| Abstract|| |
Context: Odontogenic tumors (OTs) represent a rare subset of pathologies of the oral and maxillofacial region. The classification of OTs has undergone several changes over the years due to a lack of uniform international identification criteria. The histomorphological similarity and the many variations in behavioral patterns elaborated by these lesions warrant research. Aims: Using the update from the fourth edition of the World Health Organisation Classification of Head and Neck Tumors (2017), this dental institution carried out an epidemiological study on OTs in the state of Goa (India) and compared the data obtained with similar studies on OTs done within India. Materials and Methods: The clinical and pathological data of OTs from August 1996 to December 2016 was retrieved from the oral and maxillofacial pathology department archives, belonging to the lone dental college and hospital in the state of Goa, India. Demographic data such as frequency, age, gender, and site along with pathological subtype was analysed. Statistical Analysis Used: Descriptive analysis (i.e. frequency of age, gender, and location), Chi-Square Test and Fischer Exact test. Results: The relative frequency of OTs was 2.61% of all oral biopsied specimens. The most common OT encountered was ameloblastoma (57.9%). The posterior aspect of the mandible was the most favored site (77.2%). The frequency of OTs decreased after the fourth decade. An overall marginal male predilection (55%) was seen. Conclusions: This study contributes to the establishment of a comprehensive loco-regional epidemiological database on OTs in India, aiding research on their aetio-pathogenesis and diagnosis.
Keywords: Epidemiology, jaws, odontogenic tumors
|How to cite this article:|
Syed S, Carvalho KM, Spadigam A, Dhupar A. Clinico-pathological correlations of odontogenic tumors: Some critical observations based on a 20 year institutional study and a comprehensive review of literature. Indian J Dent Res 2019;30:516-20
|How to cite this URL:|
Syed S, Carvalho KM, Spadigam A, Dhupar A. Clinico-pathological correlations of odontogenic tumors: Some critical observations based on a 20 year institutional study and a comprehensive review of literature. Indian J Dent Res [serial online] 2019 [cited 2020 Jul 8];30:516-20. Available from: http://www.ijdr.in/text.asp?2019/30/4/516/271064
| Introduction|| |
The tooth forming apparatus of the upper and lower jaw gives rise to myriad pathologies, of which odontogenic tumors (OTs) account for not more than 3% of all diagnosed oral and maxillofacial lesions.
Based on the tissue of origin, OTs were classified by the World Health Organisation (WHO) in 1971 into three categories: “Neoplasm and other tumors related to the odontogenic apparatus,” “Neoplasm and other lesions related to bone,” and “Epithelial Cysts”. In 1992, the WHO subcategorised OTs based on their tissue of origin. In 2002 and 2005, the WHO further modified the classification on the basis of advances made in the field of tumor biology and genetics.,,, An update from the fourth edition of the World Health Organisation Classification of Head and Neck Tumors: featuring Odontogenic and Maxillofacial Bone Tumors was published in January 2017. In this update, odontogenic keratocyst and calcifying odontogenic cyst were placed under the odontogenic cyst category and cemento-ossifying fibroma was placed under the OT category based on their inherent nature.
So far, there has been no consensus regarding the aetio-pathogenesis dictating the incidence-prevalence pattern of OTs. Asian studies show conflicting results with respect to the most common OT encountered.,, The Indian OT scenario, however, remains uncharted as data from roughly two-thirds of the country are unknown.,,,
The objective of this study from the state of Goa is aimed at adding to the existing Indian repository of data on OTs. This would contribute to the future establishment of a National Registry for OTs. There are no previous published reports on the epidemiology of OTs from the state. Since Goa has only one dental institution with a surgical set-up, it serves as a database for the OT burden generated in the state.
To the best of our knowledge, after a thorough literature search, this is the first epidemiological study, which is based on the WHO 2017 classification of OTs.
| Materials and Methods|| |
Retrospective data on OTs from August 1996 to December 2016 from the Department of Oral Maxillofacial Pathology was reviewed.
In this study, the oral lesions defined as OTs by the WHO 2017 Classification of Head and Neck Tumors: Odontogenic and Maxillofacial Bone Tumors were included. Clinical and surgical notes were used to assess clinical parameters of age, gender, site, and histopathology.
A review of hematoxylin and eosin stained sections of OT cases was done by three oral pathologists in order to confirm the diagnosis. Assessment of the site of occurrence of the OTs in the jaw bones was done by dividing the maxilla and mandible into three sextants each (i.e. one anterior and two posterior sextants). The anterior sextant was defined as the distal aspect of the canine on the right to the distal aspect of the canine on the left, in each jaw. The posterior sextant was defined as the mesial aspect of the first premolar to the distal aspect of the last erupted molar in the jaw.
The clinical and pathological data was analysed using the SPSS software (version 20). Descriptive analysis (i.e. frequency analysis for age, gender, and location), Chi-Square test and Fischer exact test were used to compare the parameters of age, gender, and location with tumor histopathology.
The English literature on OTs was reviewed and the available data from Indian and Asian studies was comprehensively analysed.
| Results|| |
The relative frequency of OTs was 2.61% of all oral biopsied specimens encountered between August 1996 and December 2016. Of the 114 cases of OTs recorded, only one was malignant.
Histopathological analysis revealed that the three most common OTs encountered were the ameloblastoma (57.9%), cemento-cement-ossifying fibroma (COF) (18.4%), and adenomatoid odontogenic tumor (AOT, 10.5%). The other OTs seen were odontoma (7.0%), odontogenic fibroma (OF, 1.8%), dentinogenic ghost cell tumor (DGCT, 0.9%), ameloblastic fibroma (AF, 0.9%), cementoblastoma (0.9%), odontogenic myxoma (OM, (0.9%), and ameloblastic carcinoma (AC, 0.9%).
Comparing the anatomical anterior and posterior jaw segments of maxilla and mandible, the posterior aspect of the mandible was the most favored site (77.2%). AOT was seen exclusively in anterior compartment of both jaws, showing a predisposition for the maxillary jaw. The only malignant OT was seen in anterior mandible [Figure 1].
|Figure 1: Anatomic distribution of odontogenic tumors in upper and lower jaws, as seen in this study|
Click here to view
In this study, the third and fourth decade together accounted for 96.5% of the case load. An overall marginal male predilection (55%) was seen. Gender analysis of individual OTs showed that ameloblastoma and COF did not favor any gender. AOT, DGCT, AF, AC, and OF were seen exclusively in females, whereas odontoma, cementoblastoma, and OM favored the male gender.
| Discussion|| |
The relative frequency of OTs found in this study cannot be compared with the results of other studies. This is due to the recategorisation of odontogenic keratocyst and other odontogenic lesions in the WHO classifications over a period of time, as stated in the introduction.,,,,
The male predilection shown by OTs in the state of Goa is seen in other Indian states such as such as Maharashtra, Kerala, and Gujarat.,,,, Gender distribution in Asian studies favors the male gender, as is the pattern shown by studies from Nigeria, Canada, and Egypt.,,, In contrast, a female predisposition is reported in South America and Europe.
India, Asia, Europe, and Africa report a peak incidence of OTs in the third decade of life.,,,,,
Conforming to studies across the world, the most common site for OTs was found to be the posterior aspect of the mandible. However, specific OTs such as AOT are found exclusively in the anterior segment of both the jaws.,,,,,,,,, Interestingly, it is established that the most common impacted tooth found in the oral cavity is the third molar, followed by the permanent canine that could contribute to the specific site predilection of OTs within the jaw bones.,,
Ameloblastoma is the most common OT in most Asian countries.,,,,,,,,, Studies from USA, South America, and Europe have reported odontoma as the most common OT., Only one study from India reported odontoma to be the most predominant OT. The existing data on odontomes in India, however, may not reflect the actual scenario because in most Indian states, odontomes are rarely sent for histopathological analysis.
The WHO 2017 Classification for Head and Neck Tumors has placed COF in the benign OT category because of its unique location within jaw bones and its origin from the periodontal ligament. The new classification does not seem to differentiate the peripheral and central forms of COF. It is known that the periodontal ligament contains multipotent stem cells capable of differentiating into a spectrum of mature mesenchymal cells, which include cementoblasts and osteoblasts. It is hypothesized that microtrauma to the tooth bearing area of the jaws caused by plaque and masticatory load could trigger the stem cell component in the periodontal ligament to proliferate and differentiate giving rise to deposition of cementum and bone.,,, The most common age of presentation for COF is the second and third decade and the female gender is predominantly affected., Studies reporting on the site of presentation of the peripheral COF report anterior maxilla to be the most common site, whereas posterior mandible is the preferred site of presentation for central lesions., All the cases included in this study were peripheral COFs, which showed an equal gender predilection and favored the posterior mandible.
Comparing the demographics of individual OTs within India, Andhra Pradesh and Gujarat are the only two states to report a male gender predisposition and an equal gender distribution, respectively, for AOT, compared with a female predominance shown by other states., Since OTs such as DGCT, AF, OF, cementoblastoma, and OM have very low incidence rates, comparing their demographic data becomes difficult [Table 1]. Calcifying Epithelial OT and squamous OT have not been reported in the state till date. The rarity of these OTs in other Indian states results in a difficulty in commenting on their epidemiology.
|Table 1: Odontogenic tumor demographic data in India based on published reports|
Click here to view
In the present study, the overall clinicopathological pattern of OTs reported in Goa is similar to other Indian states. It is important to note, however, that the OT scenario of a majority of north Indian states remains unknown due to paucity of published studies.
The temporospatial pattern elaborated by OTs reflects the investigative potential of epigenetics in the aetiopathogenesis of OTs. Prophylactic measures to curb any focus of inflammation and/or microtrauma in the alveolar region of jaw bones, such as elective extractions of impacted teeth, warrants further research so as to investigate their role in the incidence and prevalence patterns of OTs.
Department of Oral and Maxillofacial Surgery, Goa Dental College and Hospital for supplying the clinical data and the biopsied tissue material.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Imran A, Jayanthi P, Tanveer S, Gobu SC. Classification of odontogenic cysts and tumors – Antecedents. J Oral Maxillofac Pathol 2016;20:269-71.
] [Full text]
Philipsen HP, Reichart PA. Revision of the 1992-edition of the WHO histological typing of odontogenic tumor. A suggestion. J Oral Pathol Med2002;31:253-8.
Kramer IRH, Pindborg JJ, Shear M. The WHO histological typing of odontogenic tumours-a commentary on the second edition. Cancer 1992;70:2988-94.
Barnes L, Eveson JW, Reichart P, Sidransky D. Odontogenic tumors. In: Philipsen HP, Reichart PA, Slootweg PJ, Slater LJ, editors. World Health Organization Classification of Tumours-Pathology and Genetics of Head and Neck Tumours. Lyon: IARC Press; 2005. p. 283-327.
Wright JM, Vered M. Update from the 4th
edition of the World Health Organisation classification of head and neck tumors: Odontogenic and maxillofacial bone tumors. Head Neck Pathol 2017;11:68-77.
Goteti SH. Odontogenic tumors: A review of 675 cases in Eastern Libya. Niger J Surg 2016;22:37-40.
] [Full text]
Naz I, Mahmood MK, Akhtar F, Nagi HA. Clinicopthological evaluation of tumors in Pakistan- A seven years retrospective study. Asian Pac J Cancer Prev 2014;15:3327-30.
AlSheddi MA, AlSenani MA, AlDosari AW. Odontogenic tumors: Analysis of 188 cases from Saudi Arabia. Ann Saudi Med 2015;35:146-50.
Kowkabi M, Razavi SM, Khosravi N, Navabi AA. Odontogenic tumors in Iran, Isfahan: A study of 260 cases. Dent Res J (Isfahan) 2012;9:725-9.
Varkhede A, Tupkari JV, Sardar M. Odontogenic tumors: A study of 120 cases in an Indian teaching hospital. Med Oral Patol Oral Cir Bucal 2011;16:895-9.
Kadashetti V, Chaudhary M, Patil S, Gawande M, Shivakumar KM, Badiyani BK. Odontogenic tumors; a retrospective study of 102 cases. Int J Oral Care Res 2014;2:7-11.
Ebenezer V, Ramalingam B. A cross-sectional survey of prevalence of odontogenic tumours. J Maxillofac Oral Surg 2010;9:369–74.
Sivapathamsundaram B. Odontogenic tumor: Indian scenario. J Oral Maxillofac Pathol 2017;21:2-3.
] [Full text]
Niranjan KC, Shaikh Z. Clinicopathological correlation of odontogenic cysts and tumours in a South Indian population over a 20-year period. Int J Dent Res 2014;2:32-6.
Pandiar D, Shameena PM, Sudha S, Varma S, Manjusha P, Banyal VS, et al
. Odontogenic tumors: A 13-year retrospective study of 395 cases in a South Indian teaching institute of Kerala. Oral Maxillofac Pathol J2015;6:602-8.
Singhal A, Singhal P, Ramesh V, Balamurali PD. A retrospective study of odontogenic tumours in a South Indian population. Indian J Dent Sci 2013;3:1-3.
Gill S, Chawda J, Jani D. Odontogenic tumors in Western India (Gujarat): Analysis of 209 cases. J Clin Exp Dent 2011;3:78-83.
Luo HY, Li TJ. Odontogenic tumors: A study of 1309 cases in a Chinese population. Oral Oncol 2009;45:706-11.
Sekerci AE, Nazlım S, Etoz M, Denız K, Yasa Y. Odontogenic tumors: A collaborative study of 218 cases diagnosed over 12 years and comprehensive review of the literature. Med Oral Patol Oral Cir Bucal 2015;20:34-44.
Ramos GD, Porto JC, Vieira DS, Siqueira FM, Rivero ER. Odontogenic tumors: A 14-year retrospective study in Santa Catarina, Brazil. Braz Oral Res 2014;28:33-8.
Shin SM, Choi EJ, Moon SY. Prevalence of pathologies related to impacted mandibular third molars. Springerplus 2016;5:915.
Rafetto LK. Removal of asymptomatic third molars: A supporting view. J Oral Maxillofac Surg 2006;64:1811-5.
Siddiqui SR, Agrawal S, Monga HS, Abhishek G. Prophylactic removal of the third molars: Justified or not. J Int Oral Health 2015;7:132-5.
Mi HW, Lee M-C, Fu E, Chow L-P, Lin C-P. Highly efficient multipotent differentiation of human periodontal ligament fibroblasts induced by combined BMP4 and hTERT Gene transfer. Gene Therapy 2011;18:452-61.
Shah K, Kale AD, Hallikerimath S, Chandra S. Recurrent peripheral cemento-ossifying fibroma: Report of a recurrence case. Contemp Clin Dent 2012;3:S23-5.
Kumar SK, Ram S, Jorgensen MG, Shuler CF, Sedghizadeh PP. Multicentric peripheral ossifying fibroma. J Oal Sci 2006;48:239-43.
Eversole LR, Rovin S. Reactive lesions of gingival. J Oral Pathol 1972;1:30-8.
Ganji KK, Chakki AB, Nagaral SC, Verma E. Peripheral cemento-ossifying fibroma: Case series literature review. Case Rep Dent 2013;2013:930870.
Elarbi M, Azuzz L. Cemento-ossifying fibroma of the mandible case report. Rev Cir Traumatol Buco-Maxilo-Fac 2014;14:41-4.
Antony VV, Khan R. Peripheral Cemento- ossifying fibroma”–A case report. IOSR J Dent Med Sci 2013;6:34-7.
Nalabolu GRK, Mohiddin A, Hiremath SKS, Manyam R, Bharath TS, Raju PR. Epidemiological study of odontogenic tumours: An institutional experience. J Infect Public Health 2017;10:324-30.
Deepa J, Deepa RM, Kale A. An institutional retrospective study to assess the change in prevalence of odontogenic cysts and tumors based on WHO 2005 classification. Ann Dent Spec 2016;4:62-7.
Sriram G, Shetty RP. Odontogenic tumor: A study of 250 cases in an Indian teaching hospital. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2008;105:e14-21.
Dr. Karla M Carvalho
Liberty Apartments, Flat No: 202, Feira Alta, Mapusa - 403 507, Goa
Source of Support: None, Conflict of Interest: None