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The clinical case of an unusual ameloblastic fibroodontoma (AFO) was reported. The patient’s clinical chart as well as preoperative and postoperative radiographs and histological findings of a 20-year old man that addressed Dental Clinic at University of L’Aquila were thoroughly reviewed. The patient showed a swelling in the oral cavity and radiographic feature of a radiolucent lesion at left second premolar maxillary site. Histologic examination made diagnosis of AFO. AFO is a rare mixed odontogenic tumor with similarities to the ameloblastic fibroma (AF) and ameloblastic dentinoma. The nature and the relationships between mixed odontogenic tumours and related lesions are still controversial. Moreover is not clear if these lesions are separate pathologies or if they are different development stages of the same pathology.
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Ameloblastic fibro-odontoma: a case report
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Ameloblastic fibro-odontoma: a case report
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               Ameloblastic fibro-odontoma: a case report




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               Stefano Mummolo, DDS                                            the AFO one or more cellular foci continue differentiation
               Enrico Marchetti, DDS                                           process and produce enamel and dentin.
               Salvatore Di Martino, MD, DDS                                   This lesion is often an incidental radiographic finding. Ra-
               Luisa Scorzetti, DDS                                            diographically, the tumor appear well circumscribed,
               Giuseppe Marzo, MD, DDS                                         round-to-ovoid radiolucency, surrounded by a thin sclerotic




                                                                                                           zi
                                                                               margin (1). According to the recent WHO classification of
               University of L’Aquila, Italy                                   Odontogenic Tumors published in 2005, AFO is a beni-
               Department of Health Sciences (A. Sotgiu)                       gn tumor without invasive growth that belongs to the group




                                                                                           a
                                                                               of lesions with odontogenic epithelium with odontogenic
               Corresponding author:                                           ectomesenchyme, with or without hard tissue formation (2).
                                                                               There is considerable debate in literature regarding the re-




                                                                                        rn
               Dott. Stefano Mummolo
               Ospedale S. Salvatore                                           lationship between AFO and other mixed odontogenic tu-
               Clinica Odontoiatrica                                           mors. Some authors assert that AFO is a mature amelo-
               Edificio delta 6                                                blastic fibroma whereas other ones think it could be a pre-




                                                                               te
               67100 L’Aquila, Italy                                           cursor of odontoma (3).
               E-mail:stefano.mummolo@cc.univaq.it                             It is rare in the jaw, where only about 2% of all cases have
                                                                               been reported. Focused literature revealed that neopla-
                                                                               sm occur predominantly in children and young adults. An
                                                                     In        equivalent incidence in both upper and lower jaws and no
                                                                               gender predilection were reported (4).
                                                                               Clinically, the size of the tumor shows marked variations,
               Summary                                                         ranging from lesions detectable only microscopically, to
               Ameloblastic fibro-odontoma: a case report.                     giant tumors consisting of extensive calcified masses. Ra-
                                                           ni
                                                                               diographs usually show a well-defined radiolucent area con-
               The clinical case of an unusual ameloblastic fibro-             taining various amounts of radiopaque material of irregular
               odontoma (AFO) was reported. The patient’s clinical             size and form (2,5).
                                                io

               chart as well as preoperative and postoperative radi-           The aim of the current study was to report a clinical case
               ographs and histological findings of a 20-year old man
                                                                               of AFO and the long-term results after surgical treatment.
               that addressed Dental Clinic at University of L’Aquila
               were thoroughly reviewed. The patient showed a
                                       iz



               swelling in the oral cavity and radiographic feature of a
               radiolucent lesion at left second premolar maxillary            Case report
               site. Histologic examination made diagnosis of AFO.
                      Ed




               AFO is a rare mixed odontogenic tumor with similari-            A 20-year-old Caucasian male was referred to the Den-
               ties to the ameloblastic fibroma (AF) and ameloblastic          tal Clinic at University of L’Aquila with an asymptomatic
               dentinoma. The nature and the relationships between             intra-oral swelling. Neither dental history reported local trau-
               mixed odontogenic tumours and related lesions are               ma or infection at lesion site, nor medical history revea-
               still controversial. Moreover is not clear if these le-         led remarkable systemic diseases.
               sions are separate pathologies or if they are different         Panoramic radiography showed a rounded, well-defined,
               development stages of the same pathology.                       radiolucent lesion at upper left second bicuspid and first
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                                                                               molar edentulous sites. It contained a radiopaque mass
               Key words: ameloblastic fibro-odontoma (AFO), odonto-           of apparently calcified material in proximity of the root of
               genic tumor.
                                                                               adjacent first bicuspid (Fig. 1). CT scan showed a
                                                                               23x17mm osteolytic lesion in the left body of maxilla with
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                                                                               lobulated and well-demarcated margins (Fig. 2). The bor-
                                                                               ders of the lesion were in part radiopaque, similar to cor-
               Introduction                                                    tical bone (Fig. 3). Radiological findings were consistent
                                                                               with a benign bone tumour. Clinical examination revealed
©




               The ameloblastic fibro-odontoma (AFO) is a rare , slow-         a circumscribed swelling in the vestibule on the left side
               growing, odontogenic tumour. This benign neoplasm               of the maxilla, with an unaffected mucosa.
               has been defined by the World Health Organization               Under general anaesthesia, the tumour was removed th-
               (WHO) as “a neoplasm composed of proliferating odon-            rough intra-oral approach. The first premolar was found
               togenic epithelium in a cellular ectomesenchymal tissue         to be involved by the mineralized mass.
               with varying degrees of inductive changes and dental hard       Microscopically examination of sections stained with he-
               tissue formation” .The lesion has histologic feature and bio-   matoxylin and eosin showed scattered cords of odonto-
               logic behavior similar to the ameloblastic fibroma, but in      genic epithelium surrounded by a large amount of cellu-

               Annali di Stomatologia 2010; I (2): 11-13                                                                                    11
0373 4 Ameloblastic_Mummolo:-        30-11-2010      18:57    Pagina 12




               S. Mummolo et al.




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               Figure 1 - Panoramic radiography.




                                                                                                            zi
                                                                               Figure 3 - CT – parasagittal image.




                                                                                           a
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                                                                               te
                                                                     In
               Figure 2 - CT – axial image.
                                                        ni
               lar connective tissue. In the mass of fibrous tissue, calci-
               fied areas were seen that were consistent with mature den-
               tin formation and enamel matrix (3).
               Post-operative clinical course was uncomplicated (Fig. 4).
                                              io


               Finally, at 8 years-follow-up after implant-supported reha-
               bilitation, no signs of recurrence were reported (Fig. 5).
                                      iz



                                                                               Figure 4 - CT – axial image after surgical treatment.
               Discussion
                      Ed




               Histologic examination showed a benign ectomesenchy-
               mal neoplasia of odontogenic origin characterized by the
               proliferation of islands, nests and cords of epithelial cel-
               ls the exhibited a palisaded arrangement at the periphery
               and centrally a loose arrangement resembling the stella-
               te reticulum of the enamel organ. The mesenchyme of the
               lesion was characterized by stellate or spidle-shaped cel-
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               ls, eosinophilic substance compatible with dentinoid ma-
               terial and basophilic material compatible with elementary
               enamel was observed (5).
               The differential diagnosis of ameloblastic fibro-odontoma
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               and ameloblastic fibroma is based on the presence or ab-
               sence of elements indicative of differentiation of the
               tooth germ. The presence of both dentin and enamel is es-
               sential to call the tumor “ Ameloblastic fibro-odontoma” (6).
©




               The distinction between developing complex odontoma and
               AFO is impossible sometimes. However the presence of
               great amounts of enamel, dentin, and cementum-like tis-
               sue arranged in a haphazard pattern suggests a diagno-
               sis of odontoma (7).
               Ameloblastic fibrodentinoma is considered by some
               authors as a stage between the ameloblastic fibroma (AF)        Figure 5 - Periapical radiography – 8 years-follow-up after im-
               and AFO based on extent of histodifferentiation (8,9). Many     plant-supported rehabilitation.


               12                                                                                     Annali di Stomatologia 2010; I (2): 11-13
0373 4 Ameloblastic_Mummolo:-        30-11-2010        18:57      Pagina 13




               Ameloblastic fibro-odontoma: a case report


               authors have reported that AFO is not aggressive and can                lofac Surg 1999;57:620-623.
               be treated adequately with surgical curettage of the le-             4. Hammad H.M., Hammond H.L., Kurago Z.B., Frank J.A.
               sion without removing the adjacent teeth (10). The dif-                 Chondromyxoid fibroma of the jaws. Case report and review
               ferential diagnosis for ameloblastic fibro-odontoma should              of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol
               include ameloblastic fibrosarcoma, a rare malignant                     Endod. 1998; 85 (3):293-300.
                                                                                    5. Soares R.C., Godoy G.P., Neto J.C., de Souza L. B., Pinto




                                                                                                                        i
               counterpart to these odontogenic benign tumour, that ari-
                                                                                       L. P. Ameloblastic fibro-odontoma: report of a case presenting




                                                                                                                      al
               ses in the jaws either de novo or from preexisting or re-
                                                                                       an unusual clinical course. International journal of Pediatric
               current ameloblastic fibroma. Finally, as AFO sometimes
                                                                                       Otorhinolaryngology Extra 2006;1, 200-203.
               inhibits tooth eruption, the lesion can be microscopical-
                                                                                    6. Friedrich R. E., Siegert J., Donath k., Thorsten Jakel K. Re-
               ly differentiated from the follicular lesions around an im-




                                                                                                                    on
                                                                                       current Ameloblastic Fibro-odontoma in a 10-year-Old Boy.
               pacted teeth in which a proliferation of odontogenic re-                J Oral Maxillofac Surg 2001;59:1362-1366.
               sts can occur. (11)                                                  7. Martin-Granio-Lopez R, Lopez-Garcia-Asenjo J, De-Pedro-
                                                                                       Marina M, Dominguez-Cuadrado L. Odontoameloblatoma:
                                                                                       a case report and a review of the literature. Med Oral.




                                                                                                                  zi
               Conclusions                                                             2004;9:340-4.
                                                                                    8. Chen Y, Li TJ, Gao Y, Yu SF. Ameloblastic fibroma and rela-
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                                                                                                a
               of preservation of the involved teeth. Recurrence or local           9. Nascimento JE, Araujo LJ, Almeida LY, De-Paula AM, Bo-
               invasion is normally not observed if removed along with                 nan PR. Ameloblastic fibro-odontoma: a conservative sur-




                                                                                             rn
               any involved teeth. Malignant transformation of ameloblastic            gical approach. Med Oral Patol Oral Cir Bucal 2009 Dec 1;14
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                                                                                   te
               not justify a radical treatment of all these benign lesions.        11. De Riu G, Meloni SM, Contini M, Tullio A. Ameloblastic fi-
               As noted in the literature review, not all lesions previou-             bro-odontoma. Case report and review of the literature. J Cra-
                                                                                       nio-Maxillo-Facial-Surgery (2010) 38, 141-144.
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                                                                                   12. Navonne R, Mela F., Romagnoli R.,et al. studio clinico pa-
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               intervention. When there is recurrence accompanied by
                                                                         In            tologico di un caso di evoluzione sarcomatosa di fibroma ame-
                                                                                       loblastico. Minerva Stomatologica 1982;31:673, 1982.
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                                                           ni
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                                                                                       Ubergang in ein odontogenes Sarkom. Dtsch Z Mund Kie-
                                                                                       fer Gesichts-Chir 1991;15:90.
               References                                                          15. Howell RM, Burkes EJ. Malignant transformation of amelo-
                                                io


                                                                                       blatic fibro-odontoma to ameloblastic fibrosarcoma. Oral Surg
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                                       iz



                   Oral Surgery 2007;4: 45-50.                                         fibro-odontoma without removal of the associated impacted
                2. Costa Carvalho Silva G., Correia Jham B., Carvalho Silva            permanent tooth: report of cases. J Oral Maxillofac Surg
                   E., Rebello Horta C., Pereira Godinho SH, Santiago Gomez            1992;50: 1094-7.
                      Ed




                   R. Ameloblastic fibro-odontoma, case report. Oral Oncology      17. Philipsen H.P., Reichart P.A., Praetorius F. Mixed odonto-
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                3. Furst I., Pharoah M., Phillips J. “Recurrence of an Amelo-          lationship. Review of the literature and presentation of 134
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               Annali di Stomatologia 2010; I (2): 11-13                                                                                             13
