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Intramural Unicystic Ameloblastoma

Article in Journal of the College of Physicians and Surgeons--Pakistan: JCPSP · March 2017

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Manas Bajpai Nilesh Pardhe

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LETTER TO THE EDITOR
Intramural Unicystic
Ameloblastoma
Sir,
The term unicystic ameloblastoma (UA) was first used in
1977 by Robinson and Martinez to describe cystic
lesions with clinico-radiographic features resembling an
odontogenic cyst, but histologically showing the
presence of ameloblastomatous epithelium lining part of
the cyst cavity.1 UA accounts for 10 - 15 % of all intra -
osseous ameloblastomas. More than 90% of cases
involve the mandible, usually the posterior region. Up to
80% are associated with an unerupted mandibular third
molar. Ackerman et al. first classified UAs in 3 categories,
namely: Luminal, intraluminal and mural, depending on
their histological patterns.2 Later, this classification was
modified by Philipsen and Reichart, owing to the fact
that UAs show combination of histological patterns.3
A 22-year lady presented to a private clinic with a
painless swelling on her left side of lower jaw from 6
months. Extra-oral examination revealed a localised
swelling on the lower, left posterior region of the face. On
intra-oral examination, a pink colored, dome shaped
swelling of left posterior mandible was seen associated
to teeth #36 #37 and space of missing #38, measuring
2 x 2 cm in dimensions. On palpation, it was a firm to
hard swelling without pain. The color of the overlying
mucosa was normal without any sign of ulceration. It
was non-fluctuant and non-reducible and no discharge
was noticed, nor were pulsations felt. Cervical lymph
nodes were non-palpable.
Panoramic radiograph revealed a large osteolytic,
unilocular radiolucent lesion covered by a sclerotic
margin, surrounding impacted #38, extending from the
ramus of the left mandible to the root of #35. Resorption
of the roots of teeth #36, #37 and #35 found (Figure 1).
Based on all these findings, a provisional diagnosis of
UA was given. Osteotomy and curettage was performed
Figure 1: Large unilocular radiolucent lesion on the left mandible with
associated impacted #38.
Journal of the College of Physicians and Surgeons Pakistan 2017, Vol. 27 (2): 117-118
under general anaesthesia
with the repositioning of ID
nerve and extraction of
teeth #36, #37 and impacted
#38. The excised tissue was
sent for histopathological
evaluation. The defect was
restored with reconstruction
plates, without loss of
continuity. One-year follow-
up of the patient was un-
eventful with a satisfactory
healing and bone formation.
Macroscopically, the gross
tissue showed grey to black
Figure 2: Gross specimen.
cystic specimen with corru-
gated margins (Figure 2).
Histopathological examination of the tissue revealed a
cystic odontogenic epithelium proliferating into the
connective tissue stroma in a plexiform pattern with
anastomosing strands. The cystic epithelium also
proliferated into the luminal space. The connective
tissue stroma was fibrous and showed hyalinisation of
collagen fibres at places (Figure 3a). Stroma also
revealed islands of odontogenic epithelium with
squamous metaplasia (Figure 3b). On the basis of all the
features, a final diagnosis of intramural UA was given.
UAs may simulate odontogenic cysts clinically and
radiographically. Histological examination is the most
sensitive tool to differentiate between odontogenic cysts
and UAs. However, both clinical and radiological findings
share equal contribution to the final diagnosis.4 Small
incisional biopsy specimens may sometimes be
insufficient to arrive at definitive diagnosis, in the light of
the fact that UAs may show a combination of different
histological features. It is of utmost importance to
correlate the histopathological findings with clinical and
radiographic features to arrive at a correct diagnosis.5
Figure 3: (a) Ameloblastomatous proliferation of cystic epithelium in
connective tissue wall and lumen (Hematoxylin and Eosin X20). (b) Islands
of odontogenic epithelium in stroma with hyalinisation of collagen fibres.
(Hematoxylin and Eosin X20).
117
Letter to the editor
REFERENCES
1. Robinson L, Martinez MG. Unicystic ameloblastoma: A
prognostically distinct entity. Cancer 1977; 40:2278-85.
2. Ackermann GL, Altini M, Shear M. The unicystic ameloblastoma:
A clinicopathological study of 57 cases. J Oral Pathol 1988;
17:541-6.
3. Peter A, Reichart PA, Phillipsen HP. Odontogenic tumors and
allied lesions. Chicago: Quintessence Pub 2004; 77-86.
4. Ricci M, Mangano F, Tonelli P, Barone A, Galletti C, Covani U.
An unusual case of unicystic intramural ameloblastoma
and review of the literature. Contemp Clin Dent 2012; 3:
33-8.
118
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5. Bajpai M, Agarwal D, Bhalla A, Kumar M, Garg R. Multilocular
unicystic ameloblastoma of mandible. Case Rep Dent 2013;
835892.
Manas Bajpai and Nilesh Pardhe
Department of Oral and Maxillofacial Pathology, NIMS
Dental College, Jaipur, India.
Correspondence: Dr. Manas Bajpai, Assistant Professor,
Department of Oral and Maxillofacial Pathology, NIMS
Dental College, Jaipur, India.
E-mail: dr.manasbajpai@gmail.com
Received: July 16, 2016; Accepted: November 02, 2016.
Journal of the College of Physicians and Surgeons Pakistan 2017, Vol. 27 (2): 117-118