Amelblastic fibrom

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International Journal of Oral & Maxillofacial Pathology. 2011;2(3):59-63 ISSN 2231 2250Available online at http://www.journalgateway.com or www.ijomp.org

2011 International Journal of Oral and Maxillofacial Pathology. Published by Publishing Division, Celesta Software Private Limited. All Rights Reserved

Case ReportAmeloblastic Fibroma: Report of 3 Cases and Literature ReviewShalini Gupta, Ankita Tandon, Divya Mehrotra, OP Gupta

AbstractAmeloblastic fibroma is a rare odontogenic tumour of the jaw, in which both the epithelial andectomesenchymal elements are neoplastic and is commonly seen in the first 2 decades of life.The common clinical manifestation is a slow-growing swelling. Ameloblastic fibroma is mostcommon in adolescents and young adults, and is generally found in the mandible. We report threecases of Ameloblastic fibroma which had appeared at different anatomic sites and had causedmarked facial disfigurement.The controversy regarding its management needs to be emphasizeddue to the malignant transformation reported.Therefore detailed biological behaviour along withthe current surgical pathologic issues of Ameloblastic fibroma would be discussed in thesubsequent presentation.

Keywords: Jaw Neoplasms; Dental Tissue Neoplasms; Odontogenic tumor; Ameloblastic Fibro-odontoma; Ameloblastic Fibro-dentinoma; Ameloblastic fibroma.

Shalini Gupta, Ankita Tandon, Divya Mehrotra, OP Gupta. Ameloblastic Fibroma: Report of 3 Cases andLiterature Review. International Journal of Oral & Maxillofacial Pathology; 2011:2(3):59-63. International

Journal of Oral and Maxillofacial Pathology. Published by Publishing Division, Celesta Software PrivateLimited. All Rights Reserved.

Received on: 23/07/2011 Accepted on: 12/09/2011

IntroductionAmeloblastic fibroma (AF) is a relativelyuncommon neoplasm of odontogenic origincomprising about 1.5-4.5% of allodontogenic tumors. Since its description byKruse in 1891, there has been much debateand confusion regarding its nature andbiological behavior. In the earlier years of

the last century, AF has been recordedunder different headings, with the mostcommon one being the ameloblastoma(Adamantinoma). In 1946, Thoma andGoldman were the first to classify this tumoras a separate entity.

1AF is defined by WHO

as consisting of odontogenicectomesenchyme resembling the dentalpapilla and epithelial strands and nestsresembling dental lamina and enamel organ.No dental hard tissues are present. If thelesion has dentinoid tissue without or withenamel formation, it could be termed as

ameloblastic-fibrodentinoma (AFD) orameloblastic-fibroodontoma (AFO),respectively. This group of lesions is alsosometimes referred to as mixed odontogenictumors that histologically resemble variousstages of tooth formation.

1

Clinically the tumor grows slowly andpainlessly, expanding the jaw.Radiographically, it appears as aunilocular/multilocular area of radiolucencywith a smooth outline. Histologically, thetumor consists of strands and groups of

epithelial cells in a connective tissuebackground and does not invade bone.

2The

opinion that AF exhibits somewhat slowclinical growth, is well encapsulated andshows an innocuous benign behavior wassupported by most authors, thusconservative treatment in keeping with thebehavior of AF was recommended. On theother hand, some authors believed that AFis more aggressive than it had been thought

and a more radical therapy is needed on thebasis of reviewing recurrent or malignantlytransformed cases in the literature.

1Hence,

three cases of AF are presented throwinglight on the characteristic features as wellthe treatments adopted.

Case reportsCase 1: A 12 year old male patient visitedour outpatient department with chiefcomplaint of difficulty in swallowing andspeech for last 1 year. On intra oralexamination, an ulceroproliferative growth

roughly 2.5 cm in greatest dimensions wasappreciated posterior to maxillary left firstmolar extending to both palatal and buccalsides (Fig 1A). The growth waserythematous, showed irregular surface andappeared to interfere with local functions. Norelevant medical history was associated. Onradiographic examination, a well definedunilocular radiolucency was found encirclingimpacted maxillary left second molar withresorption of roots of maxillary left first molar(Fig 1B). The excision of entirepedunculated mass was performed along

with removal of impacted maxillary left firstmolar.


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Case 2: An 18 year old male patient with thechief complaint of right facial asymmetrysince 2 years visited our outpatientdepartment. On examination, an expansilemass of right mandibular body and ramus(Fig 2A) was noticed with mobility of

associated teeth. Medical and dentalhistories were non contributory. Onradiographic examination, a multilocularradiolucent lesion of right mandibular bodywas appreciated extending across themidline upto left mandibular first pre molar(Fig 2B). The CT findings revealed softheterogenous predominantly hyperdensetissue attenuation extending in body, ramusand condyle of mandible on right sidecausing destruction of the same. The lesioninfiltrated base and lateral border of tongueon right side. The interface of the lesion was

ill defined with masseter muscle (Fig 2C).Mandibular resection, from ipsilateralcondylar neck to contralateral canine, wasperformed with primary reconstruction usingtitanium reconstruction plate. The remainingcondylar stump was fixed with two screws,while three screws were fixed in the bodyregion.

Case 3: A 16 year old male patient visitedour outpatient department with the chiefcomplaint of pain in left lower posterior teethfor last 1.5 years. On examination, impacted

mandibular left second molar wasassociated with a radiolucent lesionencircling the crown of mandibular left thirdmolar and numerous intervening septae (Fig3A). There was extensive cortical plateexpansion. Partial mandibular resection wasperformed with immediate reconstructionusing titanium reconstruction plate andscrews.

Histopathologically, the lesions showedstrands, cords and islands of odontogenicepithelium in a primitive connective tissue

stroma. The epithelial islands and cordswere characterized by peripheralcolumnar/cuboidal hyperchromatic cells andwere frequently only two cell layers thick.The mesenchymal component consisted ofevenly distributed plump ovoid and stellatecells in a loose myxoid to predominantlyeosinophilic matrix resembling the primitivedental papilla. Mitosis was not a feature. Nohard tissue structures were detected (Fig 2D& 3B). The patients were under regularfollow up protocol to keep a check onrecurrences and the results have been

functionally and esthetically pleasant.

DiscussionAmeloblastic fibroma is a rare mixedodontogenic tumor that usually occurs inyoung patients, the youngest patientreported is a 7 week old infant.

3It has been

diagnosed at a mean age of 15 years3

with

males being more affected than females.4,5

Itcan appear either in the maxilla or mandible,with the posterior region of the mandible asits most common anatomic site.

6,5Our cases

presented clinically in both the jaws. Thepatients usually present with a hard swelling,but intraoral ulceration, pain, tenderness, ordrainage may also be observed.

7The lesion

may affect the normal eruption of teeth in thearea. An impacted tooth may be associatedwith the tumor in approximately threequarters of the cases

5and the finding was

concomitant in our cases.

The biological behaviour of AF shares thenature between hamartoma and trueneoplasm.

7Cahn and Blum postulated that

an AF could develop eventually into anodontoma if the lesion had been allowed toremain. They believed that the onlydifference between the AF and theodontoma is the patients age and thedegree of maturation of the lesions. Thiswould imply that all AF, AFD and AFOmerely represent various stages of the samelesion, and will mature over time resulting in

ultimately the formation of an odontoma.However, this continuum concept has notbeen widely accepted

1as recent evidence

suggests that AF exhibits a high recurrentrate and, in some cases, malignanttransformation.

7We have followed our cases

for almost 2 years and have found norecurrence.

Radiographically, as in our casesameloblastic fibromas are unilocular lesions,occasionally multilocular when larger, withsmooth well-demarcated borders. These

lesions are frequently associated withunerupted teeth, so may initially beinterpreted as dentigerous cysts.

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Microscopically, AF comprises strands andislands of an odontogenic epithelium in aloose and primitive connective tissuestroma, characteristic of dental papilla(embryonic dental pulp). The odontogenicepithelial cells are similar to those ofameloblastoma. Tiny islands resembling thefollicular stage of the developing enamelorgan may be observed. Some recurrentcases developed dentin formation with or

without enamel structures, and subsequentlydifferentiate over time into odontoma.

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Mitoses should not be a feature ofameloblastic fibroma. The presence ofmitosis should expand the differentialdiagnosis to include malignant entities, toinclude ameloblastic fibrosarcoma.

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Figure 1A: Clinical photograph showing

ulceroproliferative growth

Figure 1B: Orthopantomograph showingunilocular radiolucency encircling impacted

maxillary left second molar.

Figure 2A: Clinical photograph showingexpansile mass related to right mandibularbody.

Figure 2B: Orthopantomograph showingextensive radiolucency.

Figure 2C: CT scan showing an expansilegrowth involving the right mandibular area.

Figure 3A: Orthopantomograph showingunilocular radiolucency encirclingmandibular left third molar crown.

Figure 2D: Photomicrograph showing cordsof odontogenic epithelium in a primitiveconnective tissue stroma.

Figure 3B: The Photomicrograph showsepithelium in the primitive ectomesenchyme.


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In cases undergoing malignanttransformation, there are unequivocalchanges in the mesenchymal component,and the odontogenic epithelium iscompletely disappeared as malignanttransformation of AF was found to be

associated with oncogenic aberrations intumour-related genes. Mesenchymalproliferation within the tumour resulting in aloss of an epithelial component, is a usualpresentation of sarcomatous changes ofAF.

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Treatment for ameloblastic fibroma isdescribed as conservative excision.Philipsen et al. proposed that the innocuousbehavior of the lesion does not justifyaggressive initial treatment but rathermeticulous surgical enucleation with close

clinical follow-up.3

In general, a conservativeapproach, such as enucleation withcurettage of the surrounding bone, shouldbe applied for young patients. Theneoplastic nature of AF is often suggestedby the fact that some of these tumors couldrecur following surgery and that malignanttransformation from a pre-existing AF hasbeen reported.

8While uncommon, the

possibility of malignant transformation ofameloblastic fibroma into ameloblasticfibrosarcoma is well documented

7and the

recurrence rate of AF found by Trodahl et al.

was 43.5%; on the other hand by Zallen etal. it was 18.3% after reviewing the literaturewith 85 cases of AF.

8An aggressive surgical

treatment is therefore suggested by someauthors because of the possibility ofmalignant transformation of an Ameloblasticfibroma to an ameloblastic fibrosarcoma. Nomatter what the reason of recurrence is, along term follow-up is necessary.

8,9

Various immunohistochemical markers ofintra and extracellular proteins have alsobeen studied in AF and other benign

odontogenic mixed tumors.10 In AmeloblasticFibroma, odontogenic epithelial cells arefully positive for cytokeratin detected byantibody KL-1.On the other hand, onlyimmature dental papilla-like mesenchymaltissue, especially around the dental laminalike odontogenic epithelium, are positive fortenascin. Positive vimentin staining can beobserved in some areas of immature dentalpapilla like cells as well as the basementmembrane of odontogenic epithelium. Thesefindings are very well suggestive of the factthat Ameloblastic fibroma develops at an

early stage of tooth formation.1 Someauthors suggested the use of proliferating

indices, such as AgNOR, PCNA and Ki-67,to institute an appropriate treatment for eachpatient, and possibly, to suggest themalignant development.

7Among these,

PCNA is a nuclear protein which isassociated with the S phase of DNA

synthesis in association with cellproliferation. MIB-1 reacts with Ki-67 nuclearantigen associated with cell proliferationand, has been found throughout the cellcycle excepting for G0phase.

1

ConclusionEvaluation of the growth potential in AF andrelated lesions could therefore be of help inunderstanding tumor aggressiveness and inselecting appropriate surgical procedures.

Author Affiliations

1. Dr. Shalini Gupta, Professor & Head, 2. Dr.Ankita Tandon, Senior Resident, Department ofOral & Maxillofacial Pathology, CSMMU,Lucknow, 3. Dr. Divya Mehrotra, Professor,Department of Oral & Maxillofacial Surgery,CSMMU, Lucknow 4. Dr. OP Gupta, Practicinglaproscopic surgeon, Lucknow, India.

AcknowledgementWe would like to thank all the staff members fromthe Department of Oral and MaxillofacialPathology.

References

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3) Abughazaleh K, Andrus KM, KatsnelsonA, White DK. Peripheral ameloblasticfibroma of the maxilla: report of a caseand review of the literature. Oral SurgOral Med Oral Pathol Oral Radiol Endod

2008;105:e46-8.4) McGuinness NJ, Faughnan T, Bennani

F, Connolly CE. Ameloblastic fibroma ofanterior maxilla presenting as acomplication of tooth eruption: A casereport. J Orthod 2001;28(2):115-7.

5) Arnnop PP, Chaine A, Dhanuthai K,Bertrand JC, Bertolus C. ExtensiveAmeloblastic Fibroma in an AdolescentPatient: A Case Report with a Follow-Upof 4 Years. Eur J Dent 2009;3:224-8.

6) Chen Y, Wang JM, Li TJ. Ameloblasticfibroma: A review of published studies

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7) Oghli AA, Scuto I, Ziegler C,Flechtenmacher C, Hofele C. A largeameloblastic fibro-odontoma of the rightmandible. Med Oral Pathol Oral Cir

Bucal 2007;12:E34-7.8) Boi M, Hren NI. Ameloblastic fibroma.

Radiol Oncol 2006;40(1):35-8.9) Nelson BL, Folk GS. Ameloblastic

Fibroma. Head Neck Pathol 2009;3:513.

10) Chen Y, Li TJ, Gao Y, Yu SF.Ameloblastic fibroma and related

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Corresponding Author

Dr. Shalini Gupta,Head of Department,Oral & Maxillofacial Pathology,C.S.M.M.U, Lucknow, India.E mail ID: [email protected] number: 09453556510

Source of Support: Nil, Conflict of Interest: None Declared.

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