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Int. J. Oral Maxillofac. Surg. 2010; 39: 145149 doi:10.1016/j.ijom.2009.11.022, available online at http://www.sciencedirect.

com

Clinical Paper Clinical Pathology

Clinicopathological study and treatment outcomes of 121 cases of ameloblastomas


E. R. Fregnani, D. E. da Cruz Perez, O. P. de Almeida, L. P. Kowalski, F. A. Soares, F. de Abreu Alves: Clinicopathological study and treatment outcomes of 121 cases of ameloblastomas. Int. J. Oral Maxillofac. Surg. 2010; 39: 145149. # 2009 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. The aim of this paper is to evaluate the clinical, radiographic, and histopathological ndings and treatment modalities in all cases of ameloblastomas treated at the Sao Paulo Cancer Hospital, between 1953 and 2003. 121 case reports were retrieved from the medical les. Data were reviewed and statistical analysis was performed using KaplanMeyer method and Cox proportional risk model. The patients age ranged from 2 to 82 years (mean 33.2 years), with a slight female prevalence. Most cases were located in the posterior mandible (80%). Radiographically, 60% showed a multilocular pattern. 113 casees were solid ameloblastomas, and plexiforme subtype was the most common. Solid tumours were treated by wide resection, curettage and criosurgery, or curettage alone, and unicystic tumours by curettage and/or cryotherapy. The global mean recurrence rate was 22%, with a mean follow-up of 9.7 years. The ameloblastomas were predominantly solid, affecting the posterior mandible. Important factors for outcome were radiographically multilocular lesions, the presence of ruptured basal cortical bone and histologically follicular tumours.

E. R. D. E. O. P. F. A.

Fregnani1, da Cruz Perez1, de Almeida2, L. P. Kowalski3, Soares4, F. de Abreu Alves1

1 Department of Stomatology, A.C. Camargo Cancer Hospital, Sao Paulo, Brazil; 2 Department of Oral Pathology, University of Campinas Dental School, Piracicaba, Brazil; 3 Department of Head and Neck Surgery, A.C. Camargo Cancer Hospital, Sao Paulo, Brazil; 4 Department of Pathology, A.C. Camargo Cancer Hospital, Sao Paulo, Brazil

Key word: ameloblastoma. Accepted for publication 30 November 2009 Available online 31 December 2009

Ameloblastomas comprise 1358% of all odontogenic tumours, and were rst described by Cusack in 1827 and detailed by Broca in 186825. The WHO classies ameloblastomas as benign odontogenic tumours formed by odontogenic epithelium with brous mature stroma, but without odontogenic ectomesenchime2. Solid/multicystic; unicystic and peripheral variants, with distinctive clinical, demographic and biological features are well-established25. Solid ameloblastomas are the most frequent, characterized by a slow but inltrative growth pattern and
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locally aggressive, occurring mainly on the posterior mandible of young adults (2536 years). Radiographically ameloblastomas are uni- or multilocular osteolytic lesions, frequently showing cortical expansion. Microscopically, the follicular and plexiform patterns are the most common, and other subtypes include acantomatous, desmoplastic, granular and basal. It is common to nd more than one histological variant in the same tumour2,24. Characteristically, polarized cells that resemble ameloblasts surround tumour cells, nests and cystic cavities.

The term unicystic ameloblastoma derives from the macro and microscopic appearances. The lesion is essentially a well-dened single cavity lined with ameloblastomatous epithelium, and three variants are found: luminal, intraluminal and mural10,22,26. Treatment of ameloblastoma is controversial. Unicystic ameloblastomas are usually treated conservatively with curettage and cryosurgery. There is discussion in the literature about different treatment approaches for solid ameloblastomas3,5,15,28.

# 2009 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

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Table 2. Recurrences of 113 cases of solid ameloblastomas according to the treatment modality. There were no statistical differences between treatment modalities and recurrences.*. Treatment Segmental resection Bone curettage + cryoterapy Bone curettage Total
*

The aim of this study was to evaluate the clinical, radiographic, and histopathological ndings and treatment modalities of 121 cases of ameloblastomas over 50 years at a single institution.
Material and methods

N total 47 47 19 113

N with recurrence 8 14 3 25

% of recurrence 17.0 29.8 15.8 22.1

The notes of all cases diagnosed as ameloblastomas between 1953 and 2003 were retrieved from the hospital les. Clinical, radiographical, treatment and follow-up information were obtained from the patients records. This is a long-term retrospective study so there are no treatment protocols because they varied according to the department in which the patient was treated. All hematoxylineosin stained slides were reviewed and the tumours classied according to 2005 WHO criteria2. Cases without enough data or pathological specimens (slides or paran block) were excluded from the study; 121 cases were included. Follow-up was calculated from the date of the rst treatment to the date of the last available assessment. The KaplanMeyer method was used to assess the probability of recurrence, and the log-rank test was used to compare the recurrence-free survival curves. Statistical assessment of the clinical and histopathologic variables was carried out by a multivariate regression analysis Coxs proportional risk model.
Results

x2 test: P = 0.252.

Most patients were Caucasians (72%), with a slight female predominance (53%), and ages ranged from 2 to 82 years (mean 33.2 years). The clinical size of the lesions ranged from 1.5 to 25 cm, with a mean of 5.5 cm. The main complaints were swelling in the involved area (83%) and/or pain (33%). Most cases were located in the posterior region of the mandible (80%). Table 1 shows further details of the distribution of all cases
Table 1. Site distribution of 121 cases of ameloblastoma. N Maxilla Right posterior region Left posterior region Anterior maxilla Mandible Right posterior region Left posterior region Anterior mandible Peripheral Total 12 2 9 1 108 50 47 11 1 121 % 9.9 1.6 7.4 0.8 89.2 41.3 38.9 9.1 0.8 100.0

studied. Most patients were treated initially at the A.C. Camargo Hospital, but 31 cases (26%) had been treated elsewhere with bone curettage (22 cases), segmental resection (6 cases) and marsupialization (3 cases). 113 cases were classied as solid (93%), 7 (6%) as unicystic and 1 as peripheral. Regarding the solid tumours, most of them (62%) were radiographically multilocular; 65% showed expansion and 25% discontinuity of the vestibular and/or lingual bone plate. The basal plate bone presented expansion in 40% of the cases and discontinuity in 13%. Histologically, most cases were classied as plexiform (53%) or follicular (34%). Other histological subtypes (13%) identied were acantomathous (7 cases), granular (5 cases), desmoplastic (2 cases) and haemangiomatous (1 case). Regarding the treatment of the 113 solid ameloblastomas: 47 cases were treated with segmental resection, 47 with bone curettage followed by cryotherapy, and 19 cases with bone curettage only. Diagnosis of unicystic ameloblastoma was conrmed in seven cases, considering the image examinations, macroscopic and histologic ndings. The mean age was 35 years; ve cases affecting women. All cases showed unilocular images and 4 were of the mural histological variant. All cases were treated conservatively by curettage or curettage and cryotherapy.

The only patient affected by an extraosseous lesion was a 29-year-old Caucasian man. The lesion (3 cm in size) was located on the left mandibular alveolar edge and it was treated by enucleation. The histological aspect of this case was predominantly follicular. The mean follow-up time was 9.7 years and 27 patients presented recurrences, 25 of the 113 (22%) with solid ameloblastomas and 2 of 7 (29%) with unicystic lesions. Extra-osseous recurrences occurred in four solid ameloblastomas. The two unicystic cases that recurred were of the mural subtype; one had been treated with curettage associated with cryotherapy, and the other with curettage only. Table 2 shows the data for recurrences of solid ameloblastomas. The x2 test indicated that there were no signicant statistical differences between recurrences and type of treatment performed. Table 3 shows the main data for solid ameloblastomas cases, as well as the results of Coxs univariate regression for analysis of the clinical variables correlated with recurrences. Of the clinical variables, only the radiographic pattern and the basal cortical bone status were statistically signicant. Patients with radiographically multilocular lesions had 3.02 times more chance of recurrences than patients with unilocular lesions (Fig. 1). Patients who presented only expanded basal cortical bone had

Fig. 1. Disease-free survival function curves according to the radiographic pattern.

Clinicopathological study and treatment outcomes in ameloblastoma


Table 3. Main ndings of 113 cases of solid ameloblastomas and univariate analysis. Variable Age (years) Category <18 1832 >32 Afro Brazilian Caucasian Male Female 12 >12 <5 510 >10 Mandible Maxilla No Yes No Yes Segmental resection Bone curettage + cryotherapy Bone curettage Unilocular Multilocular Torn Expanded Preserved Torn Expanded Preserved No Yes Follicular Plexiform Others* N total 16 50 47 27 86 54 59 58 47 35 39 6 102 11 67 41 84 29 47 47 19 38 62 28 65 7 11 34 40 101 4 38 60 15 N (%) with recurrence 4 (25.0) 15 (30.0) 6 (12.8) 6 (22.2) 19 (23.2) 10 (18.5) 15 (25.4) 13 (22.4) 10 (21.3) 6 (17.1) 12 (30.8) 0 (0.0) 24 (23.5) 1 (9.1) 13 (19.4) 11 (26.8) 14 (16.7) 11 (37.9) 8 (17.0) 14 (29.8) 3 (15.8) 4 (10.5) 18 (29.0) 8 (28.6) 13 (20.0) 0 (0.0) 6 (54.5) 7 (20.6) 7 (17.5) 23 (22.8) 0 (0.0) 12 (31.6) 8 (13.3) 5 (33.3) HR 1.00 1.37 0.45 1.00 0.90 1.00 1.94 1.00 1.04 1.00 1.97 1.00 0.35 1.00 1.30 1.00 2.04 1.00 1.91 0.94 1.00 3.02 1.00 0.57 1.00 0.24 0.15 1.00 1.00 0.31 0.90 CI 95% 0.454.16 0.131.60 0.362.28 0.854.46 0.452.45 0.735.34 0.052.56 0.572.97 0.904.59 0.774.75 0.243.65 1.019.04 0.231.40 0.080.74 0.050.49 0.120.80 0.322.56

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P 0.581 0.216 0.830 0.117 0.923 0.183 0.299 0.535 0.086 0.161 0.932 0.048 0.224 0.013 0.002 0.015 0.844

Ethnic Gender Time of complaining (months) Clinical tumour size (cm)

Tumour site Soft tissue inltration Previous treatment Treatment

Radiographic pattern Cortical bone V e L(P)

Basal cortical bone

Pathologic fracture Main histopathological pattern

* Other histological pattern: acantomathous (seven cases/three recurrences), desmoplastic (two cases/one recurrence), granular cell (ve cases, no recurrences) and haemangiomatous (one case/one recurrence). HR: hazard ratio; CI: condence interval.

0.24 times more chance of recurrence than those with ruptured basal cortical bone. Patients with preserved basal cortical bone had 0.15 times more chance of recurrence than patients with ruptured basal cortical bone. This parameter was analysed only in mandibular cases. Considering the microscopic aspects, predominantly plexiform cases had 0.31 times more chance of recurrence than those with a follicular pattern. During the histopathological review, it was observed that 87 cases (77%) of solid tumours exhibited cystic areas (predominantly microcystic in 36 cases; macrocystic in 38 and both in 13 cases). In addition, areas with intense mesenchymal brosis were observed in 28 cases. These variables were not statistically signicant for recurrences. When the multiple regression analysis was performed, taking into consideration

the three most signicant variables (radiographic pattern, basal cortical bone status and main histological aspect), only the variable status of the basal cortical bone remained. This variable was considered the most important for recurrence of the disease.
Discussion

Epidemiologic studies have shown considerable differences in the incidence of odontogenic tumours, particularly when African and Asiatic studies are compared with those of North America1,7,17,18,23,32. These variations in incidence are justied by geographic, ethnic and socio-economic factors or by the institution where the data were collected. Data obtained from medical centres usually show a predominance of ameloblastomas, while studies from

dental centres present a greater prevalence of odontomas9. Most literature regarding odontogenic tumours is biased depending on the institution where the data were collected9. The epidemiological prospective study from SIMON et al.30 showed no racial differences. In the present study, ameloblastomas corresponded to 67% of all odontogenic tumours, which conrms the bias found in studies from medical centres. Therefore, except for odontomas, ameloblastomas are the most common odontogenic tumours, and important clinical and biological controversies persist in relation to them. In the literature, there are few studies considering the clinical-pathologic characteristics of ameloblastomas in Latin America8,16,21. The present series of 121 cases showed a mean age of 33.2 years, which is in

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recurrences reported after curettage and cryotherapy of ameloblastomas is considered high, and therefore this form of treatment should be selected for small or unicystic lesions. CARLSON & MARX3 carried out an extensive literature review, and concluded that radical resection is the only form of treatment for ameloblastomas with predictable results. The rates of recurrence for solid ameloblastomas range between 15% and 25% after radical treatment, as in the present study. The present cases treated with curettage presented a similar value of recurrence (16%), differing from the rates of 3580% described in the majority of studies11,12,14,20,28,29. This can be explained because the present cases treated only with curettage were relatively small (mean 4 cm) which is an indicator of good prognosis, as previously shown, and large tumours with ruptured basal cortical bone were treated more aggressively. The three recurrences were associated with ruptured basal cortical bone. Cases that presented large dimensions with cortical thinning and/or rupture received more radical treatment. Although controversial, it is considered that unicystic ameloblastomas with mural inltration require treatment similar to that of solid ameloblastomas. LAU & SAM15 carried out a systematic review of MAN the treatment of unicystic ameloblastomas which revealed that treatment by resection showed the lowest incidence of recurrence (4%), followed by enucleation with the application of Carnoy solution (16%) and simple enucleation (31%). In the present study, two of seven cases of unicystic ameloblastomas presented recurrences (29%), both had mural inltration; one was treated with curettage followed by cryotherapy and the other with simple curettage. The present study is more than a retrospective single institute evaluation and shows, based on a univariate regression analysis, interesting data about the prognostic factors of recurrence. A signicant number of conservative and segmental surgeries are compared with long-term follow-up. To conclude, the authors evaluated clinical, radiographic and histopathological ndings from 121 cases of ameloblastomas from a single institution, and found that the tumours were predominantly solid, and affected the posterior mandible. The data showed that cases treated by segmental resection presented less recurrence, and that radiographically multilocular lesions, the presence of ruptured basal cortical bone and histologically follicular ameloblastoma seem to be important factors for prognosis.
Funding

agreement with the meta-analysis carried out by REICHART et al.24 and with a recent multicentric study in the Latin American population16. Most studies show a similar incidence between men and women, with slight variations16,17,24. The present data showed a slightly higher number of women (53%). The posterior region of the mandible is the location of greatest incidence of both solid and unicystic ameloblastomas, and this was conrmed in the present study2,25. Solid ameloblastomas were the most common (93%) and only seven cases (6%) were considered unicystic. This accords with the majority of other studies24, although a Latin American multicentric study showed that 63% of cases were unicystic ameloblastomas16. The authors think that more uniform criteria to diagnosis ameloblastomas as unicystic or solid is necessary. Although the present series had only 7 cases considered unicystic, the mean age for these cases was higher compared with other studies22. Radiographic characteristics seem to be relevant for the prognosis of ameloblastomas4,6,19,20,27. The present solid cases with a multilocular radiographic image, presented a signicantly higher incidence of recurrences. A ruptured mandibular basal cortical bone indicated a three times higher risk of recurrence, compared with cases of preserved or expanded cortical bone. Most studies show that for solid ameloblastomas the predominant histological pattern has no relevance for recurrences. According to the present data and those of HONG et al.13, considering the two most frequent subtypes, plexiform and follicular, the latter presented a signicantly higher number of recurrences. This is controversial and more studies are necessary to conrm or refute this observation. Various studies considering treatment criteria and protocols for solid ameloblastomas have been published, but the subject remains controversial. Recent advances in the understanding of the biologic behaviour of this tumour have provided new treatment protocols4,27,28,31. Cryotherapy is a form of adjuvant treatment that can be used in benign intraosseus lesions, including ameloblastoma, with the aim of eliminating residual tumour cells inside the bone trabeculae by freezing. This has not been widely used in ameloblastomas, but it can be useful when more conservative treatment is performed. It was shown that after cryotherapy, when recurrences occur they are small and well limited, facilitating complete removal of the lesion5. The 31% of

The State of Sao Paulo Research Foundation (FAPESP).


Competing interests

None declared.
Ethical approval

Approved by the Human Research Ethics Committee of the A.C. Camargo Hospital, # 669/05.
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Address: Eduardo Rodrigues Fregnani Department of Stomatology A.C. Camargo Cancer Hospital Sao Paulo Brazil Tel.: +55 11 2189 5000 E-mail: eduardofregnani@me.com

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