CASE REPORT
Jurnal Radiologi Dentomaksilofasial Indonesia December 2023, Vol. 7, No. 3: 111-6
P-ISSN.2685-0249 | E-ISSN.2686-1321
http://jurnal.pdgi.or.id/index.php/jrdi/index
Radiographic imagery of aggressive plexiform-type
ameloblastoma in the mandible: a case report
Rakmat Putra Guru Arnawansah1*, Barunawaty Yunus2, Fadhlil Ulum Abdul Rahman2,
Yossy Yoanita Ariestiana3, Husni Mubarak4
ABSTRACT
Objectives: This case report aims to describe the unilocular radiolucent lesion, well-defined, with
radiographic characteristics of plexiform scalloped margins in the anteroposterior
ameloblastoma and its impact on surrounding mandibular region. The second panoramic
tissues in a middle-aged female patient using examination (May 23, 2022) indicated a more
panoramic radiography and computed tomography, aggressive lesion expansion, with evidence of root
along with the case management. resorption and destruction of the inferior
mandibular cortex approaching the mandibular
Case Report: A 43-year-old female patient angle. CBCT findings demonstrated a hypodense/
presented to the Dental Radiology Unit of RSGMP radiolucent lesion extending anteroposteriorly,
UNHAS with a panoramic referral letter, diagnosed superiorly, and inferiorly, leading to displacement,
clinically with anterior mandibular ameloblastoma. root resorption, and destruction of the inferior
This work is licensed under a Extraoral examination revealed an asymmetrical mandibular cortex in the inferior direction.
Creative Commons Attribution 4.0
which permits use, distribution and reproduction, facial appearance with anterior mandibular
provided that the original work is properly cited,
the use is non-commercial and no modifications or
adaptations are made.
enlargement. Intraoral examination showed mucus Conclusion: Based on the characteristics and
membrane enlargement in the anterior mandible structure of the lesion observed through various
region (teeth 37-45), soft consistency, absence of radiographic examinations, a unilocular
crepitus, no palpation tenderness, and mobility in ameloblastoma was suspected. Histopathological
several anterior mandibular teeth. The first examination confirmed the plexiform-type
panoramic radiograph (March 16, 2022) exhibited a ameloblastoma.
Keywords: Plexiform ameloblastoma, computed tomography, jaw tumor, mandible
Cite this article: Arnawansah RPG, Yunus B, Rahman FUA, Ariestiana YY, Mubarak H. Radiographic imagery of aggres-
sive plexiform-type ameloblastoma in the mandible: a case report. Jurnal Radiologi Dentomaksilofasial Indonesia 2023;7
(3)111-6. https://doi.org/10.32793/jrdi.v7i3.954
1
Oral and Maxillofacial Radiology Resi-
INTRODUCTION
dency Program, Faculty of Dentistry,
Hasanuddin University, Makassar,
Indonesia 90245
Ameloblastoma is an aggressive neoplasm specifically.5 Radiographically, ameloblastoma
2 arising from remnants of the dental lamina and appears osteolytic and is typically located in the
Department of Oral and Maxillofacial
Radiology, Faculty of Dentistry, Ha- enamel organ (odontogenic epithelium). It is tooth-bearing areas. Due to its slow growth,
sanuddin University, Makassar, Indo- characterized by aggressive yet benign growth, slow margins are usually well-defined and sclerotic.6
nesia 90245 and locally invasive.1,15 Historically, ameloblastoma Ameloblastomas often form well-defined
2
Department of Oral and Maxillofacial has been known for over half a century. The term radiolucent regions, similar to cysts, with well-
Surgery, Faculty of Dentistry, Hasanud- "ameloblastoma" was suggested by Churchill in defined margins.7 The slow growth rate is generally
din University, Makassar, Indonesia
90245 1934.2 This tumor originates from the development associated with root movement. Root resorption
2
Oral and Maxillofacial Surgery Special-
of enamel organ tissue type that doesn't can sometimes occur in relation to ameloblastoma
ist Program, Faculty of Dentistry, differentiate into enamel formation, as per the growth.5,6 Solid/multilocular ameloblastomas
Hasanuddin University, Makassar, WHO definition.3 typically exhibit soap bubble or honeycomb
Indonesia 90245
Clinical presentation of this lesion is usually appearances. Computed Tomography (CT) or Cone
asymptomatic and can be discovered during routine Beam Computed Tomography (CBCT) can aid in
* radiographic examination or due to asymptomatic surgical planning by revealing cortical destruction
Correspondence to:
Rakhmat Putra Guru Arnawansah jaw expansion. Tooth movement or malocclusion and soft tissue extension. A common appearance is
✉ rahmatdenz1987@gmail.com can sometimes be early signs. Typically, slow a unilocular or multilocular radiolucent lesion with
growth, painless swelling causing facial asymmetry, well-defined borders.8
large lesions can lead to lose teeth, pain, There are six histological subtypes of
Received on: May 2023 paresthesia, and ulceration or nasal region ameloblastoma, including follicular, plexiform,
Revised on: August 2023
Accepted on: November 2023 obstruction.2,3 acanthomatous, granular cell, basal cell, and
Radiographically, ameloblastoma is commonly desmoplastic. Plexiform ameloblastoma is one of
found in bone and detected through dental X-rays the variants with specific histopathological
or panoramic (orthopantomogram/OPG) images characteristics, featuring a fishnet-like pattern of
111 © 2023 JRDI | Published by Ikatan Radiologi Kedokteran Gigi Indonesia
CASE REPORT
proliferating epithelial cells. This lesion manifests as asymmetrical facial appearance (Figure 1).
a unilocular or multilocular radiolucent area in the Upon intraoral examination, there is
mandible or maxilla.4,12 enlargement of the anterior mandibular mucosa in
This article aims to report a rare case of the tooth region 37-45 with dimensions of
plexiform ameloblastoma in a female patient based approximately 12.4 x 5.2 x 2.5 cm, soft consistency,
on radiographic characteristics from panoramic and no crepitus, color and temperature similar to
CBCT images. surrounding tissue, no palpation tenderness,
necrotic tissue in the gingival region of teeth 34 and
35, mobility in teeth 31, 32, 37, 42, 43, 41, 44, and
CASE REPORT 45, along with poor oral hygiene conditions (Figure
2).
A 43-year-old female patient presented to the The result of the first orthopantomograph
Dental Radiology Unit of RSGMP UNHAS with a examination (March 16, 2022) showed a well-
referral letter from an Oral Surgeon for an defined, corticated unilocular radiolucent lesion in
orthopantomogram examination with a 2-month the anterior mandibular region extending up to
interval and MDCT in the following month. (Figure 3a). The following month, the second
Anamnesis results from approximately 2 years ago orthopantomograph examination (May 23, 2022)
indicated complaints of pain and swelling in the revealed the progression of a highly aggressive
anterior lower jaw. The patient's general condition lesion, irregularly unilocular, corticated in all
was good, with no drug allergies and no history of directions anteroposteriorly, superior-inferiorly,
systemic illness. Extraoral examination revealed and mediolaterally, accompanied by displacement
pain, swelling in the anterior lower jaw, and and resorption of the remaining mandibular tooth
roots (Figure 3b).
Figure 1. Extraoral view of the patient (a, c) lateral view; (b) frontal view; (d) view from below showing facial
asymmetry due to swelling in the mandibular region
Figure 2. Intraoral views of the patient: (a) Front view; (b) Upper jaw; (c) Lower jaw
Figure 3. (a) First panoramic radiograph (March 2022) (b) Second panoramic radiograph (May 2022) showing aggressive lesion in the
anterior mandibular region within a one-month interval
Jurnal Radiologi Dentomaksilofasial Indonesia 2023; 7(3); 111-6 | DOI: 10.32793/jrdi.v7i3.954 112
CASE REPORT
Figure 4. Multiplanar CBCT 3D reconstruction view of bone, bone-soft tissue, and soft tissue
Figure 5. Axial view showing a hypodense lesion measuring approximately 90.6 mm x 65 mm
Figure 6. Sagittal view showing lesion dimensions of approximately 72 x 58 mm
113 Jurnal Radiologi Dentomaksilofasial Indonesia 2023; 7(3); 111-6 | DOI: 10.32793/jrdi.v7i3.954
CASE REPORT
Figure 7. Coronal view of CBCT images
Figure 8. (a) Extraoral Incision; (b) Mandibular Segmental Resection; (c) Insertion of Reconstruction Plate; (d) Suturing
Figure 9. (a) Plexiform ameloblastoma with a proliferation of epithelial cells resembling a fishnet pattern; (b) Plexiform ameloblastoma
with string-like strands that anastomose with tumor cells
In the axial view of the mandible, the lesion 65 mm in the axial view (Figure 5).
demonstrates expansion in the anterior-posterior In the sagittal view of the right mandible, the
and medio-lateral directions. The bone in the lesion extends superiorly-inferiorly from the tooth
parasymphyseal region and mandibular body region to the base of the mandible (tooth 45 is
appears extensively destroyed (residual bone pushed far posteriorly with ½ root resorption, root
fragments in the inferior mandibular cortex) with resorption is also observed in teeth 44-43); the
remaining bone in the right-left ramus of the lesion measures approximately 72 x 58 mm in the
mandible; the lesion measures approximately 90 x left sagittal view (Figure 6).
Jurnal Radiologi Dentomaksilofasial Indonesia 2023; 7(3); 111-6 | DOI: 10.32793/jrdi.v7i3.954 114
CASE REPORT
Figure 10. Preoperative intraoral view (Left), Postoperative intraoral view (Right)
In the sagittal view of the left mandible, the resolution modalities such as 3D CBCT radiography
lesion extends superiorly-inferiorly from the tooth are needed to provide a more precise picture. The
region to the base of the mandible, with teeth 36 characteristic radiographic feature of this lesion is
and 37 remaining and root resorption observed on its unique location in the anterior mandibular
the mesial root of tooth 36); the lesion measures region. Besides its distinctive location, the lesion
approximately 57 x 55 mm in the left sagittal view appears to be destructive and aggressive, leading to
(Figure 6). mobility and resorption of the remaining
The MDCT examination results indicated the mandibular teeth. This differs from the findings of
presence of a primary bone tumor in the mandible. Bina Kashyap et al., where the lesion did not cause
The interpretation of the panoramic radiography tooth mobility or root resorption. Radiographic
and CBCT/MDCT results suggests a suspected examinations plays a crucial role in accurately
radiodiagnosis of this lesion as ameloblastoma. The determining the size and extent of the lesion. With
radiographic findings will serve as diagnostic the advancement of technology, sophisticated
support for the treatment procedure and will guide radiographic data such as MDCT and CBCT can
the oral surgeon. guide oral surgeons in treatment planning through
The patient underwent a segmental resection 3D reconstruction models, facilitating surgical
surgery under general anesthesia with intubation procedures. Radiographic examination and lesion
via tracheostomy. The incision pattern was made location are key factors in establishing a diagnosis.
approximately 1-2 cm below the mandibular Intraoral radiography, panoramic radiography,
margin, and an extraoral incision was performed MDCT/CBCT, and MRI are all used as diagnostic
using the transmandibular approach. An intraoral tools. Radiological findings may include expansion
incision was made on the right posterior vestibule of the cortical plate with scalloped margins,
extending to the vestibule of tooth region 45. multilocularity or a 'soap bubble' appearance, and
Subsequently, a segmental resection of the root resorption.4 CT is used to depict soft tissue
mandible was performed, followed by the masses, cortical bone damage, and tumor extension
placement of reconstruction plates and suturing into adjacent structures. Although MRI is not
(Figure 8). The histopathology examination results commonly used for hard tissue examination, it can
reveal the characteristics of plexiform-type provide information about tumor definition and
ameloblastoma (Figure 9). consistency.11
The management of plexiform-type
ameloblastoma follows the same principles as other
DISCUSSION types, involving surgical procedures. The goal of
ameloblastoma tumor management is complete
Plexiform-type ameloblastoma in the anterior tumor removal, aesthetic facial reconstruction, a
mandibular region is a relatively rare occurrence, favorable prognosis, and long-term follow-up
with only a few reported cases in the literature demonstrating favorable conditions. Solid
situated in the anterior region.4 Steven R Singer et ameloblastoma typically requires at least jaw
al. explain that plexiform-type ameloblastomas are resection or excision, as recurrences are reported in
generally found in the posterior mandible and the 50-90% of cases treated with curettage alone.
remaining cases occur in the maxilla, accounting for Resection followed by immediate surgical
5-15% of cases.9 Similarly, Vikrant O. Kasat et al. reconstruction is usually performed for large
mention that plexiform-type ameloblastomas are lesions. Routine follow-up for ameloblastoma cases
commonly found in the posterior mandibular region is essential, as recurrences can be observed up to
with an average age of 37.5 years.10 10-20 years after primary therapy.6
Solid-type ameloblastomas can sometimes Histological examination is integral in
present with varying radiological appearances in ameloblastoma cases. Many described histological
different radiographic examinations, making patterns for ameloblastomas may lack clinical
identification challenging. To establish an accurate relevance. Some present with a single histological
diagnosis, it is advisable to employ multiple type, while others exhibit multiple histological
radiographic techniques.13 In cases like this, high- patterns within the same lesion. Generally, all
115 Jurnal Radiologi Dentomaksilofasial Indonesia 2023; 7(3); 111-6 | DOI: 10.32793/jrdi.v7i3.954
CASE REPORT
subtypes show palisading of columnar cells around REFERENCES
epithelial nests in a pattern resembling ameloblasts
of the enamel organ. The cell nuclei resemble 1. White SC, Pharoah MJ. Oral radiology principles and
reticulum stellate cells of the enamel organ. interpretation. 7th ed. St. Louis, Missouri: Elsevier Mosby;
2014.
Plexiform-type ameloblastomas consist of two to 2. Shafer WG, Hine MK, Levy BM. Shafer’s Textbook of Oral
three layers of stellate-like cells at the periphery, Pathology, Eight edition. Elsevier; 2016. p. 130-2.
forming interconnecting nests.6,14 3. Bonanthaya K, Elavenil P, Manuel S, Kumar VV, Rai A. Oral and
Maxillofacial Surgery for the Clinican. Singapore: Springer
Nature; 2021.
4. Kashyap B, Reddy PS, Desai RS. Plexiform ameloblastoma
CONCLUSION mimicking a periapical lesion: A diagnostic dilemma. J Conserv
Dent. 2012 Jan;15(1):84-6.
5. Underhill TE, Katz JO, Pope TL Jr, Dunlap CL. Radiologic
Plexiform ameloblastoma is a rare jaw bone findings of diseases involving the maxilla and mandible. AJR
tumor with the same etiology as other types of Am J Roentgenol. 1992 Aug;159(2):345-50.
ameloblastoma, typically asymptomatic. The lesion 6. Regezi JA, Sciubba JJ, Jordan RCK. 2017. Oral Pathology:
Clinical Pathologic Correlations, Seventh Edition. St. Louis,
presents as extensive and aggressive on clinical and Missouri: Elsiever; 2017. p. 279-80.
radiographic examinations, with a specific pattern 7. Odell EW. Cawson’s Essential of Oral Pathology and Oral
commonly located in the anterior maxilla or Medicine. Elsevier; 2017. p.183-4.
8. Cohen MA, Hertzanu Y, Mendelsohn DB. Computed
mandible region. Supportive examinations, tomography in the diagnosis and treatment of mandibular
including radiography and histology, greatly aid in ameloblastoma: report of cases. J Oral Maxillofac Surg. 1985
confirming a definitive diagnosis and guiding oral Oct;43(10):796-800.
surgeons in managing this lesion. 9. Singer SR, Mupparapu M, Philipone E. Cone beam computed
tomography findings in a case of plexiform ameloblastoma.
Quintessence Int. 2009;40(8):627-30.
10. Kasat VO, Karjodkar FR, Ladda R. Plexiform unicystic
ACKNOWLEDGMENTS ameloblastoma-a case report and review of literature.
CHRISMED J Health Res. 2014;1(2):103-6.
11. Celur S, Babu KS. Plexiform ameloblastoma. Int J Clin Pediatr
None. Dent. 2012 Jan;5(1):78-83.
12. Wu YH, Chang JY, Wang YP, Chiang CP. Langerhans cells in
plexiform ameloblastoma. J Dent Sci. 2017;12(2):195-7.
13. Pramanik F, Epsilawati L, Lita YA, Herawati E. Analisis
FOOTNOTES gambaran radiologis suspek ameloblastoma tipe solid pada
radiograf CBCT 3D. Jurnal Radiologi Dentomaksilofasial
All authors have no potential conflict of interest Indonesia. 2019;3(2):15-20.
14. Brown NA, Betz BL. Ameloblastoma: A Review of Recent
to declare for this article. Informed consent was Molecular Pathogenetic Discoveries. Biomark Cancer. 2015;7
obtained from the patient for being included in this (Suppl 2):19-24.
case report. 15. Effiom OA, Ogundana OM, Akinshipo AO, Akintoye SO.
Ameloblastoma: current etiopathological concepts and
management. Oral Dis. 2018;24(3):307-16.
16. Hertog D, Bloemena E, Aartman IH, van-der-Waal I.
Histopathology of ameloblastoma of the jaws; some critical
observations based on a 40 years single institution
experience. Med Oral Patol Oral Cir Bucal. 2012;17(1):e76-82.
Jurnal Radiologi Dentomaksilofasial Indonesia 2023; 7(3); 111-6 | DOI: 10.32793/jrdi.v7i3.954 116
