Histologic Variants of Ameloblastoma : A case series

Redy Pristanto Putra1, Okky Prasetio2, and David Buntoro Kamadjaja3

1
Resident of Oral and Maxillofacial Surgery, Faculty of Dental Medicine, Universitas Airlangga, Surabaya, Indonesia
2
Head of Oral and Maxillofacial Surgery Division, Dr. Moh. Soewandhie General Hospital, Surabaya, Indonesia
3
Head of Oral and Maxillofacial Surgery Department,Faculty of Dental Medicine, UniversitasAirlangga,
Surabaya, Indonesia
redypristanto@gmail.com, okkyprasetio@gmail.com, david-b-k@fkg.unair.ac.id

Abstract
Ameloblastoma is the second most common intraosseous benign odontogenic tumor usually affecting the posterior region of mandible, is slowly
growing, locally invasive, epithelial odontogenic of the jaw with a high rate to recurrence. This is a lesion whose clinical and radiographical
features are similar to all the variants, and a definitive diagnosis is achieved by histopathology only. Ameloblastoma has histologicall patterns,
these pattern are follicular, plexiform, acanthomatous and granular cell. other less common histological variants are clear cell and desmoplastic
cells. Variants with more than one histological pattern for the same ameloblastoma could show, depending on the direction and degree of
differentiation of tumor cells. Here we present a serial case with more than one type of histologic variants of conventional ameloblastoma seen.

Keyword :Ameloblastom,Variants,histological pattern

INTRODUCTION CASE REPORT AND

Ameloblastomas are neoplasms that have
inspired great controversy and clinical interest; their
incidence, radiographic features, treatment and
behavior are still discussed quite often in the
literature.1 The World Health Organization defined
ameloblastoma as a benign but locally aggressive
tumor with a high tendency to recure, consisting of
proliferating odontogenic epithelium lying in a
fibrous stroma.2
Ameloblastomas constitute almost half
(48.9%) of the odontogenic tumors with female :
male and maxilla : mandible ratios of 1:1.7 and 1:8
respectively. It‘s a neoplasm with variable clinical
expression and accounts for 1% of all cysts/tumors
of jaws and 18% of all odontogenic neoplasm.3 It is
a true neoplasm of enamel organ type tissue which
does not undergo differentiation to the point of
enamel formation.4
Numerous cases presented have thrown light on
the variety of histological variants and patterns seen
in ameloblastoma, that may create diagnostic
challenges. Follicular and plexiform are the main
histological variants. In this case series, we report
two patients of ameloblastoma with pattern of the
tumor histological picture are predominantly
follicular differentiation, along with plexiform and
acantomatous cell type showing.
MANAGEMENT
CASE 1
45 year old, a female patient, with complaints
of a lump on the right cheek since 1 year ago,
initially the mass is small but gradually grows larger.
The mass is settled, never deflated, no complaints of
previous tooth pain, no discharge from the mass, no
history of trauma. The patient claimed there were no
other lumps in other part of the body, no significant
weight loss in the past 3 months. Patients denied
having other systemic diseases.
From the clinical examination it was found
that the general condition of the patient was good,
compost mentis, cooperative, and vital signs are
normal. On extra oral examination (Figure 1A) this
patient presented with facial asymmetry, mass in the
right mandibular region, diffuse borders, color equal
to surrounding tissue, dense elasticity consistency,
no fistula, no tenderness in palpation, no enlarged
lymph nodes, inferior border of the mandible was
still palpable.
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Figure 1. (A) Preoperative clinical photographs
showing enlargement of the right mandible. (B) Intra
oral photos. Swelling on the buccal side of the right
mandibular region.
Intra-oral examination (Figure 1B) showed
mass from the distal of the second right premolar to
the right mandibular ramus ascendens, clear borders,
6x7x6 cm in size, firm consistency, the same color
as the surrounding tissue, no lingual mass expansion,
no fistula, no ulcer, no tenderness in palpation.
OPG reviewed (Figure 2) show unilocular
radiolucency in the posterior mandibular region with
inferior border of the mandible was still intact. On
FNAB of examination the nodule in the right
mandibular region, indicated benign cystic lesion,
which can be found in Ameloblastoma.
Figure 2. Preoperative panoramic x-ray showing.
Unilocular radiolucency in the posterior mandibular
region until the mandibular ascending ramus,
inferior border of the mandible was still intact
Incisional biopsy was carried out and tissue
samples were taken from 2 sites, HPA examination
was cystic degeneration tissue, ameloblastoma
(acanthomatous, follicular and plexiform type) was
obtained in the second tissue. Right
hemimandibulectomy was done with Costochondral
Graft and Stabilize with miniplate.
Figure 3. The resected mandible involved in tumor,
(A) hemimandibulectomy specimen (B) Tumor
dissected
Results showed various type of
Ameloblastoma (Follicular, Plexiform, and
Acanthomatous) with the margin of the medial
resection free of tumors.
CASE 2
31 year old, a male patient, with complaints
of swelling in right lower jaw since past 2 years ago.
Swelling was gradual in onset, slowly progressed
since this years to attain the present size and was not
associated with pain, no discharge from the mass, no
history of trauma. Had a FNAB examination at the
Soewandhie public hospital past year. The patient
claimed there were no other lumps in other part of
the body, no significant weight loss in the past 3
months. Patients didn’t have profile of other
systemic disease.
From the clinical examination it was found
that the general condition of the patient was good,
compost mentis, cooperative, and vital sign normal.
On extraoral examination, facial asymmetry was
seen. A well defined swelling was present on the
right lower region of the mandible. The swelling was
extending anteriorly to the right corner of the mouth
and posteriorly to the right angle of mandible region,
superiorly 3 cm below the zygoma and inferiorly to
the inferior border of the mandible. Skin over the
swelling appeared normal without any secondary
changes (figure 9). Normal skin color, ,no fistula, no
ulcer.On palpation, the swelling was non tender,
clear borders, 7x5x2.5 cm in size , firm to hard in
consistency with with few soft spots in several parts.
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Same temperature as the surrounding tissue, no mass
expansion, border of right mandible is not palpable.
No tenderness in palpation, no paresthesia in right
buccal, mentale, and labial region. Enlarged of
regional lymp nodes 1 cm in size, firm in
consistence, mobile with no pain.
A A
B
Figure 4. (A) Preoperative clinical photographs
showing enlargement of the right mandible. (B) Intra
oral photos appear swelling on the buccal side of the
right mandibular region.
Intraorally, a diffused swelling was present in
the buccal aspect of teeth 33, 36, 37, 38, and
extending to lingual area. diffuse borders, hiperemi
to surrounding tissue, no fistula, no ulcer. On
palpation, swelling was non tender, clear borders,
7x5x2.5 cm in size , firm to hard in consistency
with few soft spots in mandible posterior region 44-
46.Uneven surface, same temperature as the
surrounding tissue. Clinically teeth 42, 43, 48 were
mobile.
Incisional biopsy was carried out and the
HPA examination results were taken from 2 sites,
showed Cystic Degeneration tissue, Ameloblastoma
(Acanthomatous, Follicular and Plexiform Type)
was obtained in the second tissue.
OPG reviewed (October 25, 2019) showed
well defined multilocular radiolucency with buccal
and lingual cortical expansion in relation to teeth 33
- 48. Edentoulos in region 43 – 47 Panoramic
radiograph revealed well defined multilocular
radiolucency with coarse and curved internal
septation, extending from distal aspect of teeth 33 to
3
distal aspect of 48,displaced teeth 37 and 38. The
inferior alveolar nerve canal was displaced
downward, and mandibular bone destruction with
inferior border of mandible in region 46 to 47 was
ballooning out and not intact. CT scan review
revealed solid lesion 8,5 x 5 x 5 cm in mentale to
ramus mandible dextra with destruction that area.
Figure 5. Preoperative panoramic photo of the
mandibular mass, shows multilocular radiolucency,
root resorption and mandibular bone destruction
with inferior border of mandible was not intact
Fine needle aspiration biopsy was carried out
and the puncture were taken from the nodul in the
ramus mandible dextra, benign cystic degeneration
tissue was found. Then an incisional biopsy was
carried out, and tissue samples were taken from 3
sites. Ameloblastoma (follicular, acantomathous,
and plexiform type) was obtained in the all tissue.
Right hemimandibulectomy was done with
Costochondral Graft and Stabilize with
reconstruction plate.
Figure 6. The resected mandible involved in tumor
(A) hemimandibulectomy specimen (B) Tumor
dissected
4 DISCUSSION
The ameloblastoma is a histologically almost
always benign odontogenic tumour of the jaw bones.
In 2017, the classification of these lesions underwent
4 Chapter Error! No text of specified style in document.
modifications in terminology with the introduction
of prospective views based on updates in current
genetic studies. WHO 2017 classification divided
ameloblastoma based on clinical and radiographic
characteristics, histopathology, behavioral and
subtypes or variants into four categories;
conventional, unicystic, extraosseous / peripheral,
and metastasizing ameloblastoma. The solid/
multicystic term was discarded, as it could be
confused withthe unicystic type. Desmoplastic
ameloblastoma was also reclassified as a histological
subtype and not as aclinical-pathological entity. 1
Conventional ameloblastoma is the most common,
representing 85% of all ameloblastomas, It can be
divided into follicular, plexiform, acanthomatous
and granular cell morphological patterns. other less
common histological variants are clear cell and
desmoplastic cells.In general, one-third of the
conventional type has a plexiform pattern, one-third
a follicular pattern, and the remaining third
corresponds to the other variants. Its biological
behavior is considered more aggressive due to its
higher incidence of recurrence.11
Ameloblastoma consists of a proliferation of
solid strands, cords and islands of odontogenic
epithelium supported by connective tissue stroma,
This proliferating epithelium can undergo cystic
changes. the histopathological features of
ameloblastoma include hyperchromasia, palisading
and reverse polarization of the basal nuclei of the
epithelial cells with vacuolization of the cytoplasm.
Ameloblastoma shows a variety of histological
patterns depending on the direction and degree of
differentiation of tumor cells. The histological
classification of these lesions was based on the most
predominant subtype, considering that the presence
of more than one histological subtype was observed.
The etiologic factors described relate to the onset of
the lesion after a local trauma, inflammation,
nutritional deficiencies, mutations and/or molecular
alterations, where different signaling pathways
participate.1
A uncommon for the same ameloblastoma to present
A
B
Figure 7 (A) (B). HPA result for case 1. The
presence of more than one histological subtype
conventional ameloblastoma. It presenting multiple
patterns :
(1)Follicular type of islands with Peripheral palisade
and central reticulum stellate pattern. Squamous
metaplasia formation.
(2) Central squamous metaplasia with fibrous stroma
surrounding suggestive of acanthomatous
transformation.
(3) Appear stranded basal cell image of Plexiform
type Ameloblastoma
HPA examination result Histological picture
with more than one histological subtype
conventional ameloblastoma . It presenting multiple
patterns, follicular, acantomathous, and plexiform.
In the study conducted by Fulco GM et al. 14 75.5%
of the solid lesions ameloblastomas ( now it means
conventional ameloblastoma) included were made
up by more than one histological pattern. Similiarly,
in the study carried out by Adeline et al. 15 68.6% of
the solid revealed more than one histological pattern.
Nonetheless, only 16.1% and 19.7% of the solid
lesions evaluated by Reichart, et al. and Kim and
Jang16, respectively, showed more than one
histological type. According to Adebiyi et al., and
Waldron and El-Mofty, ameloblastomas, especially
the large ones, are made up of numerous histological
patterns.9 Although there are various
histopathological types of Ameloblastoma, it is
known that they do not affect the biological
behavior of the neoplasm. It is currently accepted
that there is no relationship between histological
pattern and tumor prognosis. In addition, it is not
different histological patterns.19
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The treatment of ameloblastoma remains histological pattern and tumor behavior or

controversial because it is a benign, locally prognosis.
aggressive tumor with a high recurrence rate. In
surgical planning, it is important to consider whether References
it is a primary or recurrent tumor; the age, size,
1. Cadavid H, et al. Ameloblastomas:
location and duration of the lesion; the presence of
current aspects of the new WHO
cortical bone rupture; and soft tissue involvement.
classification in an analysis of
the treatment may be conservative or radical.(Fujita
136 cases. Surgical and Experimental
et al. 2013). In a recent study, it was observed that
Pathology (2019) 2:17
patients treated with conservative surgery had
2. Wright M, et al. Odontogenic Tumors :
significantly higher recurrence rates when compared
Where Are We in 2017. J Istanbul Univ
to cases submitted to segmental resection. According
Fac Dent 2017; 51: S10-S30
to these variables, radical treatment and Facial
3. Rikung G. Perbedaan Tipe dan Pola
reconstruction procedures with iliac crest grafts or
Distribusi Matriks Ekstraselular Tumor
microvascular fibular flaps may be required.
Ameloblastoma Berdasarkan Gambaran
(Milman et al. 2016)
Tipe Histopatologi. Digital Repository
Radical treatment consists of marginal or block
Universitas Jember. 2015.
resection, by removing the tumor and surrounding
4. Mulia VM. Sitologi Tumor
structures involved, but what needs to be considered
Odontogenik : Ameloblastoma .
from the different types of Ameloblastoma is the
Cakradonya Dent Jurnal. 2015; 7(2):807-
difference in the safety margin of the tumor area
868.
with healthy bones that can be used as a benchmark
5. Asif M, et al. Neglience or tumor
in determining the limit of the surgery. 11 From
aggresiveness: A case report of recurrent
research on resection jaw in amelobalstoma cases
ameloblastoma. SAGE open medical
and safety margins, it was found that the
case reports 2019 Volume 7: 1-4
conventional type of ameloblastoma was more
6. Priya Sahni, Kruti C Patel, Ronak J
aggressive when compared to the unicystic type. 14
Patel, Anil R Patel. Ameloblastoma with
The postoperative histopathological of unicystic type
Multiple Histologic Variants: A Case
showed infiltration up to 0.2 cm. Postoperative
Report with Review of Literature.
histopathological type Acanthomatous type showed
International Journal of Oral &
infiltration of up to 0.5 cm. Postoperative Follicular
Maxillofacial Pathology. 2014;5(4):40-
type histopathological analysis showed infiltration of
47.
up to 0.75 cm Postoperative Plexiform type
7. Cahyawati TD. Ameloblastoma. Jurnal
histopathological analysis showed infiltration of up
Kedokteran Unram 2018, 7 (1): 19-25.
to 0.75 cm.15 For large ameloblastomas, in order to
8. Khaled Shaikhi, Mirdza Neiders.
ensure the removal of micro-cyst and
Morphological Variants of
daughter cyst, resection of jaw should be done
Ameloblastoma
approximately 1.5–2 cm beyond the radiological
and Their Mimickers. North American
limit.18 In our case, marginal clearance of 2 cm was
Journal of Medicine and Science, Jan
also achieved.
2012 Vol 5 No.1, pp 20-28
9. Mosmi Lomi. Ameloblastoma of the
5 CONCLUSION mandible: A case series. International
Journal of Oral Health Dentistry,
Ameloblastoma can be divided into several types October-December, 2018;4(4):242-246
based on the histopathological features. In one case 10. Neville B, Damm DD, Allen CM,
of Ameloblastoma could show more than one Bouquot J (2002) Odontogenic cyst and
histological patterns of various types of tumors. In: Neville B, Damm D, Allen
Ameloblastoma. Although is not uncommon, a CM, Bouquot J (eds) Oral and
surgeon is expected to be more careful and maxillofacial pathology, 3rd edn.
understand the histological pattern of Saunders/Elsevier, St. Louis, pp 678–
Ameloblastoma, starting from the results of a biopsy 740.
until definitive treatment of the case. It is currently 11. Hertog D, Elisabeth Bloemena, Irene H
accepted that there is no relationship between A Aartman, Isaäc van-der-Waal ,
6 Chapter Error! No text of specified style in document.

Histopathology of ameloblastoma of the

jaws; some critical observations based on
a 40 years single institution experience.
Med Oral Patol Oral Cir Bucal. 2012 Jan
1;17 (1):e76-82.
12. Sangeeta R., Ankit M. Granular Cell
Ameloblastoma : A Case Report.
International Journal of Oral &
Maxillofacial Pathology. 2011;2(4)63-
67.
13. Saify F, Sharma N. Basal Cell
Ameloblastoma : A Rare Case Report
and Review of Literature. Oral &
Maxillofacial Pathology Journal
[ OMPJ ]. Vol 1, No 1, Juni 2010.
14. Fulco GM, et al. Solid Ameloblastomas-
Retrospective clinical and
histopathologic study of 54 cases.
Brazilian Journal of
Otorhinolaryngology 76 (2). 2010
15. Butt FM, Guthua SW, Awange DA,
Dimba EA, Macigo FG. The pattern and
occurrence of ameloblastoma in
adolescents treated at a university
teaching hospital, in Kenya: A 13-year
study. J Craniomaxillofac Surg. 2011;30.
16. Kalaiselvan S, et al. ”Evaluation of
safety margin” in ameloblastoma of the
mandible by surgical, radiological, and
histopathological methods: An evidence-
based study. J Pharm Bioallied Sci. 2016
Oct; 8(Suppl 1): S122–S125.
17. Kishore M, Panat SM, Kishore A, Joshi
A. Folikular ameloblastoma: A case
report. IJSS Case Reports and Reviews
2014; 1(1):1–3.
18. Chaitanya B, Chhaparwal Y, Pai K,
Kudva A. Hybrid Ameloblastoma : an
Amalgam of Rare and Conventional
Ameloblastoma. Contemporary Clinical
Dentistry. January-March 2016. Vol 7.
Issue 1 92.