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Case Report

Ameloblastoma of the
acanthomatous and plexiform type
in the mandible presenting as a
unilocular radiolucency
Nigel R Figueiredo, Manoj Meena, Ajit D Dinkar, Manisha Khorate
Department of Oral Medicine and Radiology, Goa Dental College and Hospital, Bambolim, Goa, India
ABSTRACT

The ameloblastoma is an odontogenic tumor of epithelial origin, which is persistent and


locally invasive, and has aggressive but benign growth characteristics. There are three
major clinicoradiographic types: Conventional solid/multicystic intraosseous, unicystic and
peripheral ameloblastoma, with the conventional solid intraosseous type being the most
common. Histopathologically, it occurs in six patterns: Plexiform, follicular, acanthomatous,
granular cell, basal cell, and desmoplastic types. This report describes a case of an
ameloblastoma in the angle and ramus region of the mandible, which radiographically appeared
as a unilocular radiolucency mimicking a dentigerous cyst or unicystic ameloblastoma, but
showed features of both the acanthomatous and plexiform patterns of a conventional/solid
ameloblastoma on histopathological analysis.
Key words: Acanthomatous, ameloblastoma, plexiform, unilocular

Introduction

Address for correspondence:


Dr.Nigel R Figueiredo,
House No.685, Santerxette, Aldona,
Bardez403508, Goa, India.
Email:nigel_06@yahoo.co.in
Date of Submission: 11-02-2014
Date of Acceptance: 10-03-2015

Access this article online


Website:
www.indjos.com
DOI:
10.4103/0976-6944.154608
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The ameloblastoma is a benign odontogenic


tumor of epithelial origin, which is thought
to represent 10% of all tumors of the jaw
bones.[1] The name ameloblastoma implies
a resemblance to cells of the enamelforming
organ.[2] This lesion was first designated as
an Adamantinoma by Malassez in 1885,
while the term ameloblastoma was coined
by Ivy and Churchill in 1930.[3]
There are three different clinicoradiographic
types: The conventional solid/multicystic
intraosseous ameloblastoma, the unicystic
ameloblastoma and the peripheral
ameloblastoma. In addition, the desmoplastic
ameloblastoma is regarded as a fourth
subtype of ameloblastoma because of its
mixed radiolucentradiopaque radiographic
appearance (resembling a fibroosseous
lesion) and unique histology.[4] These tumor
types differ in biological behavior and rate
of recurrence. Therefore, each type of
ameloblastoma requires different forms
of treatment.[5]

Histologically, ameloblastomas are


classified into follicular, plexifor m,
acanthomatous, granular cell, desmoplastic
and basal cell types, with the follicular
and plexiform patterns being the most
common.[5] Other rare histologic types
mentioned include the papilliferous, clear
cell and keratoameloblastoma. These
variants may exist singly or as a combination
of two or more types.[6] However, there
is no relationship between the individual
patterns and the behavior of the tumor or
its prognosis.[4]
This paper reports a distinctive case of an
ameloblastoma presenting as a unilocular
radiolucency in the angle and ramus region
on the right side of the mandible in a
20yearold female patient, which showed
histopathological features of both the
acanthomatous and plexiform patterns.

Case Report
A 20yearold female patient reported to
our outpatient department with a chief
complaint of swelling in the lower right
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Figueiredo, etal.: Ameloblastoma of the acanthomatous and plexiform type in the mandible

posterior region since 2months. The patient gave a history


of progressive increase in the size of the swelling with
occasional pain in the area. On extraoral examination,
a diffuse swelling was seen over the right side of the
face, over the posterior body and angle of the mandible,
extending superiorly to involve the ramus and inferiorly
to the posterior submandibular region, measuring around
54cm[Figure1].
The overlying skin was normal with no evidence of
any discharge. On palpation, the swelling was firm in
consistency, tender, nonpulsatile and noncompressible,
with no local rise in temperature. Intraoral examination
revealed a diffuse swelling in the right retromolar region
and buccal vestibule in region of 4748[Figure2]. Asingle,
subcentimetric, firm and mobile right submandibular lymph
node was palpable.

Figure 1: Extra-oral view showing a diffuse swelling over the right


side of the face

Panoramic radiography showed a large, welldefined


unilocular radiolucency with thin corticated borders in the
right angle of mandible, extending superiorly to involve
the entire ramus and anteriorly up to the 47 region. The
internal structure was completely radiolucent. An impacted
developing 48 was seen, which was displaced inferiorly to
the angle of mandible[Figure3].
An incisional biopsy was carried out, which showed strands
of odontogenic epithelium proliferating irregularly in a
sparse connective tissue stroma. Acystic lining was seen,
which was flattened in one area and 710 layers thick in
other areas. There was palisading of basal cells with reverse
polarity, hyperchromasia, and presence of stellatereticulum
like cells. The findings of the incisional biopsy, correlated
with the clinicoradiographic findings, were suggestive
of a dentigerous cyst with secondary changes of an
ameloblastoma.
Based on the findings of the incisional biopsy, the
lesion was treated by surgical resection (partial right
mandibulectomy) with reconstruction using free fibular
graft. Histopathological examination of the resected area
showed an overlying stratified squamous parakeratinized
epithelium with an underlying connective tissue stroma
containing islands and cords of proliferating odontogenic
epithelium, lined by a single layer of tall columnar cells
with hyperchromatic nuclei resembling ameloblasts,
enclosing central stellatereticulum like cells. Parts of the
stroma showed a fingerlike or latticelike arrangement
with minimal stellate reticulumlike component
(Plexiform pattern) [Figure 4]. Other areas of the stroma
showed evidence of squamous metaplasia and keratin
pearl formation (Acanthomatous pattern) [Figure 5]. Based
on the histopathologic findings, a final diagnosis of an

Indian Journal of Oral Sciences Vol. 6 Issue 1 Jan-Apr 2015

Figure 2: Intra-oral view showing a swelling over the right


retromolar area

Figure 3: Orthopantomograph showing a large unilocular


radiolucency in association with an impacted 48

ameloblastoma with both acanthomatous and plexiform


patterns was made.
Postoperative healing was uneventful and the patient has
been under regular followup for the last 2years, during

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Figueiredo, etal.: Ameloblastoma of the acanthomatous and plexiform type in the mandible

Figure 4: Histopathology (H and E stain at 100 magnification)


showing features of the plexiform pattern of ameloblastoma

Figure 5: Histopathology (H and E stain at 100 magnification)


showing features of the acanthomatous pattern of ameloblastoma

which time no evidence of complication or recurrence


has been noted.

incidentally on radiographic studies in asymptomatic


patients.

Discussion

The radiographic appearance of ameloblastomas varies


from unilocular (cystlike) to multilocular (presence
of bony septae which create internal compartments),
with welldefined sclerotic margins which may appear
scalloped or may expand the cortical plate.[7] While
conventional intraosseous ameloblastomas commonly
present as expansile multilocular radiolucencies, the
unicystic ameloblastoma usually occurs as a unilocular
radiolucency and resembles a cystic lesion both clinically
and radiographically. [5,8] However, not all unilocular
ameloblastomas are examples of unicystic ameloblastoma.
The conventional intraosseous ameloblastoma may be
unilocular, but it does not exhibit the fibrous capsule
of the unicystic variant, and does not appear to have
arisen in a cyst.[9] The tumor may be associated with an
unerupted tooth, which may be displaced and resorption
of the roots of adjacent teeth is common.[4] Lesions with
unilocular presentation are thought to be more significantly
associated with unerupted teeth.[8] Our case presented
as a large unilocular radiolucency in association with an
impacted mandibular third molar which was displaced
inferiorly to the angle of mandible region, and thus showed
clinicoradiographic features suggestive of a dentigerous
cyst or unicystic ameloblastoma.

The ameloblastoma is a true neoplasm of odontogenic


epithelium, which is persistent and locally invasive, and has
aggressive but benign growth characteristics. It is believed
to arise from the remnants of the dental lamina, cells of
the developing enamel organ, or can sometimes arise as
a result of neoplastic changes in the lining or the wall of
a nonneoplastic odontogenic cyst, most commonly the
dentigerous cysts and odontogenic keratocysts.[1] Among
the different clinicoradiographic types, the conventional
solid/multicystic intraosseous ameloblastoma is the most
common, accounting for around 86% of lesions.[7]
Ameloblastomas are most common in the third to fifth
decades of life, but have has been reported in patients
with age ranging from 10 to 90 years.[3] Some authors
describe these lesions as being more common in males,
while others state a female predilection. Approximately
80% of ameloblastomas occur in the mandible, usually in
the posterior(molarascending ramus) region, and 1015%
may be associated with a nonerupted tooth.[4] The present
case was seen in a 20yearold female, in the angle and
ramus region of the mandible, and was associated with an
impacted mandibular third molar.
Patients may present with a slowgrowing, painless
swelling or expansion of the jaw, which results in facial
asymmetry. These tumors characteristically expand within
the jaw and malocclusion or mobility of teeth may occur,
but paresthesia and pain are uncommon.[1] As the lesion
grows, it may erode through the bone and extend into the
soft tissues.[3] Lesions may also occasionally be detected
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Histopathologically, ameloblastomas show a fibrous


stroma with islands or masses of proliferating epithelium
that resemble odontogenic epithelium of the enamel
organ, with palisading of cells around proliferating
nests of odontogenic epithelium in a pattern similar to
ameloblasts. The follicular pattern, which is the most
common, consists of islands of epithelium containing
a core of loosely arranged angular cells resembling
Indian Journal of Oral Sciences Vol. 6 Issue 1 Jan-Apr 2015

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Figueiredo, etal.: Ameloblastoma of the acanthomatous and plexiform type in the mandible

stellatereticulum, surrounded by a single layer of tall


columnar cells with nuclei showing reverse polarity.
The plexiform pattern shows long anastomosing
cords or sheets of odontogenic epithelium, while
the acanthomatous type shows extensive squamous
metaplasia and keratin formation in the central portion
of the islands.[5] The present case resembled a dentigerous
cyst/unicystic ameloblastoma radiographically, but the
histopathological analysis showed separate areas of
both the plexiform and acanthomatous patterns of a
conventional solid intraosseous ameloblastoma.
Although ameloblastomas may arise from dentigerous
cysts by ameloblastic transformation of the cyst lining,
this remains a point of controversy. Two reasons for
this include: Firstly, an ameloblastoma may involve an
unerupted tooth, particularly a third molar at the angle
of mandible and may thus radiographically resemble
a dentigerous cyst; secondly, a biopsy of an ameloblastoma
may often be taken from an expanded locule lined by a
thin layer of epithelium, and may histologically appear as
a dentigerous cystwhen the tumor is entirely removed,
a diagnosis of ameloblastoma is made, which may be
misinterpreted as arising from a dentigerous cyst.[10] This
was evident in the present case where the findings of an
incisional biopsy, correlated with the radiographic findings,
suggested a dentigerous cyst with secondary changes of
an ameloblastoma, while histopathological examination
following removal of the entire tumor revealed features
suggestive of an ameloblastoma.
The treatment of ameloblastomas depends on the
clinicoradiologic variant, anatomic location and clinical
behavior of the tumor. The age and the general state
of health of the patient are also important factors.[11]
Conventional ameloblastomas are locally invasive with a
high rate of recurrence if not adequately removed. Surgical
treatment of ameloblastoma may be either conservative
or radical. The conservative approach is usually carried
out for unicystic/small unilocular lesions and includes
enucleation, curettage or surgical excision with peripheral
osteotomy.[12] Radical treatment measures are advocated
for large lesions, which include marginal or segmental
resection of the diseased section of the jaw and inclusion
of about 1 or 2 cm of apparently uninvolved bone.[6]
Supraperiosteal resection of the bone is necessary when
extensive thinning or perforation of the cortical plates
is noted. In the mandible, either fullthickness resection
or resection with preservation of the lower border is
done, while resection of maxillary lesions is defined
by the anatomic extension of the excision in partial or
total maxillectomy.[12] Therapeutic irradiation should not
be used in the treatment of ameloblastomas as it can
Indian Journal of Oral Sciences Vol. 6 Issue 1 Jan-Apr 2015

lead to osteonecrosis and has a potential for inducing


postradiation malignancies.[9]
Conventional solid intraosseous ameloblastomas have a
welldocumented tendency for recurrence, especially if
treated by conservative procedures like curettage. This is
because the lesion characteristically infiltrates between the
trabeculae of cancellous bone of the jaws, and thus extends
beyond its radiographically apparent margin.[13] Because
of their slow growth, recurrence of these lesions may be
longdelayed, and hence a longterm postoperative follow
up is essential for all patients.

Acknowledgment
Dr.Anita Spadigam, Professor and Head, and Dr.Anita Dhupar,
Asst. Professor, Department of Oral and Maxillofacial Pathology,
Goa Dental College and Hospital, Bambolim, GoaIndia, for
the histopathological analysis and photographs.

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How to cite this article: Figueiredo NR, Meena M, Dinkar AD,


Khorate M. Ameloblastoma of the acanthomatous and plexiform type
in the mandible presenting as a unilocular radiolucency. Indian J Oral
Sci 2015;6:34-7.
Source of Support: Nil, Conflict of Interest: None declared

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