Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero-Castaño N, Lo Muzio L. Ameloblastoma of the jaw and maxillary bone: clinical study and report of our experience

Ameloblastoma of the jaw and maxillary bone: clinical study and report of our experience

Di Cosola M*, Turco M*, Bizzoca G*, Tavoulari K*, Capodiferro S*, Escudero- Castaño N**, Lo Muzio L***

SUMMARY

In order to ameloblastoma´s cases series, benign tumour of epithelial origin, we will value the histological and radiographic common findings, its frequent symptomatology, and we will estimate the prevalence variation according to the age, gender, lesion localization, etc. In the other hand, we will emphasize the principal risk factors and we will classify the different treatment according to its histology, clinic and type of lesion. Key words: Solid-multicystic ameloblastoma, unicystic ameloblastoma.

Receibed: Febrero 2007. Acepted for publication: Marzo 2007.

* Department of Dentistry and , University of Bari, Italy. ** Departament of Oral Medicine and Bucofacial Surgery, Complutense University of Madrid, Spain. *** Department of Oral Pathology, University of Foggia, Italy.

Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero-Castaño N, Lo Muzio L. Ameloblasto- ma of the jaw and maxillary bone: clinical study and report of our experience. Av. Odontoestomatol 2007; 23 (6): 367-373.

INTRODUCTION Reichart and Philipsen (5), in an analysis of the three previously mentioned entities, state that the average The ameloblastoma is a relatively rare dental tumor, age of the patients with ameloblastoma is 36 years. described for the first time by Broca in 1868, and so Gardner critesizing such review( 6), calculated a 39 denominated by Churchill in 1934. year-old average age for the solid multicystic amelo- blastoma, 22 for that unicystic ameloblastma and of According to Larsonn and Almeren (1), its 51 years for the peripheral ameloblastoma. Equal incidence is 0,6 cases per million, while Shear and incidences have been found in the two sexes by Singh (2) found an incidence of 0.31 cases per Reichart. million in a white population of Witwatersrand in South Africa. Between 1975 and the beginning of the ’80, the concept that the ameloblastoma exists MATERIAL AND METHODS in three different clinical/histopatological forms was accepted (3-4): solid-multicystical, unicystical and Ten cases (10) of ameloblastoma were identified in peripheral. the department of Dentistry and Surgery of the

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University of Bari-Italy between 1990 and 2006. They maxillary bones, therefore the region in front of the were evaluated and classified clinically, histologically first molar. and radiographycally, based on the cytological criteria of Vickers and Gorlin (21) and World Health Orga- Analysing the three histological types separately we nization (25) guide lines. found that 7 of the 8 cases of solid ameloblastoma were located in the jaw, 1 of these in the posterior The clinical-pathological data was evaluated: age, sex, third, and 1 in the upper . All the unicystic site and dimensions of the lesion, primary and ameloblastomas (2) were located in the posterior third secondary symptoms and radiographic aspects and of the jaw. characteristics. Furthermore the possible presence and entity of recurrence in relationship to the histological nature of the lesions was examined. The Size of tumor size of the tumor was measured from the and the greater mesio-distal and apico- In all patients the mesio-distal dimensions were occlusal diameters were recorded. greater than 2,5 cm (Figs. 1-3).

We also considered the type of treatment, results in relationship to the hystological, clinical and pathol- Symptoms ogical criteria of the various types of lesion, with a follow-up ranging from 1 to 15 years. The symptoms were recorded in all cases: in 8 patients the main symptom was a non aching swelling of the maxillary bones, 2 were accompanied by pain. RESULTS In 4 cases the increase of volume of the lesion was described up until the hospitalisation; in 1 case pain Frequency was the only symptom. In 2 patients rhizolisi was

10 cases of ameloblastoma were identified during this study. According to the hystological classification (25) we found: 8 solid-multicystic, 2 unicystic.

Age and gender: The average age of all the patients, during the diagnosis, was 39.6 years. The male/ female relationship calculated for the whole group was 1,3:1, for the solid ameloblastomas 1,4:1 and 1,25:1for the unicisticis ameloblastomas.

All the patients were of Caucasian race and of Italian Fig. 1. nationality.

Site distribution

9 cases were located in the jaw and 1 in the upper maxilla. The ratio between the upper maxilla and jaw was 9:1. In 7 cases the ameloblastoma was located in the posterior third of the maxillary bones, including the area of the molar teeth and the structures distal to them. In 3 cases the tumor was found in the anterior and /or middle part of the Fig. 2.

368/AVANCES EN ODONTOESTOMATOLOGÍA Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero-Castaño N, Lo Muzio L. Ameloblastoma of the jaw and maxillary bone: clinical study and report of our experience

Fig. 3. observed. Paraesthesia was observed only in 1 case as a secondary symptom. In 2 cases we found the non aching ulceration of the overlying oral mucosa Fig. 4. (Fig. 4). In 4 cases the discovery of the lesion was chance, after performing an x-ray for other medical purposes. The average time between the debut to ameloblastoma were recognised and classified based the diagnosis was 9,7 months. on the Ackerman classification into: 1 type III (50%) and 1 type II (50%).

Radiographic findings Treatments and recurrence The radiographic aspect in 8 cases was multilocular and in 2 cases unilocular. In the case of maxillary The cases have been divided into 3 groups based on ameloblastoma the opacity of the maxillary sinus was the clinical entity ( multicystical or unicystical) and observed. the treatment received ( conservative or radical):

— Group 1: multicystic ameloblastoma with conser- Histological findings vative therapy, (cases from 1 to 6). — Group 2: multicystic ameloblastoma with radical The histological diagnosis and the classification therapy, (cases from 7 to 8). based on the actual criteria (25) allowed us to — Group 3: unicystic ameloblastoma with conser- indentify 8 solid-multicistic amelobblastomas. The vative therapy (cases from 9 to 10). solid follicolar ameloblastoma were 4 (50%), the plessiforme 2 (25%), the acantomatosis 1 (12,5%) Conservative surgical therapy included enucleation, and the mixed 1 (12,5%). The 2 cases of unicystic curettage, electrocautery, excision and stitching. Ra-

AVANCES EN ODONTOESTOMATOLOGÍA/369 AVANCES EN ODONTOESTOMATOLOGÍA Vol. 23 - Núm. 6 - 2007 dical surgery also includes marginal resection, seg- lesion out of 2 had dimensions greater than 3 cm at mental or total resection of the with wide the hospitalisation. margins. In the group of the unicystic ameloblastomas (cases The analysis of the first group (6 patients) showed 3 9-10) the only recurrences had with dimensions cases of recurrences. The average time between the greater than 3 cm at hospitalisation were observed. treatment and the recurrences was 3,5 years (range By each the location, in the first group we have 4 months-10 years). noticed only 3 recurrences out of a total of 6 with location in the anterior and middle third of the 2 of the 3 recurrences were treated again with maxillary bones, while the remainder 3 lesions were conservative therapy. located in the posterior third recurred. In the second group there was only one lesion located in the poste- In one case the treatment of the recurrence consisted rior third. in the resection of the jaw without continuity of the inferior edge and a bone autograft, which has not In the group of the unicystic ameloblastoma, we recurred. found one recurrence located in the posterior part of the jaw. In the second group of solid ameloblastomas the 2 patients were treated with radical surgery. DISCUSSION In this group the only case of recurrence was observed after 5 years, in spite of the resection of the jaw with The ameloblastoma is statistically more frequent in solution of continuity of the inferior edge the the molar region and branch of the jaw, while in the recurrence was diagnosed and treated with a surgical maxilla it is more often found in the molar region, extension of the borders. even if in some cases the maxillary cavity is interested.

The third group with unicystic ameloblastomas, is The 80% of these cases were solid-multicystical and composed of 2 patients with an initial conservative the remaining 20% unicystic. This data differ from treatment. Reicharts studies (5), which estimates 51% the incidence of the solid-multicystical lesion and the 49% The unicystic ameloblastomas recurred in average for the unicystic. Such discrepancy can only be after 3.5 years. determined by the small amount of patients considered in our study. From the analysis of our There was one recurrence in this group. It was cases an agreement emerged with Kramer and Regezi possible to reconstruct the following therapeutic et, to the (11), we found no correlation among clinical history: first treated with conservative therapy, symptoms, biological behavior and histological recurred for the second time and a jaw resection with subtype. According to Reichart (5) the most common solution of continuity of the inferior edge was histological type is the follicolar lesion 50% compared necessary but successful. to 33,9% proposed by literature.

According to Shaw and Katsikas (26) none of their Clinical and histological factors of risk for cases followed chemio- or radiotherapy, which are recurrence only indicated in case of remaining tumor tissue after lesion resection or as a palliative therapy. In case of The first group (cases 1-6) had 3 recurrences out of upper maxillar lesions radical resection is advised, a total of 6 with dimensions greater than 2 cm at the due to the spongy osteoarchitecture of the maxilla moment hospitalisation and 1 recurrences had which facilitates the diffusion of the tumor to the dimensions inferior than 2 cm at the time of sinus ethmoidalis, pterygoidea fossa, temporal fossa hospitalisation. In the second group (cases 7-8), 1 and base of skull (21,25). In our study we had only

370/AVANCES EN ODONTOESTOMATOLOGÍA Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero-Castaño N, Lo Muzio L. Ameloblastoma of the jaw and maxillary bone: clinical study and report of our experience one case of ameloblastoma located in the upper We believe that for the diagnostic phase the instru- maxilla, it were treated with conservative surgery. mental examinations (X-RAY, CT or MRT) are Recurrences were encountered in the patients, that essential; while intralesional byopsies are inefficient were over 65 years old, for whom this type of because they don ‘t offer a whole vision of the tumor treatment was chosen because of the limited life span and could lead to diagnostic error. Therefore we think left; but for the young patients the literature suggest that it is advisable, considering: the site and extention, a conservative approach in pediatric patients. of the lesion, age and general conditions of the patient, to remove the lesion in a conservative manner Considering the fact that the conservative treatment in a first surgical step and according to the histolgical of the maxillar bones offers local recurrence in100% aspect evaluate a possible radical resection. of the cases and a 60% of mortality rate (26-28-32), it is important that the patients with this pathology have a life long follow-up with the help of computer BIBLIOGRAFÍA tomography (CT) and magnetic resonance tomography (MRT). The therapy of the ameloblasto- 1. Larsson A Ê, Almeren H. Ameloblastoma of the ma is surgical, but it has not been established yet jaws. An analysis of a consecutive series of all which is the most suitable operation. The decision to cases reported to the Swedish Cancer Registry use a radical or conservative approach depends on during 1958-1971. Acta Pathologica et Micro- various factors: 1) the dimensions and the location biologica Scandinavica (A) 1978;86:337-49. of the lesion, 2) the growth rate and the relationship with the nearby structures, 3) the histological type, 4) 2. Shear M, Singh S. Age-standardized incidence the clinical characteristics, in the recurrences, 5) the rates of ameloblastoma and dentigerous on general conditions of health and the age of the patient the Witwatersrand, South Africa. Community (14). Among this series of factors the histological Dentistry and Oral Epidemiology 1978; 6:195-9. aspect has a decisive role: the solid multicystic ame- loblastoma and the unicystic-intramural ameloblas- 3. Gardner DG, PecË sak AMJ. The treatment of tomas, subtype III of Ackerman, need radical ameloblastoma based on pathologic and treatment, with the resection of 1-2 cm of healthy anatomic principles. Cancer 1980; 46:2514-9. bone (12-15-17), although not all the authors have found high rates of recurrence after the conservative 4. Robinson L, Martinez MG. Unicystic ameloblas- treatment of such forms (5). On the contrary the toma: a prognostically distinct entity. Cancer unicystic intraluminal ameloblastoma rarely recurrers 1977;40:2278-85. after conservative therapy, only 10 % according to Leider (15), Eversole (16), Gardner (17) and 13,7% 5. Reichart PA, Philipsen HP, Sonner S. Ameloblasto- according to Reichart (5). Therefore a conservative ma: biological prole of 3677 cases. Oral , aproach it is advisable in the case of a non invasive European Journal of Cancer 1995; 31B:86-99. and non proliferating cyst wall. The location of the lesion seems to be an important risk factor for the 6. Critique of the 1995 review by Reichart et al.$ of recurrence of the ameloblastoma in fact the most the biologic erole of 3677 ameloblastomas D.G. frequent recurrence were located in the third poste- Gardner Oral Oncology 1999;35:443-9. rior of the jaw. 7. Khan MA: Ameloblastoma in young persons: A clinicopathologic analysis and etiologic CONCLUSION investigation. Oral Surg Oral Med Oral Pathol 1984; 67:706. We think that the product of our experience, is useful for an efficient programming of treatment, taking in 8. Eversole LR, Leider AS, Strub D. Radiographic consideration numerous variables of this pathology characteristics of cystogenic ameloblastoma. and the indications found in literature. Oral Sug Oral Med Oral Pathol 1984;57:572-7.

AVANCES EN ODONTOESTOMATOLOGÍA/371 AVANCES EN ODONTOESTOMATOLOGÍA Vol. 23 - Núm. 6 - 2007

9. Gardner DG: Plexiform unicystic ameloblastoma: case report and review of the literature. A diagnostic problem in dentigenous . International Journal Oral Maxillofacial Surgery Cancer 1981; 47:1358. 1992;21:40-4.

10. Struthers P, Shear M: Root resorption by Amelo- 21. Vickers RA, Gorlin RJ. Ameloblastoma: blastomas and . Int J Oral Surg delineation of early istopatologic features of neo- 5:128, 1976. plasia. Cancer 1970;3, 699-710.

11. 258 Kramer IR. Ameloblastoma: a clinico- 22. Kramer IRH, Pindborg JJ, Shear M. Histological pathological appraisal. Br J Oral Surg 1963;1:13- typing of odontogenic tumours. 2nd ed. Berlin: 28. Springer-Verlag; 1992.

12. Regezi JA, Kerr DA, Courtney RM. Odontogenic 23. Leider AS, Eversole LR, Barkin ME. Cystic ame- tumors: analysis of 706 cases. J Oral Surg loblastoma. A clinopathologic analysis. Oral Sug 1978;36:771-8. Oral Med Oral Pathol 1985;60:624-30.

13. Ackerman GL, Altini M, Shear M: The unicystic 24. Eversole LR, Leider AS, Strub D. Radiographic ameloblastoma: A clincopathological study of 57 characteristics of cystogenic ameloblastoma. cases. J Oral Pathol 1988;17:541. Oral Sug Oral Med Oral Pathol 1984;57:572-7.

14. HP Philipsen A, PA Reichart BH. Nikai CT. Takata 25. Eggert JH, Steinhauser EW. Besonderheiten der CY. Kudo. Review Peripheral ameloblastoma: Oberkiefer-Ameloblastome Beobachtungen an biological role based on 160 cases from the 12 Fallen. Fortschr Kiefer Gesichtschir 1986;31: literature. Oral Oncology 2001; 37:17-27. 101-4.

15. Ueno S, Nakamura S, Mushimoto K, Shirasu R. 26. Shaw HJ, Katsikas DK. Ameloblastoma of the A clinicopathologic study of ameloblastoma. maxilla. A clinical study with four cases. J Journal Oral Maxillofacial Surgery 1986;44:361-5. Laryngol Otol. 1973;87:873-84.

16. Waldron CA, El-Mofty SK. A histopathologic study 27. Eichhorn T, Glanz H, Kleinsasser O. Amelo- of 116 ameloblastomas with special reference to blastome des Oberkiefers. Bericht uber 6 Falle the desmoplastic variant. Oral Surgery Oral Me- und Literaturzusammenstellung. HNO 1982; dicine Oral Pathology 1987;63:441-7. 30:1-8.

17. Rick G, Howell F, Pindborg J. The peripheral 28. Batsakis JG, Mclatchey KD. Ameloblastoma of Ameloblastoma: a clinicopathologic study of 18 the maxilla and paripheral ameloblastomas. Am cases. Journal Oral Pathology 1985;14:85. Otol Rhinol Laryngol 1983;92:532-4.

18. Niccoli-Filho W, Gomes da MGME, Raldi FV, 29. Pilz G, Nitzschke M. Uber die Spatrezidivierung Seraidarian PI. Peripheral ameloblastoma. der Ameloblastome. Stomatol DDR 1979;29: Journal Nihon University School of Dentistry 107-11. 1997;39:34-7. 30. Sandler KA, Novo RM, Rudner BE. A study of 19. Buchner A, Sciubby JJ. Peripheral epithelial ameloblastoma- age, sex, location statistics. NY odontogenic tumors: a review. Oral Surgery Oral State Dent J 1983; 49: 682-4. Medicine Oral Pathology 1987;63:688-97. 31. Young DR, Robinson M, Ameloblastoma in 20. Nauta JM, Panders AK, Schoots CJF, Vermey A, children: report of a case. Oral Sug Oral Med Roodenburg JLN. Peripheral ameloblastoma. A Oral Pathol 1962; 15: 1155-62.

372/AVANCES EN ODONTOESTOMATOLOGÍA Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero-Castaño N, Lo Muzio L. Ameloblastoma of the jaw and maxillary bone: clinical study and report of our experience

32. Sehdev MK, Huvos AG, Strong EW, Gerold FP, 34. Gardner DG. A pathologyst’s approach to the Willis GW. Proceedings: Ameloblastoma of treatment of ameloblastoma. J Oral Maxillofac maxilla and mandible. Cancer 1974; 33: 324- Surg 1984; 43: 161-6. 33. 35. Philipsen HP, Reichart PA. Revision of the 1992 33. Menakanit W. Ameloblastoma of the jaw: a 10 edition of the WHO histological typing of odonto- year analysis (1966-1975). J med Assoc Thai genic tumors. A suggestion. J Oral Pathol Med 1979; 62: 6-9. 2002;31:253-8.

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