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Ameloblastic carcinoma of the jaws: Review of the literature

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DOI: 10.4103/jdas.jdas_4_17

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Volume 6 | Issue 2 | July-December 2017
Print ISSN 2277- 4696
E ISSN 2277- 6672
Journal of Dental and Allied Sciences · Volume 6 · Issue 2 · July-December 2017 · Pages 55-??

JDAS

Journal of
Dental &
Allied Sciences
Official Publication of Sinhgad Dental College & Hospital, Pune

www.jdas.in
Review Article

Ameloblastic Carcinoma of the Jaws: Review of the Literature


Ramat Oyebunmi Braimah, Chibuzo Uguru1, Kizito Chioma Ndukwe2
Department of Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto, 1Department of Oral and Maxillofacial Surgery, University of
Nigeria, Enugu Campus, Enugu, 2Department of Oral and Maxillofacial Surgery and Oral Pathology, Obafemi Awolowo University Teaching Hospitals Complex,
Ile‑Ife, Osun State, Nigeria

Abstract
Ameloblastic carcinoma is a rare odontogenic malignancy that combines the histological features of ameloblastoma with cytological atypia,
even in the absence of metastases. The major prognostic factor is the clinical course of the disease which includes its aggressiveness, local
destruction, and distant metastatic spread preferentially through hematologic route if neglected. Histologically, ameloblastic carcinoma retains
the features of ameloblastic differentiation and exhibits cytological features of malignancy in a primary or recurrent tumor. Because it is a very
rare lesion, it poses a great difficulty in diagnosis. En bloc removal with 1–2 cm of normal bone margin has been regarded as the safest surgical
modality to ensure disease‑free survival. Literature search was carried out using the Boolean operator “And” between ameloblastoma and
carcinoma on PubMed. Retrieved articles were extensively reviewed for epidemiology, etiology, diagnosis, treatment options, and prognosis
of ameloblastic carcinoma.

Keywords: Ameloblastic carcinoma, cytological atypia, hematologic route, metastasis, odontogenic malignancy

Introduction carcinoma[3] with histologic features of malignancy found in


both the primary and the metastases.[6]
Ameloblastic carcinoma is a rare odontogenic tumor
accounting for 1.5%–2.0% of all odontogenic tumors.[1] Only Ordinarily, the tumor cells in ameloblastic carcinoma resemble
seventy cases have been reported in English literature from the cells seen in ameloblastoma, however they show cytologic
1984 to 2011.[2] In 1982, the term “ameloblastic carcinoma” atypia. The tumor usually has direct extension, lymph node
was introduced by Elzay[3] to depict a malignant epithelial involvement, and metastasis to distant sites especially
odontogenic tumor that histologically retains the features of the lungs. Because it is a very rare lesion, it poses a great
ameloblastic differentiation and exhibits cytological features difficulty in diagnosis. Literature search was carried out using
of malignancy in a primary or recurrent tumor. In the last the Boolean operator “And” between ameloblastoma and
WHO classification update published in 2005, it is defined “as carcinoma on PubMed. Retrieved articles were extensively
a rare odontogenic malignancy that combines the histological reviewed for epidemiology, etiology, diagnosis, treatment
features of ameloblastoma with cytological atypia, even in options, and prognosis of ameloblastic carcinoma.
the absence of metastases.”[2] It has characteristic histologic
features and clinical behavior that requires a more aggressive Epidemiology
surgery than that of ameloblastoma. Malignant ameloblastoma
Ameloblastic carcinoma shows no age group predilection
differs from ameloblastoma due to the presence of metastases,
appears more frequently in men (two‑third of cases) and
they both have the same benign histology.[4] Malignant
ameloblastoma and ameloblastic carcinoma have been used
interchangeably before in the past. However, it is now agreed Address for correspondence: Dr. Ramat Oyebunmi Braimah,
Department of Dental and Maxillofacial Surgery,
that malignant ameloblastoma has the ability to metastasizes Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria.
despite its benign histology in both the primary and the E‑mail: robdeji@yahoo.com
metastatic lesion.[5] On the other hand, ameloblastic carcinoma
demonstrates histologic features of both ameloblastoma and
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DOI: How to cite this article: Braimah RO, Uguru C, Ndukwe KC. Ameloblastic
10.4103/jdas.jdas_4_17 carcinoma of the jaws: Review of the literature. J Dent Allied Sci
2017;6:70-3.

70 © 2017 Journal of Dental and Allied Sciences | Published by Wolters Kluwer - Medknow
Braimah, et al.: Ameloblastic carcinoma of the jaws

involves more often the mandible (two‑third of cases). carcinoma reported by Kruse et  al., [11] 34.6% revealed
According to Dhir et  al.,[7] age range of the patients varies metastasis while 23.1% demonstrated local recurrence. Out
widely with a range of 51–84  years and a mean age of of the 34.6% of metastasis, 26.9% was metastasis to the
53.5 years. No sex or race predilection has been noted;[8,9] lungs, while only one case involved neck lymph nodes.[11]
however, a male to female ratio ranging from 1.2:1 to Dorner et al.[20] have also reported metastasis of ameloblastic
2.7:1[7,10,11] have been documented. Ramesh et al.[12] showed a carcinoma to the lungs. This high percentage of pulmonary
contrary sex ratio of female:male to be 3:2 showing a female metastasis emphasizes the importance of its detection using
preponderance. It occurs primarily in the mandible in about either computed tomography or positron emission tomography
80% of cases reported.[7] Racial predilection of blacks over scans, as well as the need for long‑term follow‑up.[21]
whites have also been reported.[13]
Radiographic Features
Etiology Ameloblastic carcinoma and ameloblastoma can have a
The etiology of ameloblastic carcinoma is largely unknown comparable radiographical features; however, definite imaging
and still controversial.[11] Most cases arise spontaneously features may aid the diagnostic feature.[22] Both lesions can be
without a previous history of cancer (de novo) with few unilocular or multilocular radiolucencies with distinct borders
cases arising following malignant transformation of in ameloblastomas but ill‑defined borders in ameloblastic
ameloblastoma.[14] Ndukwe et  al.,[10] in a multicenter study carcinomas. The borders may show slight marginal sclerosis
of ameloblastic carcinoma in Nigerians reported that most without periosteal new bone formation.[22] There is loss of
of the cases (85%) arise de novo with only 15% presenting lamina dura and resorption of tooth apex.[22] In ameloblastic
as carcinoma ex‑ameloblastoma. Researchers speculate that carcinoma, there is often the presence of focal radiopacity,
genetic and immunologic abnormalities, environmental apparently reflecting dystrophic calcification.[2,23,24]
factors (e.g., exposure to ultraviolet rays, certain chemicals,
ionizing radiation), diet, stress, and/or other factors may play Diagnostic Criteria
contributing roles in causing specific types of cancer.[15] In
The diagnostic criteria of an ameloblastic carcinoma that
individuals with cancer, malignancies may develop due to
distinguishes it from ameloblastoma are based largely on
abnormal changes in the structure and orientation of oncogenes
cytologic atypia and increased mitotic figures.[25] When
or tumor suppressor genes.[15] Oncogenes control cell growth
ameloblastic carcinoma arise de novo, the microscopic
while tumor suppressor genes control cell division and ensure
distinction from ameloblastoma is not very obvious and may
that cells die at the proper time (apoptosis).[15] The specific
be subjective.[17] The presence of numerous mitotic figures are
cause of changes to these genes is, however, unknown.
unusual in ameloblastoma, cases where they are adequately
Conversely, current research suggests that abnormalities of
numerous most likely will justify the diagnosis of ameloblastic
deoxyribonucleic acid, which is the carrier of the body’s
carcinoma.[17] Existing literature suggests that the diagnosis
genetic code, are the underlying basis of cellular malignant
of ameloblastic carcinoma is based on several arbitrary
transformation.[15] These abnormal genetic changes may occur
features, however four have been identified namely: (1) higher
spontaneously for unknown reasons or, more rarely, may be
proliferative mitotic index emphasized by higher mitotic
inherited.[15] Hypermethylation of p16 gene have been reported
activity,[26] higher proliferating cell nuclear antigen expression
to be involved in the malignant transformation of initial benign
and higher Ki67; [27] (2) nuclear atypia such as nuclear
ameloblastoma to an ameloblastic carcinoma.[16] Ameloblastic
pleomorphism and basilar hyperplasia;[17] (3) hyperchromatic
carcinoma may develop from the epithelial tissue that remains
nuclei of basaloid cells;[17] and (4) other features of malignancy
after the development of the teeth and associated structures.[2]
such as perineural or perivascular invasion.[17]
In some cases, it results from malignant transformation of
an existing ameloblastoma or a benign odontogenic cyst.[14] The main differential diagnosis of ameloblastic carcinoma
Benlyazid et al.[2] have reaffirmed this position of ameloblastic is the basaloid variant of squamous cell carcinoma.[28] The
carcinoma as arising de novo, or from odontogenic cyst, or distinguishing features of ameloblastic carcinoma from
from ameloblastoma. The majority have been said to originate squamous cell carcinoma include, the jigsaw puzzle‑type
de novo, and the remaining are malignant transformation of nesting of the tumor cells, the presence of stellate reticulum,
an odontogenic cyst or ameloblastoma.[17] and the distinctive cystic degeneration of the nests.[28] Another
possible differential diagnosis is craniopharyngioma because of
Clinical Features its similarities to odontogenic neoplasia and partially because
of its location in the cranial base.[6]
Unlike ameloblastoma, ameloblastic carcinomas are more
aggressive with perforation of the cortical plate, extension
into surrounding soft tissue,[18] lower lip paresthesia,[17] and Treatment
persistent pain.[18] Furthermore, numerous recurrent lesions Surgery is the mainstay of treatment,[12,29] including prophylactic
and metastasis, usually to cervical lymph nodes, bone, liver, and therapeutic excision of involved lymph nodes.[30] En bloc
and brain have been reported.[5,19] In the series of ameloblastic removal with 1–2 cm of normal bone margin has been regarded

Journal of Dental and Allied Sciences  ¦  Volume 6  ¦  Issue 2  ¦  July-December 2017 71


Braimah, et al.: Ameloblastic carcinoma of the jaws

as the safest surgical modality to ensure disease‑free survival.[22]


This method has resulted in local recurrence rates of <15%.[20]
Some other authors have advocated 2‑ or 3‑cm bony margins
by the means of an en bloc removal. [8,19] Ameloblastic
carcinomas have been reported to recur locally 0.5–11 years
after definitive therapy.[31] Some authors advocated the use
of adjuvant radiotherapy,[29] however others have questioned
its effectiveness.[11,32] Philip et al.[33] have suggested adjuvant
radiotherapy in patients with positive resection margins,
multiple positive lymph nodes, extracapsular spread,
perineural invasion, and those contraindicated for salvage
surgery. Similarly, Roy Chowdhury et  al.[17] have reported
that radiotherapy and chemotherapy seem to be of narrow
value; however, these methods need to be considered when
there is a locally advanced or metastatic disease not amenable
to surgical resection. Chemotherapy as primary treatment
for nonmetastatic disease has been poor.[34] However, when
there is metastatic disease, Ramadas et al.[35] found the use of
cisplatin, adriamycin, and cyclophosphamide to be valuable.
Methotrexate and leucovorin have been also used.[19]

Prognosis
The major prognostic factor is the clinical course of the disease Figure 1: Differences between ameloblastic carcinoma and malignant
which include its aggressiveness, local destruction, and distant ameloblastoma
metastatic spread preferentially through hematologic route if
neglected.[17] In addition, this relatively high risk of distant Financial support and sponsorship
metastasis differs with that of squamous cell carcinomas that Nil.
spread rather by the lymphatic way.[17] Distant metastasis is
Conflicts of interest
usually fatal and may appear as early as 4 months or as late
There are no conflicts of interest.
as 12 years postoperatively.[31] However, once metastases
occurred, the median survival have been reported to be
2 years.[28] It is also important to note that distant metastasis References
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