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Official Publication of Asociacin Ayuda Enfermo Neuroquirufgico

Journal of
Cranio-Maxillary
Diseases
Volume 2 Issue 1 J anuary-J une 2013
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ISSN: 2278-9588
Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013 80
An aggressive ameloblastic fibroma in a
9-year-old child treated with buccal fat graft
Amina Sultan, Dayashankara Rao JK
1
, Harsha Jain
2
Department of Pediatric and Preventive Dentistry, Faculty of Dentistry, Jamia Millia Islamia, New Delhi,
1
Department of
Oral and Maxillo Facial Dentistry, S.G.T. Dental College and Hospital, Gurgaon, Haryana,
2
Department of Oral and Maxillo
Facial Surgery, Sharda Dental College, Greater Noida, Utar Pradesh, India
ABSTRACT
Ameloblastic fbroma is a relatively uncommon neoplasm of odontogenic origin which is characterized by
simultaneous proliferation of both epithelial and mesenchymal tissue without the formation of enamel and dentin.
It is a slow-growing tumor and commonly located in mandibular molar area, often over an unerupted tooth. It is
usually diagnosed in the second decade of life. Its occurrence in posterior maxilla is a rare or uncommon entity
and only few cases have been reported so far in children. The management of such lesions in maxilla is to avoid
any disfgurement and at the same time not compromising its complete removal as it has tendency to recur and
possibility of malignant change into ameloblastic fbrosarcoma. This report is a documentation of a case, of a
9yearold female child with an aggressive ameloblastic fbroma of posterior region of maxilla. The patient has
been followed up for 2 years; the outcome has been functionally and aesthetically satisfactory and there have been
no signs of recurrence.
Keywords: Ameloblastic fibroma, buccal fat pad graft, surgical excision
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DOI:
10.4103/2278-9588.113583
Correspondence to:
Dr. Dayashankara Rao JK, Department of Oral and Maxillo Facial Dentistry, S.G.T. Dental College and Hospital, Farukh Nagar Road, Budhera,
Gurgaon, Haryana, India. E-mail: dsraojk@yahoo.com
in association with an unerupted tooth or tooth
migration or displacement.
[2-5]
It will frequently cause no complaint but pain
tenderness or mild swelling of the jaw may induce
the patient to seek aid from the dentist.
[1]
Nevertheless its importance lies in its tendency to
recur and in the possibility of malignant change into
ameloblasticfibrosarcoma.
[7]
This report is a documentation of a case of a
9-year-old female child with clinically aggressive
AF with an atypical localization (posterior region of
maxilla) treated with surgical excision followed by
closure of the defect with the buccal fat pad.
CASE REPORT
A 9-year-old girl child reported to the department
of Pedodontics, SGT Dental college, Gurgaon,
INTRODUCTION
The ameloblastic fibroma (AF) is a relatively
uncommon neoplasm of odontogenic origin, which
is characterized by simultaneous proliferation of
both epithelial and mesenchymal tissue without the
formation of enamel and dentin.
[1,2]
AF tends to occur in younger patients. Most
lesions are diagnosed within first two decades of life.
This tumor is slightly more common in males than in
females.
[1,3-6]
The most common site of occurrence is
the posterior site of mandible.
[1-3]
It is a slow-growing
tumor, often asymptomatic and usually occurs
Case Report
Sultan, et al.: Ameloblastic fbroma, buccal fat graft
81 Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013
with a complaint of a painless swelling on the right
side of maxilla for the last2 years. The patient gave
a history of a small nodule, on facial aspect of
gingiva in posterior region that gradually increased
up to the present size of 5-6 cm over a period of
2 years. Intraoral examination presented a soft tissue
lesion in the right maxillary posterior alveolar region
extending from deciduous canine region to soft
palate posteriorly and toward mid palatine area and
also extraorally. The swelling was large, lobulated,
nontender, and pedunculated and 6 2 cm in size.
It was irregular in shape and soft in consistency.
The overlying mucosa was normal and did not
bleed on probing. No discharge or redness was
present [Figure 1].
Extraoral examination revealed facial asymmetry
with an edematous swelling on right side of cheek
extending from maxillary right permanent canine to
first permanent molar. Skin color was normal. She
had no other symptoms and apparently in good
health [Figure 2].
Patient gave a history of past treatment involving
an incisional biopsy of that area; about 1 year back,
at some local hospital along with the medical line of
treatment (antibiotics).But the swelling increased in
size considerably without any response to the given
treatment.
A panaromic radiograph Orthopantamogram
(OPG) showed a wellcircumscribed unilocular
radiolucent lesion in right maxillary posterior region
[Figure 3]. The radiolucency measured 4 2.5 cm
and it was associated with displacement of second
premolar, first and second permanent molars toward
the periphery of the lesion (maxillary sinus).
Erupted teeth were not mobile and there was no
sign of root resorption.
Based on clinical and radiographic findings, a
presumptive preoperative diagnosis of dentigerous
cyst and ameloblastic fibrosarcoma, was done.
Under naso endotracheal general anesthesia
the complete lesion was excised and curetted via
intraoral approach. The cut surface was grey white
in appearance. Through horizontal incision along
the buccal mucosa the buccal fat pad was released
and spread over the bony defect keeping its pedicle
intact [Figure 4]. Primary closure of the oral mucosa
along with the buccal fat graft was done by using
3-0 vicryl [Figure 5]. Right side deciduous canine and
lateral incisor were extracted prior to the excisional
biopsy while first and second permanent molars were
left untouched. After the whole lesion was removed
Figure 1: Intraoral picture showing the extension of the
lesion crossing the midline of palate
Figure 2: Extraoral swelling with the diffuse swelling over
the right side of the face
Figure 3: OPG radiograph showing soft tissue density
lesion in the right maxilla
Sultan, et al.: Ameloblastic fbroma, buccal fat graft
Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013 82
the resultant bony cavity was smooth and had an
intact surface. The excised tissue mass was send for
biopsy.
The specimens sent for the pathological
examination were in two pieces from different sites
of the lesion. The tissues taken were 4 2 1 cm
and 2 1 6 cm in sizes.
The diagnosis of AFwas confirmed according to
the histological findings [Figures 6 and 7].
The postoperative healing was uneventful and
there was no incidence of any wound break down.
There was no fistula formation. On follow-up
examinations every month, there has been no
evidence of recurrence after surgery [Figure 8].
Both the premolars have erupted into the oral
cavity. The postoperative OPG taken after 3 months
showed occlusal movement of the permanent
molars and eruptive movement of permanent lateral
incisor [Figure 9].
DISCUSSION
Ameloblastic fibroma is a true histological
biphasic tumor because the epithelium and
mesenchymal component are part of neoplastic
process. The two tissue components represent an
early stage of odontogenesis before the formation
of calcified structures of enamel and dentin.
[8]
Figure 4: Complete surgical enucleation of the lesion and
exposure of the buccal fat Figure 5: Primary closure of the huge defect with buccal
fat graft
Figure 6: H and E staining, A: Section showing odontogenic
epithelium in form of cords and small islands with loosely
arranged epithelial cells resembling stellate reticulum in the
center and stellate cells in loose matrix 10 ( 4 magnifcation)
Figure 7: Odontogenic epithelium in form of islands
with juxtra epithelial hyalinization of mesenchymal
portion.10 10 magnifcation
Sultan, et al.: Ameloblastic fbroma, buccal fat graft
83 Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013
According to the revised WHO classification AF
consists of odontogenic ectomesenchyme resembling
the dental papilla and epithelial strands and nests
resembling dentallamina and enamel organ. No
dental hard tissues are present. Rarely, tumors with the
histo morphology of AF may form dysplastic dentin,
and are called ameloblastic fibrodentinomas (AFD).
[9]
In the present case, the AF was diagnosed to be
present in 9-year-old female child in the posterior
region of maxilla. Ameloblastic fibromatends to
occur in younger patient, with average age of patient
being 14.6 years and with 40% of the patients under
the age of 10 years.
[10]
The age limits are widespread
i.e., from6 months to 42 years.
[10,11]
Also the sex
predication varies from no preference
[7,12]
to males
are more frequently affected than females.
[5,10,11,13]

Over 80% of the tumors occur in mandible, the usual
site being the canine-molar region. Only four cases
of tumors in maxillary anterior region have been
reported.
[11,14]
Ameloblastic fibroma exhibits somewhat slower
clinical growth than simple ameloblastoma and does
not tend to infiltrate among trabeculae of bone.
Instead it enlarges by gradual expansion so that
periphery of the lesion often remains smooth.
[1,14]
The
lesion may displace erupted teeth although the teeth
may remain vital.
[15]
In our patient, however, rapid growth was
observed, probably due to small size of maxilla
rather than the biological behavior of tumor.
Most patients present with a painless, slow-growing
and expanding tumor commonly associated with
an unerupted tooth. Often the associated tooth is
displaced in an apical direction indicating that the
origin of lesion is superior to the tooth.
[16]
The patient,
in this case also reported with an asymptomatic, rapidly
expanding swelling on right side of maxilla in posterior
region. Second premolar and first permanent molar
were unerupted, while first premolar was partially
erupted. Deciduous right lateral incisor was retained.
The childs medical history was unremarkable.
No constant significant differences between the
appearance of the simple ameloblastoma and that
of the AF are found radiographically.
[1]
The tumor
appears as a well-defined radiolucent image,
usually unilocular, as in our case, which can be
difficult to distinguish from other odontogenic or
non-odontogenic fibroma, odontogenic myxoma,
fibrous dysplasia in the osteolytic stage and central
giant cell granuloma. The presence of dental
inclusions may give a false appearance of follicular
cyst; and resorption of tooth roots is very rare.
[7]
On
the basis of review by Trodahl, the radiographic
lesion varied in size from 1 to 8.5cm in diameter.
Most of the lesion (3:1) were multilocular; only the
smaller ones were unilocular.
[11]
The OPG in this case
revealed a large expansile- 4 2.5 cm well-corticated
unilocular radiolucent lesion located in region of
posterior maxilla. The lesion had displaced first and
second permanent molars into the maxillary sinus.
Microscopically AF is composed of young
cellular mesenchymal tissue that resembles or
duplicates the dental papilla of tooth germ. Within
this young mesenchyme are thin strands and islands
of odontogenic epithelium.
[6]
Analysis of sections of
the lesion taken from our patient showed fragments
Figure 8: Three months post-op picture showing good
healing with out any fstula
Figure 9: Three months post-op OPG with out any evidence
of the disease
Sultan, et al.: Ameloblastic fbroma, buccal fat graft
Journal of Cranio-Maxillary Diseases / Vol 2 / Issue 1 / January 2013 84
of tumor tissue composed of both mesenchymal
and epithelial components. Epithelial cells were
arranged in cords, nests, trabeculae, and strands
of cuboidal to low columnar cells. No evidence of
cellular atypia was seen.
All these histological findings suggested that it
was a true mixed tumor.
Proper management of AF has been a recent topic
of debate. Although, initially it was believed that AF
was innocuous lesion that seldom recurred after
simple local excision or curettage, but subsequent
reports seemed to indicate a substantial risk of
recurrence after conservative therapy.
[3]
As the lesion
is well-circumscribed recurrence are considered to
be the result of inadequate initial removal of what
are frequently multilocular lesions.
[8]
We opted for
excision and curettage. A wide excision of the tumor
has been recommended unless the extent of surgery
would result in significant cosmetic deformity.
[7]

Other support enucleation or curettage as the initial
treatment with a modified block resection reserved
for recurrence.
[11]
In the present case the tumor
was massive measuring 5-6 cm. The location
was posterior maxilla, in contrast to its usual site
i.e. posterior mandible. It was intra osseous having
perforated the buccal cortical plate to present as
a soft tissue lesion, an indication of its aggressive
nature. First permanent molar was left untouched.
Clinically, eruption of unerupted and malpositioned
teeth should be evaluated carefully. In this case also,
both the premolars and permanent lateral incisor
erupted spontaneously while permanent molars
showed some drifting movement occlusally.
Although rare, malignant transformation can
occur and long-term follow-ups is recommended.
Regardless of the form of treatment, patient
with AF must be followed up for longer period to
enable the early detection of possible recurrence or
development of ameloblastic fibrosarcoma.
[17]
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How to cite this article: Sultan A, Rao JD, Jain H. An aggressive
ameloblastic fbroma in a 9-year-old child treated with buccal fat graft. J
Cranio Max Dis 2013;2:80-4.
Source of Support: Nil. Confict of Interest: None declared.
Submission: May 04, 2012, Acceptance: December 11, 2012

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