buco-maxilo-fac. 2014;14(1):15-20. Pazdera J, Kolar Z, Zboril V, Tvrdy P, Pink R. Odontogenic keratocysts/keratocystic odontogenic tumours: biological characteristics, clinical manifestation and treatment. Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub. 2014;158(2):170-74.