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Ameloblastic Fibroma of the Maxilla – A Rare Presentation

A.V.S. Hanumanth Rao1, Anvita Panga2, G.Madhusudhan Reddy3, B.Vijay Kumar3

term follow-up of ameloblastic fibroma is recommended.4 This

ABSTRACT report describes an interesting case of ameloblastic fibroma that
Introduction: Ameloblastic fibroma is a rare tumour which occurs affected the maxilla of a young girl with cheek swelling.
predominantly in children. It usually arises from the mandibular
CASE REPORT
dentition although it can arise in maxilla. It is a benign tumor of
mixed odontogenic origin. The common clinical manifestation is A 12-year-old female patient reported to the Department of
a slow-growing swelling and noneruption of teeth. ENT with the complaint of a painless swelling of right cheek
Case report: We present a case of ameloblastic fibroma of since 20 days associated with epiphora of the right eye since
the maxilla in a 12 year old girl who presented to OPD with 20 days. External examination showed a diffuse swelling of
facial swelling. After detailed examination and investigations the right maxilla. Intra-orally, a firm, non-tender swelling was
we diagnosed the case as a dentigerous cyst and the patient
noted in the buccal vestibule extending from the region of the
underwent enucleation through Caldwell Luc approach under
right maxillary central incisor to the first premolar. Swelling
general anaesthesia. Histopathological examination revealed
ameloblastic fibroma. The patient is under long term follow-up
also extended over the corresponding palatal surface. The right
with no signs of recurrence. canine tooth was missing (Figure-2). The remaining ENT and
Conclusion: This case emphasizes the importance of careful general examination was normal.
differential diagnosis in ENT, while reporting a rare lesion and its CT scan was in favour of mixed dense soft tissue lesion showing
atypical location. cystic and solid components seen in the right maxillary sinus.
Mild enhancement of solid components. Expansion of the sinus
Keywords: Odontogenic tumor, Ameloblastic fibroma, Maxilla, seen with thinning of walls. Tooth like structure seen in the
Dentigerous cyst superior part of the lesion. No obvious intra-orbital extension
was seen. Possibilities included: Dentigerous cyst, Fungal
granulomatous lesion and Retention cyst (Figure-1).
INTRODUCTION With these data a provisional diagnosis of dentigerous cyst was
Ameloblastic fibroma is a true mixed odontogenic tumour made. After surgical profile and pre anaesthetic check-up, tumor
involving both epithelial and mesenchymal tissues. The enucleation along with impacted tooth through Caldwell Luc
tumor is of odontogenic origin and its occurrence is quiet rare approach under general anaesthesia was planned. After mucosal
accounting to only 2.5% of all tumors of this category.1 It occurs incision antero-inferior wall of maxillary sinus was found
predominantly in children and therefore remains an important eroded by the lesion (Figure-2). After enucleation and proper
diagnostic consideration.2 It usually arises from the mandibular hemostasis, the wound was closed by 3/0 catgut. The specimen
dentition although it can arise in maxilla.1 It is characterized was subjected to histopathological examination (Figure-3).
by simultaneous neoplastic proliferation of mesenchymal On histopathological examination, the specimen revealed
and epithelial components, with no formation of hard dental islands of epithelial cells in ectomesenchyme resembling the
tissues.3 Knowledge of the malignant potential of ameloblastic dental papilla. The islands consist of a peripheral columnar
fibroma should assist in determining the management of these ameloblastic cells with reverse polarity in fibromyxoid
benign tumours, and may prevent malignant transformation to stoma. Some of the areas show a zone of hyalinization and
ameloblastic fibrosarcoma.4 formation of dentinoid at the periphery (Figure-4). Based on the
Patients with ameloblastic fibroma of the maxilla usually present histopathological examination, a final diagnosis ameloblastic
with intraoral findings including a mass on the anterior upper fibroma was arrived at. Post operative healing was good and
jaw, swelling of the alveolar process, and non-eruption of teeth. no sign of recurrence of AF was recorded during the follow-up
Microscopically, the lesion shows cords and islands of period.
neoplastic odontogenic epithelium interspersed by neoplastic DISCUSSION
mesenchymal tissue that resembles the dental papilla. If lesions
composed of similar elements are found with deposition of Mixed odontogenic tumors comprise a rare group of lesions
dentin then they are termed as ameloblastic fibrodentinoma and with diverse histopathological types and clinical behaviour.
if dentin and enamel both are found then it is called ameloblastic
fibroodontoma.5 Professor, 2Senior Resident, 3Assistant Professor, Department of ENT,
1

Radiographically, ameloblastic fibroma appears as a well- Kakatiya Medical College, Warangal, Telangana, India
defined, unilocular or multilocular radiolucent lesion, with
sclerotic radiopaque margins. Ameloblastic fibroma may be Corresponding author: Dr. Anvita Panga, 11-4-105, Srihari Nagar,
initially identified in routine radiographic examination.6 Moosapet, Hyderabad, Telangana, India
The most appropriate treatment method for ameloblastic
How to cite this article: A.V.S. Hanumanth Rao, Anvita Panga, G.
fibroma is still uncertain. A conservative approach is suggested
Madhusudhan Reddy, B.Vijay Kumar. Ameloblastic fibroma of the
by several authors.7-10 However, tumors may recur following maxilla – a rare presentation. International Journal of Contemporary
surgical removal and progress to malignancy.4,6,7 Thus, long- Medical Research 2016;3(7):1882-1883.

1882
International Journal of Contemporary Medical Research
Volume 3 | Issue 7 | July 2016 | ICV: 50.43 | ISSN (Online): 2393-915X; (Print): 2454-7379
Rao, et al. Ameloblastic Fibroma of the Maxilla

children and adolescence. After proper clinical, radiological and

histopathological examination, conservative surgery is enough
in most of the cases of AF. Patients must be kept under long
term follow-up and special care should be taken for recurring
tumors. The present case emphasizes the importance of careful
differential diagnosis in ENT, while reporting a rare lesion and
its atypical location.
REFERENCES
1. Pereiraa K, Bennetta K, Elkinsa T P, Qub Z. Ameloblastic
fibroma of the maxillary sinus. IntJ PedOtorhinolaryngol.
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tooth and Caldwell Luc approach 2. Jones AV, Franklin CD. An analysis of oral and maxillofacial
pathology found in children over a 30-year period. Int J
Paediatr Dent. 2006;16:18-30.
3. Cohen DM, Bhattacharyya I. Ameloblastic fibroma,
ameloblastic fibro- odontoma, and odontoma. Oral
MaxillofacSurgClin North Am. 2004;16:375-84.
4. Kobayashi K, Murakami R, Fujii T, Hirano A. Malignant
transformation of ameloblastic fibroma to ameloblastic
fibrosarcoma: case report and review of the literature. J
CraniomaxillofacSurg. 2005; 33: 352-5.
5. Kramer R H, Pindborg J, Shear M. The WHO Histological
Figure-3: Cut section of the specimen showing the tooth; Figure-4:
Typing of Odontogenic Tumours. CANCER 1882,70(11).
Histopathological slide
6. Chen Y, Wang JM, Li TJ. Ameloblastic fibroma: A review
of published studies with special reference to its nature and
There is still confusion regarding the nature and relationship of
biological behavior. Oral Oncol. 2007;43:860-8.
these tumors and other related lesions in the existing literature. 7. Kousar A, Hosein MM, Ahmed Z, Minhas K. Rapid
Mixed odontogenic tumors generally comprise of Ameloblastic sarcomatous transformation of an ameloblastic fibroma of
fibroma (AF), Ameloblastic fibrodentinoma and ameloblastic the mandible: Case report and literature review. Oral Surg
fibro-odontoma. Oral Med Oral Pathol Oral Radiolendod. 2008;98;e80-e85.
AF has both epithelial and mesenchymal neoplastic 8. Barnes l, eveson JW, Reichart PA, Sidransky P (editors).
proliferation.8 This tumor is usually diagnosed between the first Pathology and genetics of tumours of the head and neck:
and second decades of life and the majority of cases of occur World Health Organization classification of tumours:
International Histological Classification of Tumors. 3rd ed.
in the mandible.6,9 A few cases of the AF have been reported in
lyon: IARC Press, 2005.
the maxilla.9 The clinical manifestations of AF are not specific 9. Dimitrakopoulos I, Psomaderis K, Zaramboukas T.
and the lesion is frequently discovered as an incident finding Ameloblastic fibroma of the mandible associated with root
in a routine radiographic examination, such as cysts and other resorption and unerupted teeth: a case report. Quintessence
odontogenic tumors.8 Radiographically, ameloblastic fibroma Int .2008;38:523-527.
must be differentiated from other similar lesions such as 10. Kim SG, Jang HS. Ameloblastic fibroma: report of a case.
ameloblastoma, dentigerous cysts, odontogenic keratocysts and J Oral MaxillofacSurg. 2002;60:216-218.
fibrosarcoma ameloblastic.4,10
While uncommon, the possibility of malignant transformation Source of Support: Nil; Conflict of Interest: None
of AF into ameloblastic fibrosarcoma has been reported by
Submitted: 13-05-2016; Published online: 13-06-2016
Chen etal.6 and Kobayashi et al.4 In those studies malignant
change only occurred in the second recurrence. But according
to Kousar et al.7 rapid sarcomatous transformation of an AF
occurred within 6 months. All authors agree that a long term
follow-up is necessary.
Ameloblastic fibroma should generally be treated with
conservative surgery. Only larger lesions or recurrent lesions
should be treated with radical surgery. Recurrence rate is
estimated to be around 33.3%, but there is a lower incidence
among younger patients.6 Therefore enucleation via Caldwell
Luc approach was considered sufficient in this patient, due to
her young age.
CONCLUSION
Ameloblastic fibroma is a rare tumor and has a good prog­
nosis. AF and other related lesions should be kept in mind
while diagnosing the swellings of the maxillofacial region in

International Journal of Contemporary Medical Research 1883

ISSN (Online): 2393-915X; (Print): 2454-7379 | ICV: 50.43 | Volume 3 | Issue 7 | July 2016