Ameloblastoma of The Anterior Mandible

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Case Report

Ameloblastoma of the anterior


mandible
Department of Oral and Maxillofacial Hariram, Shadab Mohammad, Laxman R. Malkunje,
Surgery, King George’s Medical
University, Lucknow, Uttar Pradesh, Nimisha Singh, Sugata Das, Gagan Mehta
India

ABSTRACT

Ameloblastoma or adamantinoma is the rarest of the three forms of tumor of the odontogenic
type. They are benign, locally aggressive neoplasms arising from ameloblasts, which typically
occur at the angle of the mandible, and are often associated with an un‑erupted tooth and
must, therefore, be differentiated from a dentigerous cyst which will be centered on the crown.
When in the maxilla (less common), they are located in the premolar region, and can extend
up in the maxillary sinus. Ameloblastoma is reported to constitute about 1-3% of tumors and
cysts of the jaws. The tumor is by far more common in the mandible than in the maxilla and
Address for correspondence: shows predilection for various parts of the mandible in different racial groups. The relative
Dr. Hariram, frequency of the mandible to maxilla is reported as varying from 80-20% to 99-1%. Here, we
Department of Oral and are representing a case of ameloblastoma of anterior mandible which was considered as a
Maxillofacial Surgery, King rare site of occurrence.
George’s Medical University,
Lucknow, Uttar Pradesh, India.
E‑mail: [email protected]
Key words: Ameloblastoma, anterior mandible, rare

Introduction painless, and present with bony deformity. Wide surgical


excision with adequate safe margins is the treatment of
Ameloblastoma is a benign, slow growing, locally choice. We present a rare case ameloblastoma of anterior
invasive odontogenic tumor. [1] It is the second most mandible arising from symphysis. Segmental resection
common odontogenic neoplasm. It accounts for 11% of mandible was done and stabilized with reconstruction
of all odontogenic tumors. It occurs in all age groups plates.
but the lesion is most commonly diagnosed in the third
and fourth decades.[2] Exact etiology of ameloblastoma
is not known. Ameloblastoma may arise from: Cell rest Case Report
of enamel organ either remnant of dental lamina or
remnant of Hertwigs sheath; epithelium of odontogenic A 60‑year‑old lady reported to the Department of Oral
cyst; and disturbances of developing enamel organ.[3] and Maxillofacial Surgery (OMFS) with chief complaint
The tumor frequently develops in the mandible (80%), of swelling at anterior region of mandible since 6 months.
maxilla (16%) while peripheral adamantinoma located Patient gave history of trauma at chin region due to Road
in the soft tissue account for remaining 4%. [4] In the Traffic Accident (RTA) 3 years back. At that time, patient
mandible, it frequently involves molar and mandibular was conservatively managed by some private clinician.
angle (70%), premolar (20%), and rarely anterior After 6 months of trauma, patient noticed an extra oral
region (10%). [4] The tumor is usually asymptomatic, swelling over anterior mandible with pus discharge
intraorally. After 1 year of time, patient noticed loosening
of mandibular anterior teeth. Patient again went to a
Access this article online
private clinician where she was again conservatively
Quick Response Code:
Website: managed with medication. Since past 2 years pus used
www.njms.in to appear intermittently from same place, which used to
stop on use of antibiotics. But since last 6 months, there
DOI:
is gradual increase in swelling of anterior mandible for
10.4103/0975-5950.140173 which patient reported to OMFS department where
patient was examined, evaluated and incisional biopsy of

National Journal of Maxillofacial Surgery | Vol 5 | Issue 1 | Jan-Jun 2014 | 47


Hariram, et al.: Anterior mandibular ameloblastoma

lesion was done which was suggestive of ameloblastoma. Incisional biopsy revealed epithelial islands admixed with
Then patient was admitted to OMFS ward for further fibrocollagenous tissue. Outermost layer of epithelial island
management. was composed of tall columnar cells with polarization of
the nuclei away from the basement membrane. The central
Extra‑oral inspection showed a large swelling in anterior portion of the island was composed of loose network of
region of mandible over chin area crossing midline, cells showing squamous metaplasia [Figure 3].
measuring approximately 8 × 7 cm in size. Face was
asymmetrical, deviated towards left. Scar mark was Orthopantomogram (OPG) of mandible showed
present over right side of chin. Color of the overlying multilocular expansile lytic lesion involving the symphysis
skin was normal. On palpation, the swelling was bony and bilateral body of mandible. All mandibular teeth were
hard. It was non‑compressible, non‑fluctuant, and lost except one molar tooth on either side [Figure 4].
slightly tender. Temperature of overlying skin was
normal. Swelling extends to involve bilateral body of Computed tomography (CT) scan revealed: Plain and
mandible [Figure 1]. contrast axial and coronal CT scans of face region revealed
large multiloculated cystic expansile lesions arising from
Intra‑oral inspection showed a large swelling seen symphysis menti and bilateral body of mandible, and
involving buccal vestibule, floor of the mouth, and extending up to bilateral angle of mandible. Margins of
lower alveolus region. Mouth opening was adequate. the lesions were sclerotic and scalloped. Overlying all
On palpation, intraoral swelling was hard and mild mandibular teeth was lost except one molar tooth on
tender. Sinus opening with respect to lower right and either side. Bilateral masseter muscles and tongue was
left canine (may be socket of teeth). There was bleeding normal in CT attenuation and dimensions. No obvious
from lower left canine region on palpation [Figure 2]. cervical lymphadenopathy [Figures 5 and 6].

On the basis of above findings, resection of mandible and


reconstruction with reconstruction plate was planned,
under general anesthesia. The mandible was resected
bilaterally at the body. The specimen was taken out and
the defect was reconstructed using reconstruction plate.

Figure 1: Extraoral photograph of patient

Figure 2: Intraoral photograph of patient

Figure 3: Histopathological picture Figure 4: Orthopantogram of patient

National Journal of Maxillofacial Surgery | Vol 5 | Issue 1 | Jan-Jun 2014 | 48


Hariram, et al.: Anterior mandibular ameloblastoma

Figure 5: CT scan-coronal view Figure 6: CT scan-axial view

Figure 8: Postoperative orthopantogram of patient

Discussion
Figure 7: Intraoperative photograph of patient

Ameloblastoma or admantinoma is a benign odontogenic


tumor of epithelial origin. It was described in 1827 by
Cusack and designated as an adamantinoma in 1885 by the
French physician Louis‑Charles Malassez and renamed as
ameloblastoma in 1930 by Ivey and Churchill.[5]

Ameloblastoma are seen in wide range of age but are


usually diagnosed between the 4th and 5th decades of
life except in unicystic variety (20-30 years). No gender
predominance is noted.[6] However, some workers have
seen involvements of females frequently.

Etiology of ameloblastoma is unknown. In most cases,


ameloblastoma are usually asymptomatic and found
on routine x‑rays; when they attain considerable size
Figure 9: Postoperative photograph of patient
then they present with jaw expansion. They may cause
displacement of tooth or root resorption. In our case,
The genioglosus and the geniohyoid muscles were tied patient had lower jaw swelling with bony deformity and
to the reconstruction plate and the incision was closed gradual spontaneous fall of lower central incisor and
[Figures 7‑9]. canine teeth, difficulty in chewing of food.

Histopathological findings of excised specimen confirmed Radiologically, ameloblastoma are osteolytic, being
the diagnosis of ameloblastoma. usually lucent and frequently multilocular with

National Journal of Maxillofacial Surgery | Vol 5 | Issue 1 | Jan-Jun 2014 | 49


Hariram, et al.: Anterior mandibular ameloblastoma

well‑defined sclerotic margins which may appear Due to high rate of recurrence of ameloblastoma,
scalloped or expand the cortical plate, tooth roots may long‑term follow up is recommended for more than
move or be resorbed.[7] Approximately, 80% of the tumors 10 years. Regular follow up of the patient should be done,
are found in the mandible.[8,9] The maxilla is infrequently irrespective of the treatment done.
affected. It occurs in the posterior maxilla in 98% of cases
and anterior in 2%. The molar/ramus area is the most
frequently involved in Japanese[10,11] and Whites[12] more References
than 70% of the ameloblastomas involve this region. In
1. Torres‑Lagares  D, Infante‑Cossío P, Hernández‑Guisado  JM,
blacks, ameloblastomas occur more frequently in the
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Surg 1991;19:272‑4.
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treatment of choice and involves complete removal of 9. Olaitan  AA, Adekeye  EO. Unicystic ameloblastoma of the mandible:
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Several factors influences in rate of recurrence which of 3677 cases. Eur J Cancer B Oral Oncol 1995;31:86‑99.
includes clinicopathological variant of tumor, anatomic 13. Grucia  B, Stauffer  E, Buser  D, Bornstein  M. Ameloblastoma of the
site, safe margins during surgery, and histological follicular, plexiform and acanthomatous type in the maxillary sinus:
variant. There are three variant of ameloblastoma A case report. Quintessence Int 2003;34:311‑4.
14. Lau  SL, Samman  N. Recurrence related to treatment modalities of
designated as solid or multicystic or conventional,
unicystic ameloblastoma: A systematic review. Int J Oral Maxillofac Surg
unicystic, and peripheral. The solid variety has the 2006;35:681‑90.
greatest propensity for local infiltration and recurrence.[2] 15. Gardner  DG, Pecak  AM. The treatment of ameloblastoma based on
The dense cortical bone of the mandible prevents the pathologic and anatomic principles. Cancer 1980;46:2514‑9.
tumor from spread than maxilla.[15] Inadequate surgical 16. Montoro  JR, Tavares  MG, Melo  DH, Franco Rde  L, Mello‑Filho  FV,
margins are frequent cause of recurrence. Treatment of Xavier SP, et al. Mandibular ameloblastoma treated by bone resection
and immediate reconstruction. Braz J Otorhinolaryngol 2008;74:155‑7.
ameloblastoma by curettage leave small tumor island in
17. Anastassov GE, Rodriguez ED, Adamo AK, Friedman JM. Case report.
bone, which may later cause recurrence.[16] Aggressive ameloblastoma treated with radiotherapy, surgical ablation
and reconstruction. J Am Dent Assoc 1998;129:84‑7.
Role of radiotherapy, as a useful treatment modality in case 18. Grunwald V, Le Blanc S, Karstens JH, Weihkopf T, Kuske M, Ganser A,
ameloblastoma, is not established.[17] Role of chemotherapy et al. Metastatic malignant ameloblastoma responding to chemotherapy
is not yet well‑defined, however, few reports showed little with paclitaxel and carboplatin. Ann Oncol 2001;12:1489‑91.
response with Cisplatin and Paclitaxel.[18] 19. Bachmann  AM, Linfesty  RL. Ameloblastoma, solid/multicystic type.
Head Neck Pathol 2009;3:307‑9.

Malignant transformations of ameloblastoma are rarely


seen, accounting for less than 1% of cases. Malignant How to cite this article: H, Mohammad S, Malkunje LR, Singh N, Das
ameloblastoma may arise de novo or transformation of S, Mehta G. Ameloblastoma of the anterior mandible. Natl J Maxillofac
Surg 2014;5:47-50.
pre‑existing ameloblastoma. Most common site of spread
are lung, cervical lymph nodes, and rarely brain.[19] Source of Support: Nil. Conflict of Interest: None declared.

National Journal of Maxillofacial Surgery | Vol 5 | Issue 1 | Jan-Jun 2014 | 50


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