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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 30  |  Issue : 1  |  Page : 85-87

Desmoplastic ameloblastoma of maxilla: Radiologic–Pathologic correlation


1 Department of Oral Medicine and Radiology, Jodhpur Dental College, Jodhpur, Rajasthan, India
2 Department of Oral Medicine and Radiology, Mahatma Gandhi Dental College and Hospital, Jaipur, Rajasthan, India

Date of Submission01-Feb-2018
Date of Acceptance06-Mar-2018
Date of Web Publication23-Apr-2018

Correspondence Address:
Dr. Niharika S Rathore
Department of Oral Medicine and Radiology, Jodhpur Dental College, Jodhpur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_9_18

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   Abstract 


Desmoplastic ameloblastoma (DA) is a rare variant of ameloblastoma, and less than 170 patients have been reported in the literature. The present case deserves special importance because of its rare occurrence, unfamiliar appearance, potentially aggressive nature, and high chances of misdiagnosis. Moreover, the radiographic features of this lesion rarely point towards ameloblastoma. Desmoplastic variant of ameloblastoma tends to infiltrate between bone trabeculae. The risk of recurrence is high. This report is an attempt to help the dental community in developing familiarity with the clinical presentation and at the same time advocating to develop a high index of suspicion in recognizing such cases.

Keywords: Adenoid odontogenic tumor, ameloblastoma, desmoplastic ameloblastoma, maxilla


How to cite this article:
Rathore NS, Yadav N, Shakya H, Jamdade A. Desmoplastic ameloblastoma of maxilla: Radiologic–Pathologic correlation. J Indian Acad Oral Med Radiol 2018;30:85-7

How to cite this URL:
Rathore NS, Yadav N, Shakya H, Jamdade A. Desmoplastic ameloblastoma of maxilla: Radiologic–Pathologic correlation. J Indian Acad Oral Med Radiol [serial online] 2018 [cited 2022 Jun 26];30:85-7. Available from: https://www.jiaomr.in/text.asp?2018/30/1/85/230901




   Introduction Top


Desmoplastic ameloblastoma (DA) was first reported by Eversole et al. in 1984 and was recently included in the World Health Organization's Classification of Head and Neck Tumors (WHO-2005).[1] DA is extremely uncommon, accounting for only 4–13% of all ameloblastomas.[2]

DA is a tumor with specific clinical, radiological, and histological features. DA unlike other variants of ameloblastoma is more frequently seen in the anterior region of jaw, especially the alveolar bone or dentulous areas.[3]

We present one such case of DA in anterior region of maxilla in 22-year-old male patient mimicking adenomatoid odontogenic tumor.


   Case Report Top


A 22-year-old male patient came to the department with a slow growing painless swelling in right anterior tooth region since 5 months causing gradual drifting and mobility of adjacent teeth accompanied with paresthesia. No history of trauma, or prior dental procedure was given. Paresthesia on right side. No lymphadenopathy was present. Intraorally [Figure 1], there was a solitary bony hard nontender well defined ovoid swelling of size approximately 2.5 cm anterioposteriorly extending from labial frenum to distal of 13 cm and 1.5 cm superiorinferiorly extending from attached gingiva of 11, 12, 13 involving alveolar gingival and part of labial mucosa with overlying mucosa same as adjacent. 11, 12, 13 showed drifting with marked extrudation of 11, 12 and palatal tilt of 11 associated with grade 2 mobility. Palatal view [Figure 2] revealed a solitary well defined ovoid bony hard nontender swelling of 1.5 cm in diameter extending from midline to attached gingiva of 11, 12, 13. Vitality testing showed all three nonvital. Aspiration was not possible as no area of perforation was felt.
Figure 1: Extent of swelling buccally

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Figure 2: Palatal extension

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Provisional diagnosis of adenoid odontogenic tumor was given with differential diagnosis of calcifying odontogenic cyst, central giant cell granuloma, ameloblastoma (desmoplastic variant), and central odontogenic fibroma.

Periapical radiograph [Figure 3] and maxillary occlusal view [Figure 4] of region concerned showed large solitary diffuse multilocular radiolucency with areas of calcification, root resorption of 11 with teeth displacement of 11, 12, 13. To rule out the superior extend or any other bony lesion panoramic radiograph was taken which showed right nasal floor involvement [Figure 5]. Incisional biopsy was done which confirmed it DA. Histological section showed islands and cords of odontogenic epithelium in a densely collagenized stroma which predominates. Some epithelium islands appear compressed and angular and the peripheral cells layer is inconspicuous. Few islands showed acanthomatous centers. Stromal metaplastic bone is seen [Figure 6]. The details of the procedure were explained to the patient and a written informed consent was obtained. The patient was subjected to routine radiological and hematological examination. Before going for surgical resection a CT, 3D CT [Figure 7]a and [Figure 7]b was advised which confirmed the extent of the lesion. A partial maxillectomy for tumor resection was performed and the involved teeth were removed. No recurrence has been observed in the 3-year follow-up period.
Figure 3: Periapical radiograph showing mixed radiolucency

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Figure 4: Occlusal view – diffuse large multilocularity

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Figure 5: Panoramic view showing right nasal floor involvement

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Figure 6: Histopathological image (×40) showing epithelial islands and collagenous stroma

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Figure 7: (a) Computed tomography axial view shows area of thickening in right maxillary sinus (b) 3D Computed tomography showing right nasal floor extension

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   Discussion Top


In view of the paucity of DA case series, it continues to remain one of nature's secrets. Up till now, less than 170 patients have been reported in the literature. A painless swelling or bony expansion is the most conspicuous clinical manifestation in most of the cases consistent with this case report. The mean age at initial presentation is 42.3 years (range 17–70 years) with age and gender predilection similar to that of other ameloblastomas.[4] According to previous reports, DA lesions tend to be relatively small (2 cm or less), with the exception of one unusual case that expanded into the maxillary sinus. This tendency makes DA different from conventional ameloblastoma.[5],[6] In this case, the tumor appeared to originate around the right maxillary lateral incisor and canine, because the roots of these teeth diverged, as has been described in a previous report.[7]

In this case there was root divergence with periapical resorption. Root resorption (92%) of adjacent teeth is commonly observed in conventional ameloblastoma,[7] while it is seldom observed in DA (33%) indicating its indolent growth pattern.

DA exhibits a more aggressive behavior than other types of ameloblastoma. This aggressiveness may be due to: 1) potential to grow to a large size; 2) the common location in the maxilla leading to an early invasion of adjacent structures; 3) the diffuse radiographic appearance; and 4) histologic finding of bone invasion.[8]

Three radiological presentations of DA are mentioned in the literature as follows: type I (osteofibrosis type) which has radiolucent as well as radiopaque appearance; type II (radiolucent type) which has a completely radiolucent appearance; and type III (compound type) which has radiolucent as well as radiopaque appearance combined with a large radiolucent change.[3] Radiographic features of our case showed mixed radiodensities which were consistent with that of osteofibrosis type (type 1) which is the most common pattern; the compound type is the least common. The lesion is characterized by osseous metaplasia within the dense fibrous septa and this may be the cause for mixed radiographic appearance, it may not be due to mineralized product by the tumor.

Radiographically, DA shows diffuse border indicating its infiltrative process which accounts for higher imaging to guarantee the actual extent of the lesion, which in turn would reduce its chances of recurrence. Furthermore, the lesion is close to many vital structures and the thin cortical bone is a weak barrier and the tumor usually infiltrates into marrow spaces.

The radiological and histological findings of poor encapsulation and ill-defined borders warrants a radical surgical approach along with long-term follow-up.[8]

Histopathology is the gold standard for diagnosing such lesions. Although, the possibility of misdiagnosis of DA as another odontogenic tumor is high if biopsy specimen is not sufficient enough to warrant the presence of characteristic palisading layer of ameloblastoma in all the epithelial clusters. Ameloblastoma is the second most common tumor, only next to odontoma. Clinicians have considered ameloblastoma as a low-grade malignant tumor.[9] Not all ameloblastomas behave this aggressively and it is therefore important to distinguish between clinical types of ameloblastoma to deliver qualitative treatment to patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Eversole LR, Leider AS, Hansen LS. Ameloblastomas with pronounced desmoplasia. J Oral Maxillofac Surg 1984;42:735-40.  Back to cited text no. 1
[PUBMED]    
2.
Waldron CA, el-Mofty SK. A histopathologic study of 116 ameloblastomas with special reference to the desmoplastic variant. Oral Surg Oral Med Oral Pathol 1987;63:441-51.  Back to cited text no. 2
[PUBMED]    
3.
Nair PP, Bhat GR, Neelakantan S, Chatterjee R. Desmoplastic ameloblastoma of mandible. BMJ Case Rep 2013;17:2013-200082.  Back to cited text no. 3
    
4.
Sun ZJ, Wu YR, Cheng N, Zwahlen RA, Zhao YF. Desmoplastic ameloblastoma - A review. Oral Oncol 2009;45:752-9.  Back to cited text no. 4
[PUBMED]    
5.
Higuchi Y, Nakamura N, Ohishi M, Tashiro H. Unusual ameloblastoma with extensive stromal desmoplasia. J Craniomaxillofac Surg 1991;19:323-7.  Back to cited text no. 5
[PUBMED]    
6.
Kishino M, Murakami S, Fukada Y, Ishida T. Pathology of the desmoplastic ameloblastoma. J Oral Pathol Med 2001;30:35-40.  Back to cited text no. 6
    
7.
Durmus E, Kalayci A, Ozturk A, Gunhan O. Desmoplastic ameloblastoma in the mandible. J Craniofac Surg 2003;14:873-5.  Back to cited text no. 7
[PUBMED]    
8.
Gardner DG, Pecak AM. The treatment of ameloblastoma based on pathologic and anatomic principles. Cancer 1980;46:2514-9.  Back to cited text no. 8
[PUBMED]    
9.
Gardner DG. Some current concepts on the pathology of ameloblastomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod1996;82:660-69.  Back to cited text no. 9
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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