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Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population
International Journal of Pediatric Otorhinolaryngology 90 (2016) 241e244
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: http://www.ijporlonline.com/
Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population Aline Priscila Ataíde, Felipe Paiva Fonseca, Alan Roger Santos Silva, Jacks Jorge Júnior, rcio Ajudarte Lopes, Pablo Agustin Vargas* Ma Department of Oral Diagnosis, Oral Pathology and Semiology Divisions, Piracicaba Dental School, University of Campinas, Piracicaba, Brazil
a r t i c l e i n f o
a b s t r a c t
Article history: Received 31 July 2016 Received in revised form 21 September 2016 Accepted 22 September 2016 Available online 24 September 2016
Objectives: Oral lesions affecting infants account for approximately 10% of all samples from diagnostic services and studies investigating the distribution of these lesions in pediatrics from different geographic areas are desired to improve the diagnostic knowledge of clinicians. Therefore, the aim of this study is to describe the distribution of oral lesions in a southeastern Brazilian population. Methods: The oral pathology files of the University of Campinas was retrospectively reviewed for all cases diagnosed from 2000 to 2014 affecting patients 16-years-old and younger. Data on gender and diagnosis were retrieved from patients' oral pathology reports and included in a Microsoft Excel® database. Results: Out of 34,138 cases, 2539 affected pediatric patients (7.4%) with a higher incidence in those with 13e16 years-old. Salivary gland disease was the most common group of lesions (37.1%), followed by mucosal pathology (13.6%) and odontogenic cysts (11.3%). Mucous extravasation cyst was the most common lesion (36.3%), followed by fibrous hyperplasia (5.6%) and dental follicle (5.2%). Dental lesions were uncommon (7.9%) and malignancies rare (0.4%). Conclusions: Our results were similar to previous studies and the small differences observed were more likely result of methodological variability and characteristics of the service of origin from where samples were collected. © 2016 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pediatrics Oral lesions Infants Salivary glands Oral cavity
1. Introduction Oral lesions affecting pediatric patients represent approximately 10% of the total amount of lesions diagnosed in specialized diagnostic centers [1], and because parents frequently look for medical assistance even before oral medicine specialists, not only dentists but also otolaryngologists, pediatricians, head and neck surgeons, dermatologists and other medical specialists must be aware of the epidemiological distribution of these lesions. However, the number of studies investigating oral lesions in this population is small and most of them aimed to determine the
* Corresponding author. Department of Oral Diagnosis (Pathology), Piracicaba Dental School, University of Campinas, Avenida Limeira 901, Piracicaba, S~ ao Paulo, Caixa Postal 52, CEP: 13414-903, Brazil. E-mail addresses: [email protected] (A.P. Ataíde), felipepfonseca@ hotmail.com (F.P. Fonseca), [email protected] (A.R. Santos Silva), jacks@ fop.unicamp.br (J. Jorge Júnior), [email protected] (M.A. Lopes), pavargas@ fop.unicamp.br (P.A. Vargas). http://dx.doi.org/10.1016/j.ijporl.2016.09.027 0165-5876/© 2016 Elsevier Ireland Ltd. All rights reserved.
distribution of specific group of lesions like odontogenic tumors, salivary gland diseases, jaw cysts and others [2e4]. Moreover, the lack of uniformity in the design of the studies that investigate patients with different age ranges, through widely different period of time and distribute lesions in distinct classification categories, impairs an appropriate understanding of the incidence of lesions in different geographic locations, since results cannot be adequately compared [5,6]. In Brazil very few series describing the incidence of oral lesions in pediatric patients were published to date [7e12] and because of its large geographic extension and socio-demographic heterogeneity, differences in the epidemiology of these lesions throughout the national territory could be found, demanding more studies to be carried out. Therefore, in this survey we investigated the distribution of oral lesions affecting pediatric patients retrieved from a single reference center on oral pathology located in the southeastern region of Brazil with the purpose of determining if there is any difference in the incidence of these lesions when compared with other regions of the globe.
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2. Material and methods A retrospective analysis of the oral pathology files of the Piracicaba Dental School e University of Campinas (Piracicaba/Brazil) over a 15-year period from January 2000 to December 2014 was conducted and all cases diagnosed in patients 16 years-old or younger were retrieved and tabulated in a Microsoft Excel® file for epidemiologic description. In addition to age, data on gender and final diagnosis were retrieved from patients' oral histopathology reports. The frequency of each disease, male to female ratio, mean age (and standard deviation), age range and the percentage of each lesion were calculated and provided. In an attempt to better compare our results with those previously described in literature, all lesions were grouped in one of 12 categories according to criteria proposed by Jones et al. [13] and posteriorly used by Ha et al. [14] because they represent comprehensive categorization of oral and maxillofacial lesions in pediatrics.
3. Results In the 15-year period investigated, 34,138 lesions were diagnosed, from which 2539 affected patients 16 years-old or younger, representing 7.4% of all specimens received in the Department. A slight female predominance was found (1416 vs 1123 cases, respectively) with a male:female ratio of 0.79. It was observed that patients 13e16 years old were the most affected (39.6%) patients (Fig. 1). As summarized in Table 1, salivary gland diseases was the most frequent subgroup of lesions, followed by mucosal pathology and odontogenic cysts. Mucous extravasation cyst represented by far the most common entity in both the salivary gland diseases (97.8%) group and in the whole specimens retrieved (36.3%) (Table 2 and S1). Fibrous hyperplasia was the most common lesion in the mucosal pathology group (41.6% of the group and 5.6% of all specimens) (Table S2), dentigerous cyst was the most frequent diagnosis in the odontogenic cysts group (44.6% of the group and 5.0% of all specimens) (Table S3), gingival hyperplasia predominated in the gingival and periodontal pathologies group (41.4% of the group and 4.5% of all specimens) (Table S4), dental follicle was the most common entity in the dental pathology group (64.5% of the group and 5.2% of all specimens) (Table S5) and cases classified as nondiagnostic because the appropriate clinic and microscopic correlation could
Table 1 Oral pathology distribution according to their classifications in pediatric patients in Brazil (2000e2014), Australia (1945e2003) and UK (1973e2002).
Salivary gland disease Mucosal pathology Odontogenic cysts Gingival and periodontal pathology Dental pathology Miscellaneous pathology Odontogenic tumors Bone pathology Connective tissue pathology Non-odontogenic cysts Malignant tumors Total
Present study (2016)
Ha et al. [14]
Jones et al. [13]
Total
%
Total
%
Total
%
943 344 287 273
37.1 13.6 11.3 10.8
80 222 219 137
6.3 17.3 17.1 10.7
840 533 519 439
19.6 12.5 12.2 10.3
203 166 137 73 56 48 9 2539
7.9 6.5 5.4 2.9 2.2 1.9 0.4 100.0
318 84 128 47 23 13 9 1280
24.8 6.6 10.0 3.7 1.8 1.0 0.7 100.0
973 335 243 143 146 67 31 4269
22.7 7.9 5.7 3.4 3.4 1.6 0.7 100.0
The italic values are the percentage values as indicated by the symbol % in italic.
Table 2 Most frequent oral lesions affecting pediatric patients in this study. Diagnoses
Total
%
Mucous extravasation cyst Fibrous hyperplasia Dental follicle Dentigerous cyst Nondiagnostic Gingival hyperplasia Squamous papiloma Radicular cyst Odontoma Pyogenic granuloma Peripheral ossifying fibroma Hereditary gingival fibromatosis Central giant cell lesion Keratocyst Odontogenic cyst (not specified) Focal epithelial hyperplasia
922 143 131 128 115 113 90 85 80 44 38 38 36 34 32 32
36.3 5.6 5.2 5.0 4.5 4.4 3.5 3.4 3.2 1.7 1.5 1.5 1.4 1.3 1.2 1.2
The italic values are the percentage values as indicated by the symbol % in italic.
not be done represented the most frequent diagnosis in the miscellaneous group (69.3% of the group and 4.5% of all specimens) (Table S6). Odontoma was the most common odontogenic tumor/hamartoma (58.4% of the group and 3.2% of all specimens) (Table S7), central giant cell lesion was the most common condition in bone pathology (49.3% of the group and 1.4% of all specimens) (Table S8), lymphangioma predominated in the connective tissue pathology group (25% of the group and 0.6% of all specimens) (Table S9), simple bone cyst was the most frequent in the non-odontogenic cyst group (56.3% of the group and 1.1% of all specimens) (Table S10) and Langerhans cell histiocytosis and lymphomas represented the most common malignancies (33.3% of the group each and 0.1% of all specimens each) (Table S11). 4. Discussion
Fig. 1. Distribution of oral lesions in the pediatric population studied according to the age of the patients.
Oral lesions affecting pediatric patients are relatively uncommon in specialized diagnostic centers ranging from 6.6% [8] to over 20.6% [6] of all histopathological specimens, also demonstrating a wide variability of entities recognized. Because of the large discrepancies in the methodologies used in the literature that include different periods of time, age ranges and diseases categorization, what avoid the appropriate analysis of data, we opted for following the guidelines used by Jones et al. [13] and Ha et al. [14] who
243
MEC: mucous extravasation cyst; UOC: Unspecific odontogenic cyst; UIP: Unspecific inflammatory process; CGCG: Central giant cell granuloma; IFH: Inflammatory fibrous hyperplasia. a Inflammatory fibrous hyperplasia, peripheral giant cell granuloma, peripheral ossifying granuloma and fibroma had the same frequency. b Radicular cyst, periapical granuloma, melanic pigmentation, central giant cell lesion and hemangioma had the same frequency.
MEC Dentigerous cyst Fibrous hyperplasia Gingival hyperplasia Radicular cyst Odontoma UIP Pulpitis UOC Pulp necrosis MEC Dental follicle Dentigerous cyst UIP Radicular cyst Periapical granuloma Odontoma Pyogenic granuloma Nondiagnostic IFHa MEC UIP Fibrous hyperplasia Epithelial hyperplasia Gingival hyperplasia Papiloma Pyogenic granuloma Periapical granuloma Dentigerous cyst Radicular cystb MEC Dental follicle Pyogenic granuloma Dentigerous cyst Odontoma Keratocyst Radicular cyst CGCG Papilloma Fibrous dysplasia MEC Dental follicle Fibroma Odontogenic cyst CGCG Radicular cyst Dentigerous cyst Odontoma Chronic inflammation Pyogenic granuloma MEC Fibrous hyperplasia Dental follicle Dentigerous cyst Nondiagnostic Gingival hyperplasia Squamous papiloma Radicular cyst Odontoma Pyogenic granuloma
MEC Fibroma Pyogenic granuloma Dentigerous cyst Ameloblastoma Odontoma Radicular cyst Papilloma Traumatic ulcer Peripheral ossifying fibroma
Lima et al. [8] Southern Brazil 625 cases 0e14 yrs Vale et al. [9] Northeastern Brazil 315 cases 0e18 yrs Martins-Filho et al. [10] Northeastern Brazil 564 cases 0e18 yrs ^ a et al. [11] Pesso Northeastern Brazil 360 cases 0e19 yrs Cavalcante et al. [12] Northeastern Brazil 1240 cases 0e16 yrs Current study et al. (2016) Southeastern Brazil 2539 cases 0e16 yrs
Table 3 The 10 most frequent oral lesions observed in pediatric patients described in Brazilian studies.
described the distribution of pediatric oral lesions in United Kingdom and Australia, respectively, to better compare our results with previous reports. Although dental pathology represented the most common group of lesions in both studies, whereas salivary gland pathology was the most common in ours possibly because dental hard tissue abnormalities are usually not frequently submitted to microscopic exam in our laboratory, mucosal pathology, odontogenic cysts and gingival/periodontal pathology were the second, third and fourth most frequent categories and mucous extravasation cyst represented the most common entity in all three studies, suggesting that differences in the frequency of the main oral pediatric lesions is small and might be influenced by characteristics of each diagnostic service. Moreover, the highest incidence of mucous extravasation cyst in lower lip and fibrous/gingival hyperplasia is direct related to mechanical trauma what is relatively common in pediatric patients. As previously described, we also observed that older pediatric patients present a higher frequency of lesions, what may be consequence not only of the epidemiological features of each disease, but also because clinicians tends to postpone biopsies or surgeries considering that most of the pediatric lesions are reactive or benign in nature as exemplified in our study, where only nine cases were diagnosed as malignant (0.4%) [11,12,14]. Oral surgeries in pediatrics are usually small and may be under-representative for microscopic analyses, as a consequence, nondiagnostic cases accounted for 4.5% of our cases and comprised 2.4% of Jones et al. [13], 2.7% of Ha et al. [14] and 10.1% of Kezler et al. [5]. Although dental pathology did not account for the most common subgroup in our study as above mentioned, we believe that this finding more likely represents a peculiar feature of our service that very uncommonly receives dental hard tissue for analysis. Therefore, we agree with Ha et al. [14] who stated that dental lesions are usually under-represented in microscopic-based surveys. Similarly, gingivitis and odontomas can also have their frequency altered because these lesions are mostly diagnosed in a clinical and radiographic basis, respectively. Moreover, the origin of the data retrieved may also influence the results obtained, since hospitalbased studies are more likely to provide higher frequencies of benign and malignant tumors than dental school-based studies [15]. However, because population-based research on oral lesions would be very difficult to be done, we believe that microscopicbased studies well represent the distribution of these diseases, especially if the abovementioned limitations are considered in the interpretation of the results. In our sample we found a homogenous distribution of lesions between both genders, with a slight preponderance for females that accounted for 55.8% of the cases leading to a M:F ratio of 0.8:1.0. This fare distribution was described before [7,16] and was ^a et al. [11] to be consequence of the parents speculated by Pesso awareness with their children, since a higher frequency of females is found in adult populations [17], possibly representing the higher concern demonstrated by females than males. Despite small differences, the group of most frequent lesions observed in our study is in accordance with that observed in other international samples, with mucous extravasation cyst, dental follicles/dentigerous cyst and reactive periapical/gingival lesions ranking first in the lists. Because Brazil is an extensive and heterogeneous country both geographically and socioeconomically, we attempted to identify differences in the frequency of lesions by comparing our results with those previously published. There seems to be no study performed in Brazilian Central and Northern regions, but some authors have already described the distribution of oral lesions in pediatric patients from Northeast, South and Southeast regions of Brazil (Table 3). In Brazilian studies pediatric patients accounted from 6.6% [8] to 13.1% [9] of the samples, with a
Sousa et al. [7] Southeastern Brazil 2356 cases 0e14 yrs
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A.P. Ataíde et al. / International Journal of Pediatric Otorhinolaryngology 90 (2016) 241e244
slight female predominance leading to a male/female ratio from 0.7 to 1.0 [9,10,12], both results are similar to ours. Up to 76 lesions could be described in a single analysis [7], whereas we identified 108 different entities, possibly because of the strict clinicopathological correlation usually done in our service. Methodologically, studies were very heterogeneous, using age range from 0 to 14, 0e16, 0e18 and 0-19 years-old, investigating lesions from 5 to over 20 years period and aggregating them in very different categories. On the other hand, only minor differences were found in the distribution of the most frequent lesions. As in this survey, mucous extravasation cyst predominated in all studies and dental follicle/ ^a et al. dentigerous cyst were also very common. However, Pesso [11] reported a high frequency of ameloblastomas and MartinsFilho et al. [10] described a large number of keratocysts; whereas odontoma was the most common odontogenic tumor found in most of the studies. Malignant neoplasms were always very rare [8]. Understanding the frequency and distribution pattern of oral lesions among pediatric patients is important not only for oral pathologists and dental pediatricians, but also to different medical specialists who first deal with these patients. In our daily practice, although no official statistics are available in our service, we frequently receive patients referred by otolaryngologists, pediatricians and dermatologists due to the presence of oral lesions. Therefore, we believe that the current results will improve the diagnostic algorithm of all these health professionals. Finally, this study demonstrated that distribution of oral lesions is very similar in different regions of the globe, with minor differences that may be consequence of methodological differences in the studies performed and in the characteristics of the services from where lesions were retrieved. Funding Dra. Aline Priscila Ataide was supported with a scholarship of the National Council for Scientific and Technology Development (CNPq) e Brazil. Ethics committee approval This study was approved by the Institutional Ethics Committee. Conflict of interest statement The authors state that they have no potential conflict of interest that could bias the results obtained in the current study.
Appendix A. Supplementary data Supplementary data related to this article can be found at http:// dx.doi.org/10.1016/j.ijporl.2016.09.027. References [1] D.J. Brierley, C.K.M. Chee, P.M. Speight, A review of paediatric oral and maxillofacial pathology, Int. J. Paediatr. Dent. 23 (2013) 319e329. [2] Q.G. Fang, S. Shi, Z.N. Li, X. Zhang, F.Y. Liu, C.F. Sun, Epithelial salivary gland tumors in children: a twenty-five-year experience of 122 patients, Int. J. Pediatr. Otorhinolaryngol. 77 (2013) 1252e1254. [3] N. Li, X. Gao, Z. Xu, Z. Chen, L. Zhu, J. Wang, et al., Prevalence of developmental odontogenic cysts in children and adolescents with emphasis on dentigerous cyst and odontogenic keratocyst (keratocystic odontogenic tumor), Acta. Odontol. Scand. 72 (2014) 795e800. [4] K.S. Perry, A.T. Tkaczuk, J.F. Caccamese, R.A. Ord, K.D. Pereira, Tumors of the pediatric maxillofacial skeleton: a 20-year clinical study, JAMA Otolaryngol. Head. Neck. Surg. 141 (2015) 40e44. [5] A. Keszler, M.B. Guglielmotti, F.V. Dominguez, Oral pathology in children. Frequency, distribution and clinical significance, Acta. Odontol. Latinoam. 5 (1990) 39e48. ~ iga, C.R. Me ndez, R.R. Kauterich, D.C. Paniagua, Paediatric oral pa[6] M.D. Zun thology in a Chilean population: a 15-year review, Int. J. Paediatr. Dent. 23 (2013) 346e351. [7] F.B. Sousa, A. Etges, L. Correa, R.A. Mesquita, N.S. Araujo, Pediatric oral lesions: a 15-year review from Sao Paulo, Brazil, J. Clin. Paediatr. Dent. 26 (2002) 413e418. [8] S.G. Lima, S.T. Fontes, L.M. Araujo, A. Etges, S.B. Tarquinio, A.P. Gomes, A survey of oral and maxillofacial biopsies in children: a single-center retrospective study of 20 years in Pelotas Brazil, J. Appl. Oral. Sci. 16 (2008) 397e402. [9] E.B. Vale, F.M.M. Ramos-Perez, G.L.C. Rodrigues, E.J.A. Carvalho, J.F.L. Castro, D.E.C. Perez, A review of oral biopsies in children and adolescents: a clinicopathological study of a case series, J. Clin. Exp. Dent. 5 (2013) e144ee149. [10] P.R.S. Martins-Filho, T.S. Santos, M.R. Piva, H.F. Silva, L.C.F. Silva, A.C. Mascarenhas-Oliveira, et al., A multicenter retrospective cohort study on pediatric oral lesions, J. Dent. Child. 82 (2015) 84e90. ^ a, T.D.B. Alves, N.C.N. Santos, H.L.R. Santos, A.C.S. Azevedo, [11] C.P. Pesso J.N. Santos, et al., Epidemiological survey of oral lesions in children and adolescents in a Brazilian population, Int. J. Pediatr. Otorhinolaryngol. 79 (2015) 1865e1871. [12] R.B. Cavalcante, E. Turatti, A.P.B. Daniel, G.F. Alencar, Z. Chen, Retrospective review of oral and maxillofacial pathology in a Brazilian paediatric population, Eur. Arch. Paediatr. Dent. 17 (2016) 115e122. [13] A.V. Jones, C.D. Franklin, An analysis of oral and maxillofacial pathology found in children over a 30-year period, Int. J. Paediatr. Dent. 16 (2016) 19e30. [14] W.N. Ha, E. Kelloway, F. Dost, C.S. Farah, A retrospective analysis of oral and maxillofacial pathology in an Australian paediatric population, Aust. Dent. J. 59 (2014) 221e225. [15] E.R. Fregnani, R.Z. Fillipi, C.R. Oliveira, P.A. Vargas, O.P. Almeida, Odontomas and ameloblastomas: variable prevalences around the world? Oral. Oncol. 38 (2002) 807e808. [16] K. Dhanuthai, M. Banrai, S. Limpanaputtajak, A retrospective study of paediatric oral lesions from Thailand, Int. J. Paediatr. Dent. 17 (2007) 248e253. [17] E. Kelloway, W.N. Ha, F. Dost, C.S. Farah, A retrospective analysis of oral and maxillofacial pathology in an Australian adult population, Aust. Dent. J. 59 (2014) 215e220.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: http://www.ijporlonline.com/
Distribution of oral and maxillofacial lesions in pediatric patients from a Brazilian southeastern population Aline Priscila Ataíde, Felipe Paiva Fonseca, Alan Roger Santos Silva, Jacks Jorge Júnior, rcio Ajudarte Lopes, Pablo Agustin Vargas* Ma Department of Oral Diagnosis, Oral Pathology and Semiology Divisions, Piracicaba Dental School, University of Campinas, Piracicaba, Brazil
a r t i c l e i n f o
a b s t r a c t
Article history: Received 31 July 2016 Received in revised form 21 September 2016 Accepted 22 September 2016 Available online 24 September 2016
Objectives: Oral lesions affecting infants account for approximately 10% of all samples from diagnostic services and studies investigating the distribution of these lesions in pediatrics from different geographic areas are desired to improve the diagnostic knowledge of clinicians. Therefore, the aim of this study is to describe the distribution of oral lesions in a southeastern Brazilian population. Methods: The oral pathology files of the University of Campinas was retrospectively reviewed for all cases diagnosed from 2000 to 2014 affecting patients 16-years-old and younger. Data on gender and diagnosis were retrieved from patients' oral pathology reports and included in a Microsoft Excel® database. Results: Out of 34,138 cases, 2539 affected pediatric patients (7.4%) with a higher incidence in those with 13e16 years-old. Salivary gland disease was the most common group of lesions (37.1%), followed by mucosal pathology (13.6%) and odontogenic cysts (11.3%). Mucous extravasation cyst was the most common lesion (36.3%), followed by fibrous hyperplasia (5.6%) and dental follicle (5.2%). Dental lesions were uncommon (7.9%) and malignancies rare (0.4%). Conclusions: Our results were similar to previous studies and the small differences observed were more likely result of methodological variability and characteristics of the service of origin from where samples were collected. © 2016 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pediatrics Oral lesions Infants Salivary glands Oral cavity
1. Introduction Oral lesions affecting pediatric patients represent approximately 10% of the total amount of lesions diagnosed in specialized diagnostic centers [1], and because parents frequently look for medical assistance even before oral medicine specialists, not only dentists but also otolaryngologists, pediatricians, head and neck surgeons, dermatologists and other medical specialists must be aware of the epidemiological distribution of these lesions. However, the number of studies investigating oral lesions in this population is small and most of them aimed to determine the
* Corresponding author. Department of Oral Diagnosis (Pathology), Piracicaba Dental School, University of Campinas, Avenida Limeira 901, Piracicaba, S~ ao Paulo, Caixa Postal 52, CEP: 13414-903, Brazil. E-mail addresses: [email protected] (A.P. Ataíde), felipepfonseca@ hotmail.com (F.P. Fonseca), [email protected] (A.R. Santos Silva), jacks@ fop.unicamp.br (J. Jorge Júnior), [email protected] (M.A. Lopes), pavargas@ fop.unicamp.br (P.A. Vargas). http://dx.doi.org/10.1016/j.ijporl.2016.09.027 0165-5876/© 2016 Elsevier Ireland Ltd. All rights reserved.
distribution of specific group of lesions like odontogenic tumors, salivary gland diseases, jaw cysts and others [2e4]. Moreover, the lack of uniformity in the design of the studies that investigate patients with different age ranges, through widely different period of time and distribute lesions in distinct classification categories, impairs an appropriate understanding of the incidence of lesions in different geographic locations, since results cannot be adequately compared [5,6]. In Brazil very few series describing the incidence of oral lesions in pediatric patients were published to date [7e12] and because of its large geographic extension and socio-demographic heterogeneity, differences in the epidemiology of these lesions throughout the national territory could be found, demanding more studies to be carried out. Therefore, in this survey we investigated the distribution of oral lesions affecting pediatric patients retrieved from a single reference center on oral pathology located in the southeastern region of Brazil with the purpose of determining if there is any difference in the incidence of these lesions when compared with other regions of the globe.
242
A.P. Ataíde et al. / International Journal of Pediatric Otorhinolaryngology 90 (2016) 241e244
2. Material and methods A retrospective analysis of the oral pathology files of the Piracicaba Dental School e University of Campinas (Piracicaba/Brazil) over a 15-year period from January 2000 to December 2014 was conducted and all cases diagnosed in patients 16 years-old or younger were retrieved and tabulated in a Microsoft Excel® file for epidemiologic description. In addition to age, data on gender and final diagnosis were retrieved from patients' oral histopathology reports. The frequency of each disease, male to female ratio, mean age (and standard deviation), age range and the percentage of each lesion were calculated and provided. In an attempt to better compare our results with those previously described in literature, all lesions were grouped in one of 12 categories according to criteria proposed by Jones et al. [13] and posteriorly used by Ha et al. [14] because they represent comprehensive categorization of oral and maxillofacial lesions in pediatrics.
3. Results In the 15-year period investigated, 34,138 lesions were diagnosed, from which 2539 affected patients 16 years-old or younger, representing 7.4% of all specimens received in the Department. A slight female predominance was found (1416 vs 1123 cases, respectively) with a male:female ratio of 0.79. It was observed that patients 13e16 years old were the most affected (39.6%) patients (Fig. 1). As summarized in Table 1, salivary gland diseases was the most frequent subgroup of lesions, followed by mucosal pathology and odontogenic cysts. Mucous extravasation cyst represented by far the most common entity in both the salivary gland diseases (97.8%) group and in the whole specimens retrieved (36.3%) (Table 2 and S1). Fibrous hyperplasia was the most common lesion in the mucosal pathology group (41.6% of the group and 5.6% of all specimens) (Table S2), dentigerous cyst was the most frequent diagnosis in the odontogenic cysts group (44.6% of the group and 5.0% of all specimens) (Table S3), gingival hyperplasia predominated in the gingival and periodontal pathologies group (41.4% of the group and 4.5% of all specimens) (Table S4), dental follicle was the most common entity in the dental pathology group (64.5% of the group and 5.2% of all specimens) (Table S5) and cases classified as nondiagnostic because the appropriate clinic and microscopic correlation could
Table 1 Oral pathology distribution according to their classifications in pediatric patients in Brazil (2000e2014), Australia (1945e2003) and UK (1973e2002).
Salivary gland disease Mucosal pathology Odontogenic cysts Gingival and periodontal pathology Dental pathology Miscellaneous pathology Odontogenic tumors Bone pathology Connective tissue pathology Non-odontogenic cysts Malignant tumors Total
Present study (2016)
Ha et al. [14]
Jones et al. [13]
Total
%
Total
%
Total
%
943 344 287 273
37.1 13.6 11.3 10.8
80 222 219 137
6.3 17.3 17.1 10.7
840 533 519 439
19.6 12.5 12.2 10.3
203 166 137 73 56 48 9 2539
7.9 6.5 5.4 2.9 2.2 1.9 0.4 100.0
318 84 128 47 23 13 9 1280
24.8 6.6 10.0 3.7 1.8 1.0 0.7 100.0
973 335 243 143 146 67 31 4269
22.7 7.9 5.7 3.4 3.4 1.6 0.7 100.0
The italic values are the percentage values as indicated by the symbol % in italic.
Table 2 Most frequent oral lesions affecting pediatric patients in this study. Diagnoses
Total
%
Mucous extravasation cyst Fibrous hyperplasia Dental follicle Dentigerous cyst Nondiagnostic Gingival hyperplasia Squamous papiloma Radicular cyst Odontoma Pyogenic granuloma Peripheral ossifying fibroma Hereditary gingival fibromatosis Central giant cell lesion Keratocyst Odontogenic cyst (not specified) Focal epithelial hyperplasia
922 143 131 128 115 113 90 85 80 44 38 38 36 34 32 32
36.3 5.6 5.2 5.0 4.5 4.4 3.5 3.4 3.2 1.7 1.5 1.5 1.4 1.3 1.2 1.2
The italic values are the percentage values as indicated by the symbol % in italic.
not be done represented the most frequent diagnosis in the miscellaneous group (69.3% of the group and 4.5% of all specimens) (Table S6). Odontoma was the most common odontogenic tumor/hamartoma (58.4% of the group and 3.2% of all specimens) (Table S7), central giant cell lesion was the most common condition in bone pathology (49.3% of the group and 1.4% of all specimens) (Table S8), lymphangioma predominated in the connective tissue pathology group (25% of the group and 0.6% of all specimens) (Table S9), simple bone cyst was the most frequent in the non-odontogenic cyst group (56.3% of the group and 1.1% of all specimens) (Table S10) and Langerhans cell histiocytosis and lymphomas represented the most common malignancies (33.3% of the group each and 0.1% of all specimens each) (Table S11). 4. Discussion
Fig. 1. Distribution of oral lesions in the pediatric population studied according to the age of the patients.
Oral lesions affecting pediatric patients are relatively uncommon in specialized diagnostic centers ranging from 6.6% [8] to over 20.6% [6] of all histopathological specimens, also demonstrating a wide variability of entities recognized. Because of the large discrepancies in the methodologies used in the literature that include different periods of time, age ranges and diseases categorization, what avoid the appropriate analysis of data, we opted for following the guidelines used by Jones et al. [13] and Ha et al. [14] who
243
MEC: mucous extravasation cyst; UOC: Unspecific odontogenic cyst; UIP: Unspecific inflammatory process; CGCG: Central giant cell granuloma; IFH: Inflammatory fibrous hyperplasia. a Inflammatory fibrous hyperplasia, peripheral giant cell granuloma, peripheral ossifying granuloma and fibroma had the same frequency. b Radicular cyst, periapical granuloma, melanic pigmentation, central giant cell lesion and hemangioma had the same frequency.
MEC Dentigerous cyst Fibrous hyperplasia Gingival hyperplasia Radicular cyst Odontoma UIP Pulpitis UOC Pulp necrosis MEC Dental follicle Dentigerous cyst UIP Radicular cyst Periapical granuloma Odontoma Pyogenic granuloma Nondiagnostic IFHa MEC UIP Fibrous hyperplasia Epithelial hyperplasia Gingival hyperplasia Papiloma Pyogenic granuloma Periapical granuloma Dentigerous cyst Radicular cystb MEC Dental follicle Pyogenic granuloma Dentigerous cyst Odontoma Keratocyst Radicular cyst CGCG Papilloma Fibrous dysplasia MEC Dental follicle Fibroma Odontogenic cyst CGCG Radicular cyst Dentigerous cyst Odontoma Chronic inflammation Pyogenic granuloma MEC Fibrous hyperplasia Dental follicle Dentigerous cyst Nondiagnostic Gingival hyperplasia Squamous papiloma Radicular cyst Odontoma Pyogenic granuloma
MEC Fibroma Pyogenic granuloma Dentigerous cyst Ameloblastoma Odontoma Radicular cyst Papilloma Traumatic ulcer Peripheral ossifying fibroma
Lima et al. [8] Southern Brazil 625 cases 0e14 yrs Vale et al. [9] Northeastern Brazil 315 cases 0e18 yrs Martins-Filho et al. [10] Northeastern Brazil 564 cases 0e18 yrs ^ a et al. [11] Pesso Northeastern Brazil 360 cases 0e19 yrs Cavalcante et al. [12] Northeastern Brazil 1240 cases 0e16 yrs Current study et al. (2016) Southeastern Brazil 2539 cases 0e16 yrs
Table 3 The 10 most frequent oral lesions observed in pediatric patients described in Brazilian studies.
described the distribution of pediatric oral lesions in United Kingdom and Australia, respectively, to better compare our results with previous reports. Although dental pathology represented the most common group of lesions in both studies, whereas salivary gland pathology was the most common in ours possibly because dental hard tissue abnormalities are usually not frequently submitted to microscopic exam in our laboratory, mucosal pathology, odontogenic cysts and gingival/periodontal pathology were the second, third and fourth most frequent categories and mucous extravasation cyst represented the most common entity in all three studies, suggesting that differences in the frequency of the main oral pediatric lesions is small and might be influenced by characteristics of each diagnostic service. Moreover, the highest incidence of mucous extravasation cyst in lower lip and fibrous/gingival hyperplasia is direct related to mechanical trauma what is relatively common in pediatric patients. As previously described, we also observed that older pediatric patients present a higher frequency of lesions, what may be consequence not only of the epidemiological features of each disease, but also because clinicians tends to postpone biopsies or surgeries considering that most of the pediatric lesions are reactive or benign in nature as exemplified in our study, where only nine cases were diagnosed as malignant (0.4%) [11,12,14]. Oral surgeries in pediatrics are usually small and may be under-representative for microscopic analyses, as a consequence, nondiagnostic cases accounted for 4.5% of our cases and comprised 2.4% of Jones et al. [13], 2.7% of Ha et al. [14] and 10.1% of Kezler et al. [5]. Although dental pathology did not account for the most common subgroup in our study as above mentioned, we believe that this finding more likely represents a peculiar feature of our service that very uncommonly receives dental hard tissue for analysis. Therefore, we agree with Ha et al. [14] who stated that dental lesions are usually under-represented in microscopic-based surveys. Similarly, gingivitis and odontomas can also have their frequency altered because these lesions are mostly diagnosed in a clinical and radiographic basis, respectively. Moreover, the origin of the data retrieved may also influence the results obtained, since hospitalbased studies are more likely to provide higher frequencies of benign and malignant tumors than dental school-based studies [15]. However, because population-based research on oral lesions would be very difficult to be done, we believe that microscopicbased studies well represent the distribution of these diseases, especially if the abovementioned limitations are considered in the interpretation of the results. In our sample we found a homogenous distribution of lesions between both genders, with a slight preponderance for females that accounted for 55.8% of the cases leading to a M:F ratio of 0.8:1.0. This fare distribution was described before [7,16] and was ^a et al. [11] to be consequence of the parents speculated by Pesso awareness with their children, since a higher frequency of females is found in adult populations [17], possibly representing the higher concern demonstrated by females than males. Despite small differences, the group of most frequent lesions observed in our study is in accordance with that observed in other international samples, with mucous extravasation cyst, dental follicles/dentigerous cyst and reactive periapical/gingival lesions ranking first in the lists. Because Brazil is an extensive and heterogeneous country both geographically and socioeconomically, we attempted to identify differences in the frequency of lesions by comparing our results with those previously published. There seems to be no study performed in Brazilian Central and Northern regions, but some authors have already described the distribution of oral lesions in pediatric patients from Northeast, South and Southeast regions of Brazil (Table 3). In Brazilian studies pediatric patients accounted from 6.6% [8] to 13.1% [9] of the samples, with a
Sousa et al. [7] Southeastern Brazil 2356 cases 0e14 yrs
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slight female predominance leading to a male/female ratio from 0.7 to 1.0 [9,10,12], both results are similar to ours. Up to 76 lesions could be described in a single analysis [7], whereas we identified 108 different entities, possibly because of the strict clinicopathological correlation usually done in our service. Methodologically, studies were very heterogeneous, using age range from 0 to 14, 0e16, 0e18 and 0-19 years-old, investigating lesions from 5 to over 20 years period and aggregating them in very different categories. On the other hand, only minor differences were found in the distribution of the most frequent lesions. As in this survey, mucous extravasation cyst predominated in all studies and dental follicle/ ^a et al. dentigerous cyst were also very common. However, Pesso [11] reported a high frequency of ameloblastomas and MartinsFilho et al. [10] described a large number of keratocysts; whereas odontoma was the most common odontogenic tumor found in most of the studies. Malignant neoplasms were always very rare [8]. Understanding the frequency and distribution pattern of oral lesions among pediatric patients is important not only for oral pathologists and dental pediatricians, but also to different medical specialists who first deal with these patients. In our daily practice, although no official statistics are available in our service, we frequently receive patients referred by otolaryngologists, pediatricians and dermatologists due to the presence of oral lesions. Therefore, we believe that the current results will improve the diagnostic algorithm of all these health professionals. Finally, this study demonstrated that distribution of oral lesions is very similar in different regions of the globe, with minor differences that may be consequence of methodological differences in the studies performed and in the characteristics of the services from where lesions were retrieved. Funding Dra. Aline Priscila Ataide was supported with a scholarship of the National Council for Scientific and Technology Development (CNPq) e Brazil. Ethics committee approval This study was approved by the Institutional Ethics Committee. Conflict of interest statement The authors state that they have no potential conflict of interest that could bias the results obtained in the current study.
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