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British Journal of Oral and Maxillofacial Surgery 58 (2020) 1017–1022

New World Health Organization classification of


odontogenic tumours: impact on the prevalence of
odontogenic tumours and analysis of 1231 cases from Turkey
M. Soluk-Tekkesin a,∗ , S. Cakarer b , N. Aksakalli a , C. Alatli a , V. Olgac a
a Institute of Oncology, Department of Tumour Pathology, Istanbul University, Istanbul, Turkey
b Dentistry Faculty, Department of Oral and Maxillofacial Surgery, Istanbul University, Istanbul, Turkey

Accepted 19 June 2020


Available online 16 July 2020

Abstract

The aims of this study were to describe the frequency of odontogenic tumours (OT) based on the World Health Organization’s (WHO)
4th edition of Head and Neck Tumours in Turkey, to compare the results with other regions and to assess the frequency changes of OT
worldwide after the new WHO classification. OT were selected from the pathology department’s files between 1971-2018. In a total of 1231
OT, 1215 (98.7%) were benign, whereas malignant OT were only 16 cases (1.3%). The three most common tumours were ameloblastoma
(n = 366, 29.7%), odontoma (n = 335, 27.2% both complex and compound types), and odontogenic myxoma (n = 190, 15.4%), respectively.
After the 2017 classification, the decrease of OT frequency was found among 20%-42% in the selected epidemiological series because of
re-classification of some lesions. The pattern of incidence in the Turkish population is similar to that in other populations. However, there
are some differences in the frequency of the tumour types. It is obvious that the relative frequency of odontogenic tumours worldwide will
change based on the new classification. It should be kept in mind that this is not a real decrease of OT cases. These marked changes in the
frequency and prevalence of OT is just related to reclassification of some entities.
© 2020 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Odontogenic tumours; WHO; update; ameloblastoma; odontogenic keratocyst; classification

Introduction ter understanding of the interactions between odontogenic


epithelium and ectomesenchyme and update of the new
Odontogenic tumours (OT) are a heterogeneous group of genetic and molecular data. The origin based subclassifica-
lesions derived from epithelial and mesenchymal elements tion (epithelial, mixed, and mesenchymal) was first defined
of the tooth-forming apparatus that make these lesions in 1992, and this was continued in the 2005 classification and
unique to the jaws. They show diverse clinical behaviour and the last WHO classification which was published in January
histopathological types, which range from hamartomatous 2017.2,3 The new edition includes newly described or newly
lesions to malignancy. added lesions (sclerosing odontogenic carcinoma, primordial
The first World Health Organization (WHO) classifica- odontogenic tumour, odontogenic carcinosarcoma, cemento-
tion of odontogenic tumours was published in 1971.1 This ossifying fibroma), reclassified entities (ameloblastomas,
dynamic classification is constantly renewed with the bet- odontogenic carcinomas/sarcomas, ameloblastic fibromas)
and removal of tumours (keratocystic odontogenic tumour,
∗ Corresponding author at: Institute of Oncology, Istanbul University,
calcifying cystic odontogenic tumour, odontoameloblas-
34093 Capa- Istanbul, Turkey, Tel: +90 532 748 70 57.
toma). 2,3
E-mail address: msoluk@istanbul.edu.tr (M. Soluk-Tekkesin).

https://doi.org/10.1016/j.bjoms.2020.06.033
0266-4356/© 2020 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
1018
Table 1
Number, age, gender, and anatomical distribution of 1231 odontogenic tumours according to histopathological type.
WHO 2017 classification No. Percentage of all OT Mean age (years) M:F ratio Maxilla Mandible

Anterior Premolar Molar Anterior Premolar Molar

M. Soluk-Tekkesin et al. / British Journal of Oral and Maxillofacial Surgery 58 (2020) 1017–1022
Malignant odontogenic tumours (n = 16, 1.3%)
Ameloblastic carcinoma 3 0.24 50.6 2:1 0 0 0 1 0 2
Primary intraosseous carcinoma, NOS 5 0.41 46.8 1.5:1 2 0 0 0 0 3
Sclerosing odontogenic carcinoma 0 – – – – – – – – –
Clear cell odontogenic carcinoma 4 0.33 37.5 1:1 0 0 3 0 1 0
Ghost cell odontogenic carcinoma 0 – – – – – – – – –
Odontogenic carcinosarcoma 1 0.08 30 Woman 0 0 0 0 0 1
Odontogenic sarcomas 3 0.24 50 2:1 2 0 0 0 0 1
Benign odontogenic tumours (n = 1215, 98.7%)
Epithelial origin (n = 431, 35.01%)
Ameloblastoma, conventional 303 24.61 36.1 0.95:1 5 4 20 27 30 217
Ameloblastoma, unicystic type 52 4.2 34.6 0.5:1 1 0 5 8 3 35
Ameloblastoma,extraosseous/peripheral 11 0.9 39.7 1.7:1 1 0 1 1 2 6
Metastasising (malignant) ameloblastoma 0 – – – – – – – – –
Squamous odontogenic tumour 11 0.9 41.5 1.75:1 2 2 1 3 2 1
Calcifying epithelial odontogenic tumour 22 1.8 37.1 1:1 1 3 5 1 3 9
Adenomatoid odontogenic tumour 32 2.6 23.2 0.4:1 13 0 1 9 7 2
Mixed (epithelial-mesenchymal) origin (n = 394, 32.0%)
Ameloblastic fibroma 15 1.21 20.4 1.14:1 0 0 0 3 4 8
Primordial odontogenic tumour 0 – – – – – – – – –
Odontoma (all types) 335 27.21 26.85 1.17:1
Compound type 144 11.7 23.9 1.05:1 49 9 6 42 13 25
Complex type 191 15.51 29.0 1.27:1 31 1 26 23 8 102
Developing odontoma (ameloblastic fibrodentinoma/odontoma) 40 3.25 22.5 2.07:1 5 1 7 4 1 22
Dentinogenic ghost cell tumour 4 0.33 40.5 0.3:1 0 0 0 0 0 4
Mesenchymal origin (n = 360, 29.25%)
Odontogenic fibroma 9 0.73 32.5 1.25:1 1 1 1 0 5 1
Odontogenic myxoma/myxofibroma 190 15.44 34.9 0.6:1 34 20 19 28 33 56
Cementoblastoma 34 2.76 31.9 0.7:1 0 0 0 0 9 25
Cemento-ossifying fibroma 127 10.32 31.63 0.42:1 5 9 11 22 33 47
Peripheral odontogenic tumours (n = 30, 2.44%, excluding peripheral ameloblastoma)
Odontogenic fibroma 27 2.2 36.7 1.5:1 8 3 2 4 6 4
Calcifying epithelial odontogenic tumour 2 0.16 33.5 All men 1 0 0 0 0 1
Dentinogenic ghost cell tumour 1 0.08 15 Woman 1 0 0 0 0 0
Total 1231 100 32.1 0.9:1 162 53 108 176 160 572
M. Soluk-Tekkesin et al. / British Journal of Oral and Maxillofacial Surgery 58 (2020) 1017–1022 1019

Table 2
The frequency differences in the studies of OTs in regard to different classification.
First author, year, and Country No. of OT cases No. of OT cases without Decrease of OT OKC order among OTs in
reference (2005 WHO) OKC and COC (2017 WHO) (%) the original series
Luo 200824 China 1.309 754 (OKC:522, C0C:26) 42 Most common
Tawfik 201013 Egypt 82 66 (OKC:16, COC:0) 20 Second most common
Servato 20128 Brazil 240 155 (OKC:76, COC:9) 35 Most common
Siriwardena 201212 Sri Lanka 1.677 1222 (OKC:431, COC:24) 27 Second most common
da Silva 20155 Brazil 289 167 (OKC:100, COC:22) 42 Most common
Nalabolu 201611 India 161 108 (OKC:53, COC:0) 33 Second most common
Ismail 20186 Malaysia 173 123 (OKC:38, COC:12) 30 Second most common

OT: Odontogenic tumour, OKC: Odontogenic keratocyst, COC: Calcifying odontogenic cyst.

One of the major changes in the last classification is ically in accordance with the 2017 WHO classification of
the removal of “keratocystic odontogenic tumour” from the benign and malignant odontogenic tumours. Many epidemi-
neoplastic category in the 2005 classification back into the ological studies of odontogenic tumours have been published
cyst category with “odontogenic keratocyst (OKC)” name. according to the 2005 WHO classification, so the numbers
Actually, the redefinition of OKC as a tumour in 2005 had were recalculated (the keratocystic OT and calcifying OT
produced a relative increase in the prevalence and frequency data were removed) in those reported for comparative pur-
of OT4 and OKC occurred one of the most common OT in poses.
many studies.5–7 All data analysis and graphs were performed using
The aims of the present study were to assess the frequency Microsoft Office Excel 2011. This retrospective study was
of odontogenic tumours based on the new classification approved by the Research Ethics Committee of Istanbul Uni-
within the Turkish population, to compare the results with versity (Number: 795/2018) in compliance with the Helsinki
other regions and their geographic variation, and to show that Declaration.
the relative frequency of OT will decrease worldwide after
the new classification.

Results

Material and methods Among 53,869 oral biopsies in the records of the Depart-
ment of Tumour Pathology at Istanbul University between
A total of 1231 odontogenic tumours were selected from the January 1971- January 2018, 1231 cases (2.3%) were diag-
Oral and Maxillofacial Pathology Unit files between 1971- nosed as OT (Table 1). The three most common tumours
2018. These tumours were identified using the clinical details, were ameloblastoma (n = 366, 29.7% including unicystic
including age, sex, and anatomical location. To analyse the and peripheral ameloblastomas), odontoma (n = 335, 27.2%
site distribution, the jaws were divided into the anterior, both complex and compound types), and odontogenic myx-
premolar, and molar areas. The mandibular molar site also oma/fibromyxoma (n = 190, 15.4%), respectively. Malignant
included angle and ramus areas. The recurrent cases were (n = 16, 1.3%) and peripheral OT (n = 41, 3.3%) were a small
detected and calculated as one case. The cases with a lack of proportion of this series. The mean age was about 32 years
clinical information were excluded from the study. All cases (range 2 - 91) and there was a slight female predilection
were reviewed and, if necessary, reclassified histopatholog- (53.5%). The most common site was the mandible molar

Table 3
Comparison of the present study with previous studies of OT series from different regions that updated in numbers based on the 2017 WHO classification.
First author, year, and Country No.* Period Most common The most Mean age* M:F ratio*
reference (years) OTs* common sites* (years)
Luo 200824 China 754 21 Ameloblastoma, odontoma Mand. molar, mand. anterior 34.48** 1.13:1
Tawfik 201013 Egypt 66 16 Ameloblastoma, odontoma Mand. molar, mand. premolar 29.57** 1.06:1
Servato 20128 Brazil 155 31 Odontoma, ameloblastoma Mand. molar, mand. premolar 29.0** 0.9:1
Siriwardena 201212 Sri Lanka 1222 30 Ameloblastoma, odontoma Mand. molar, mand. premolar 30.8** 0.9:1
da Silva 20155 Brazil 167 10 Ameloblastoma, odontoma Mand., max. (no subvision) 35.0** 1:1.3**
Nalabolu 201611 India 108 13 Ameloblastoma, odontoma Mand. molar, mand. anteior NA 1.91:1
Ismail 20186 Malaysia 123 8 Ameloblastoma, odontoma Mand. molar,max. anterior 33.5** 1.4:1**
Present study 2018 Turkey 1231 48 Ameloblastoma, odontoma Mand. molar, mand. anterior 32.1 0.9:1

NA: not applicable, Mand: mandible, Max: maxilla.


∗ The results were re-calculated by excluding the odontogenic keratocyst and calcifying odontogenic cyst from the series.
∗∗ The numbers were not re-calculated due to limited knowledge, used original numbers.
1020 M. Soluk-Tekkesin et al. / British Journal of Oral and Maxillofacial Surgery 58 (2020) 1017–1022

region, followed by the mandible anterior and maxillary ante- Odontoma were evaluated as complex and compound
rior sites. types in the present study as reported in the series from
Table 2 shows the frequency changes of OT after 2017 Chile18 and Estonia.19 The patients afflicted with compound
classification in some selected regional series that the odontoma were younger than those with the complex type as
decrease of OT frequency was found among 20% - 42%. reported Tawfik et al,13 Fernandes et al,20 and Tamme et al.19
The patients in the second decade of life showed a higher fre-
quency of odontomas, which was in keeping with the previous
Discussion reports.11–20 Males were affected slightly more than females
in both types of odontomas, in contrast with the series from
The last classification of OT was published in January 2017. Chile18 and Estonia19 that showed that females were affected
One of the most notable changes in the last classification more commonly. Most complex odontomas were found in the
is that “keratocystic odontogenic tumour” and “calcify- posterior mandible whereas compound odontomas were most
ing cystic odontogenic tumour” of 2005 are now classified commonly observed in the anterior maxilla.
under the developmental odontogenic cysts with the name of Odontogenic myxoma/myxofibroma was found as the
“odontogenic keratocyst” and “calcifying odontogenic cyst”, third commonest tumour (15.4%) in our series. The preva-
respectively.2 Gaitán-Cepeda et al4 and Servato et al8 demon- lence was higher than in previous reports. 11,13,21,22 On
strated nicely that there was a huge increase in the frequency the other hand, an African series reported that the odonto-
of the OT in their series like 92% and 50%, respectively, after genic myxoma was found as the second common tumour
the reclassification of OKC as a tumour in 2005. with a prevalence of 12%.23 In our series, females were
Another study from Brazil also showed that after the affected more than males (1.7:1) similar to the reports of
2005 classification, OKC occupied first place among the OT Siriwardena et al,10 Tawfik et al,13 Chrysomali et al,22 and
and their study demonstrated a huge increase of 464.2% Luo and Li.24 The lesions were commonly observed at the
in OT incidence.9 The present study did not include these mandibular molar area, consistent with the results of reported
two cysts to reflect real incidence and they were also series.10,13,22,25
excluded from the comparing studies due to a real compari- The mixed OT classification of 2005 had included
son (Table 3). Recently a large, detailed epidemiological data ameloblastic fibroma, ameloblastic fibrodentinoma, and
of OT from UK was published in the line with 2017 WHO ameloblastic fibro-odontoma entities. The last two were
classification.10 excluded from the 2017 classification because there is some
The mandible was more commonly involved than the max- evidence that once dental hard tissues are formed, these
illa by all tumours with a ratio of 2.74:1. The most common lesions are programmed to develop into odontomas.2,3 How-
sites were mandibular molar, anterior mandible, and anterior ever, in our experience, there are some destructive, large cases
maxillary areas, respectively. These results show similarity of these entities that are difficult to diagnosis them as a “devel-
with those reported by Nalabolou et al11 and Servato et al8 oping odontoma”. This debate may be clarified by the next
regarding the first and third common site. On the other hand, classification.
Siriwardena et al12 reported that the second most common Two of the 40 cases of “developing odontoma” were
site was the anterior maxilla, different from our results. ameloblastic fibrodentinoma, others were ameloblastic fibro-
In the present study, the three most common tumours odontomas in our study. However, we agree that some
were ameloblastoma, odontoma and odontogenic myx- “developing odontoma” cases histopathologically resemble
oma/myxofibroma, respectively. Ameloblastoma was found ameloblastic fibro-odontoma, especially associated with an
as the most common tumour (29.7%) in our series agree- unerupted tooth, and these should be distinguished from true
ing with the results of Siriwardena et al,10 Nalabolu et ameloblastic fibro-odontoma. Therefore, the detailed evalu-
al,11 and Tawfik et al.13 Almost no gender predilection was ation of history, clinical, radiological and histopathological
observed in our series in contrast with the previous reports of features are essential to make a proper diagnosis.
South India,11 Egypt,13 and Nigeria14 that showed male pre- Cemento-ossifying fibroma was firstly included in the
dominance. The most frequently reported affected area was odontogenic tumour classification in 1971 under the ‘cemen-
mandibular posterior, and the conventional type was most tomas’ group with the name of “cementifying fibroma”, then
common type observed in the study. excluded from the 1992 and 2005 OT classifications and men-
Odontoma was found as the second most common tumour tioned under the bone-related lesions.1–3 In 2017, it was again
(27.2%) in our series different from the series of USA,15 classified under the odontogenic tumours-mesenchymal ori-
Canada16 , Mexio17 , Chile,18 and Estonia,19 which reported gin and distinguished from the juvenile ossifying fibromas.
that the odontoma was found as the most common tumour. However, it is still a fibro-osseous lesion and discussed in
The important reasons for variability of most common detail with the other ossifying fibromas in the fibro-osseous
tumours in different populations can be associated with the lesions section of the last WHO edition.2,3 The prevalence of
differences in the submission of lesion samples or limited cemento-ossifying fibromas in our series was found as 10.3%.
access to histopathology services,10 therefore the real fre- It is not possible to make a proper comparison between the
quency may tend to be underestimated. reported OT series and the present study due to classifica-
M. Soluk-Tekkesin et al. / British Journal of Oral and Maxillofacial Surgery 58 (2020) 1017–1022 1021

tion differences used, but our results were compatible with in OT cases after the 2005 classification did not reflect real
the other series of fibro-osseous lesions of the jaws in terms increment of OT, this is not a real decrease of OT cases. These
of female predominance in middle-age and location in the marked changes in the frequency and prevalence of OT is just
posterior mandible region.26 related to reclassification of some entities.
When comparing odontogenic malignancies, the percent-
age of the malignant OT in our series was found as 1.3%,
differently than the reports from UK,10 Chinese,21,24 and Conflict of interest
African27,28 populations which showed more than 5% of
prevalence. The studies from North and South America have We have no conflicts of interest.
reported the percentage of OT malignancy as 1.6%.16–18,20
Primary intraosseous carcinoma was the most common
malignant tumour in our series (0.4%), in keeping with the Ethics statement/confirmation of patients’ permission
reports from China24 , Egypt,13 and Sri Lanka.12 The lesion
was found commonly at the posterior of the mandible, as This retrospective study was approved by the Research Ethics
in previous reports.12,13,24 On the other hand, the report Committee of Istanbul University (Number: 795/2018) in
from Brazil8 demonstrated that ameloblastic carcinoma was compliance with the Helsinki Declaration. Patients’ permis-
the most common malignant tumour with a prevalence of sion was not necessary.
1.3%. In our series, clear cell odontogenic carcinoma was
found as the second common malignant tumour (0.32%) and
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