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Oral Oncology EXTRA (2006) 42, 2225

http://intl.elsevierhealth.com/journal/ooex

CASE REPORT

Giant ameloblastoma of jaw successfully treated


by radiotherapy
Rastogi Madhup a,*, Srivastava Kirti a, M.L.B. Bhatt a,
M. Srivastava a, Singh Sudhir a, A.N. Srivastava b
a
b

Department of Radiotherapy, King Georges Medical University, Chowk, Lucknow, UP 226003, India
Department of Pathology, King Georges Medical University, Lucknow, UP 226003, India

Received 12 August 2005; accepted 15 August 2005

KEYWORDS

Summary Ameloblastomas are uncommon tumors, which were thought to be radioresistant. However there is lack of well-documented evidence in the literature
concerning their relative radio responsiveness or radio resistance nature. The present article reports a case of ameloblastoma of jaw, successfully treated by radiotherapy.
c 2005 Elsevier Ltd. All rights reserved.

Ameloblastoma;
Adamantinoma;
Jaw;
Megavoltage
radiotherapy

Introduction
Ameloblastoma (Adamantinoma) are generally considered to be radio resistant tumors. However,
there is lack of well-documented evidence in the
literature concerning their relative radio responsiveness or radio resistance. Many of the old
reports mean little because radiotherapy was changed dramatically with the introduction of mega
voltage irradiation in the late 1950s, allowing for
better distribution of the radiation to the tumor
than had previously been possible. The present
article reports a case of ameloblastoma of jaw,

* Corresponding author. Tel.: +91 522 2240016, +91 983


9074801.
E-mail address: drmadhup1@redimail.com (R. Madhup).

successfully treated by radiotherapy. An attempt


has been made to answer the question of the value
of modern therapeutic irradiation in the treatment
of ameloblastoma.

Case report
A 64 years old male referred to our head and neck
cancer clinic in October 2003 with chief complaints
of gradually increasing painless swelling in the right
side of face for the past 8 years. Physical examination revealed a huge firm to hard growth on right
side of the upper and lower alveolus. A firm to hard
growth was present in the right infratemporal fossa. No significant lymphadenopathy was found in
the cervical and supra clavicular area. All the baseline investigations including complete hemogram,

1741-9409/$ - see front matter c 2005 Elsevier Ltd. All rights reserved.
doi:10.1016/j.ooe.2005.08.004

Radiotherapy in Ameloblastoma
liver function test, renal function test, X-ray chest
and USG abdomen were well within normal limits.
A contrast enhanced CT scan face revealed a big
soft tissue mass on right side of the neck and face
with distorted normal anatomy (Fig. 1). The mass
was invading the oral cavity, buccal mucosa and
pressing over the oropharynx. Mass was also invading the right infratemporal fossa. There was a

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central area of necrosis with patchy contrast
enhancement seen. Histopathological examination
revealed dense cluster of pleomorphic tumor cells
with big hyperchromatic nuclei (Fig. 2). There were
clusters of tumor cells with large oval to roundish
nuclei with ill-defined pink cytoplasm and dispersed chromatin confirming the diagnosis of
ameloblastoma.

Figure 1 A contrast enhanced CT scan face showing a big soft tissue mass on right side of the face and neck. The mass
was invading the oral cavity, buccal mucosa and pressing over the oropharynx. Mass was also invading the right
infratemporal fossa. There was a central area of necrosis with patchy contrast enhancement seen.

Figure 2 (H&E 1000in microscope) smear shows a dense cluster of pleomorphic tumor cells with big
hyperchromatic nuclei. There were clusters of tumor cells with large oval to roundish nuclei with ill-defined pink
cytoplasm with dispersed chromatin.

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R. Madhup et al.

Figure 3 A contrast enhanced CT scan face of the same patient in June 2005 showing near to total resolution of the
disease with distorted anatomy.

Due to the advance and inoperable nature of


tumor, patient was referred to us from department
of surgical oncology for the favor of radiation therapy. Patient was subjected to radiation therapy
after metastatic work up. He was given 60 Gy in
30 fractions using 2 Gy fraction, 5 days a week by
parallel and opposed lateral fields with 2:1 weightage on the affected side by Theratron 780C
machine shielding the eye and brain. The radiotherapy was well tolerated. The soft tissue swelling
showed dramatic response and started resolving
slowly which was assessed by serial clinical and
radiological examination. At present he is having
a facial asymmetry of right side with near to complete resolution of the disease in the latest CECT
(June 2005) and a KPS of 100 (Fig. 3). He has completed 2 years of radiation therapy with no further
disease activity.

Discussion
Term Adamantinoma was introduced by Malassez1
in 1885 to denote the odontogenic tumor that had

been recognized by Cusack2 in 1827 and described


later by Falksson.3 Ivey and Churchill in 1930
changed the name to Ameloblastoma.4 The ameloblastoma is an odontogenic epithelial tumor histologically similar to the embryonic tissue that
produces enamel during tooth formation.5 The biological behavior of these tumors has been well
established, in that they are slow growing, locally
invasive tumors with a high rate of recurrence if
not removed adequately.6 Despite the extensive
literature, the principles of treatment are still
not well established, although most authors
reviewing surgical treatments of ameloblastoma
see m to regard ameloblastomas as radioresisitant,
and hence find no place for irradiation in the treatment armamentarium.5,7,8 These results were obtained in the premegavoltage days of radiation
therapy, Sehdev et al.8 in their review of the
Memorial Sloan-Kettering experience, mentioned
11 patients treated during the period of 1921
1951 prior to the introduction of mega voltage
external irradiation. Three patients had persistent
disease and six had an initial response followed
by later recurrence. Gardner and Pecak6 in 1980

Radiotherapy in Ameloblastoma
studied the role of radiation therapy in ameloblastoma and concluded, There appears to be
general agreement in the literature that the
ameloblastoma is radioresistant and that consequently radiotherapy should not be used in its
treatment. However, there is very little evidence reported, especially recently to substantiate
this belief. In 1982, Reynolds and Pacyniak9 wrote
an important, well-reasoned paper on the effect
of irradiation on ameloblastoma in which they
discussed the basic principles of radiotherapy.
They also analyzed, using modern radiotherapeutic
principles. These authors concluded that clinicians
still do not know if ameloblastomas are radioresistant or radioresponsive but indicated that they believed radiotherapy had a possible role in the
treatment of those patients who were poor surgical
risk, or were unwilling to accept extensive surgical
procedures. A second important paper is that of
Atkinson et al in 1984.10 They published a series
of 10 cases of ameloblastoma treated by megavoltage irradiation, seven with primary irradiation only
and three with both irradiation and surgery. They
concluded, on the basis of their experience and
an evaluation of literature, that ameloblastomas
are radioresponsive. Since then, there are few
reports published in the literature supporting
the role of radiation therapy as a useful modality of treatment in case of ameloblastomas.1115
We, here present a case of advanced ameloblastoma responding well to radiation therapy supporting the view of radioresponsive nature of this
tumor.
There are a number of reasons why our present
knowledge is inadequate concerning the relative
radiosensitivity or radioresponsiveness of ameloblastomas. But, it is apparent from this case and
from the literature, that the ameloblastoma is
not an inherently radioresistant tumor. We believe
that properly applied megavoltage radiation techniques have a useful role in the management of
these tumors, particularly in those cases where a
full surgical excision would be technically difficult
because of bulk and local invasion or where other
medical factors including age, would make radical
surgery inappropriate.

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Conclusion
This case further highlights the role radiotherapy in
advanced, inoperable ameloblastoma of jaw as a
definitive therapeutic option. It further warrants
that advanced, inoperable ameloblastoma be subjected to definitive megavoltage radiation therapy
to define the role of radiotherapy in this tumor.

References
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4. Ivey RH, Churchill HR. The need of a standardized surgical
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