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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 1  |  Page : 61-65

Acanthomatous ameloblastoma: A rare presentation


Department of Oral Medicine and Radiology, St. Joseph Dental College, Eluru, Andhra Pradesh, India

Date of Web Publication8-Sep-2016

Correspondence Address:
Raga Geethika Velugubantla
St. Joseph Dental College, Eluru, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.189993

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   Abstract 

Ameloblastoma is the most frequent odontogenic tumor arising from dental epithelium. It is characterized by its histological resemblance to the enamel organ of the developing tooth germ; but enamel formation is not observed. Acanthomatous ameloblastoma is considered an aggressive odontogenic tumor characterized by irregular verrucous masses adjacent to the tooth, consisting of sheets of keratinizing odontogenic epithelium. Here we are reporting a case of 35-year-old male with swelling on left body of the mandible region which was diagnosed as acanthomatous ameloblastoma based on clinical, radiographic and histopathological features.

Keywords: Acanthomatous ameloblastoma, histopathological, keratinizing, odontogenic


How to cite this article:
Pachigolla R, Velugubantla RG, Chennoju SK, Erva S. Acanthomatous ameloblastoma: A rare presentation. J Indian Acad Oral Med Radiol 2016;28:61-5

How to cite this URL:
Pachigolla R, Velugubantla RG, Chennoju SK, Erva S. Acanthomatous ameloblastoma: A rare presentation. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2019 Nov 12];28:61-5. Available from: http://www.jiaomr.in/text.asp?2016/28/1/61/189993


   Introduction Top


Ameloblastoma is a neoplasm of odontogenic epithelium, especially of enamel organ type tissue that has not undergone differentiation to the point of hard tissue formation.[1] It has been postulated that the epithelium of origin is derived from one of the following sources: (1) cell rests of the enamel organ, (2) epithelium of odontogenic cysts, (3) disturbances of the developing enamel organ, (4) basal cells of the surface epithelium, or (5) heterotropic epithelium in other parts of the body.[2] Ameloblastoma is classified according to WHO and the International Agency for Research on Cancer, 2003 as a benign tumor with odontogenic epithelium, mature fibrous stroma and without odontogenic ectomesenchyme.[3] It is further classified into: Solid/multicystic, extraosseous/peripheral, desmoplastic and unicystic.[4] The multicystic ameloblastoma has histological variants including follicular, plexiform, acanthomatous, granular cell, desmoplastic and basal cell types. Here, we are reporting a case of 35-year-old male patient with swelling on left body of the mandible region.


   Case Report Top


A 35-year-old male patient reported with swelling over the left lower third of the face since 2 years. The swelling was initially small, gradual in onset and became aggressive in growth since 6 months. It was also associated with mild, intermittent pain that aggravated while eating and relieved on taking medication since 6 months. No secondary associated symptoms were noted. Two years back, he had undergone extraction in left posterior mandible region in relation to 37 and root canal treatment in relation to 36. Medical, family, and personal histories were non-contributing.

Extraoral examination revealed a single diffuse swelling of the left lower third of the face [Figure 1]. It was 5 × 2 cm in size, which extended from the right parasymphyseal region, crossing the midline to 2 cm in front of the left angle of mandible anteroposteriorly and 2 cm below the left ala–tragus line to the lower border of the mandible superoinferiorly. The edges appeared indistinct; skin over the swelling appeared tense and glossy, without any change in color. On palpation, the swelling was non-tender and firm to hard in consistency. No local rise in temperature was noted. Intraorally, single diffuse swelling was present that extended from distal side of 42 to distal side of 36, which was 5 × 2 cm in size [Figure 2]. Vestibular obliteration with lobulations was noted. Edges were indistinct. A missing lower left second molar tooth due to extraction was noted. On palpation, the swelling was non-tender and firm to hard in consistency. Based on the history and clinical features, a keratocystic odontogenic tumor was provisionally diagnosed. Ameloblastoma and central giant cell granuloma were considered as differential diagnoses.
Figure 1: Extraoral swelling involving the left body of mandible

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Figure 2: Intraoral swelling crossing the midline

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Radiological investigations were performed and a mandibular occlusal radiograph revealed a large multilocular radiolucency mesial to left third molar with buccal cortical plate expansion. A well-defined increased radiolucency in the left premolar region and a multilocular radiolucency with honeycomb pattern present anteriorly were also observed [Figure 3]. Panoramic radiography showed a large multilocular radiolucency on the left side of the mandible, 0.5 cm mesial to the left third molar, extending anteriorly up to mesial side of right canine region. Coronal radiopacity involving enamel, dentine and pulp in relation to 36 was seen [Figure 4]. Axial and coronal CT of mandible showed osteolytic lesion that extended from distal side of 43 to mesial side of 38, crossing the midline. The internal structure showed a varying radiodensity comprising of hypodense area in the body of mandible and anteriorly hyperdense areas representing honeycomb pattern, which was surrounded by a buccal cortical bone [Figure 5] and [Figure 6].
Figure 3: Occlusal radiograph showing multilocular radiolucency with honeycomb pattern anteriorly

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Figure 4: Orthopantomograph showing multilocular radiolucency

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Figure 5: Axial CT showing osteolytic lesion

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Figure 6: Coronal CT showing osteolytic lesion with honeycomb pattern anteriorly

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The patient was subjected to surgery under general anesthesia. Lower lip split incision was performed [Figure 7]. Transmucosal incision was done from 36 to 43 region. Pathological site was exposed and the tumor was surgically enucleated along with curettage of the site including removal of 31, 32, 33, 34, 35, 36, 41, 42, 43 [Figure 8]. Bone wax was applied and hemostasis attained. Drain was placed and suturing was done layer by layer. Antibiotics, analgesics and anti-inflammatory drugs were given postoperatively.
Figure 7: Lower lip split incision

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Figure 8: Exposed pathologic site

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The excised specimen was sent for histopathological examination [Figure 9]. Examination of hematoxylin and eosin stained sections showed islands of epithelium resembling enamel organ in a mature fibrous connective tissue stroma. The islands of epithelium were peripherally lined by a single layer of tall columnar ameloblast-like cells with reverse polarity. The central core of epithelial islands showed loosely arranged angular cells resembling stellate reticulum. Most of them showed squamous metaplasia associated with keratin formation. Few of the islands showed cells with nuclear pleomorphism and hyperchromatism with sheet-like proliferation of spindle cells [Figure 10]. Based on these findings, a final diagnosis of acanthomatous ameloblastoma was considered.
Figure 9: Excised specimen along with removed teeth

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Figure 10: Hematoxylin and eosin stained sections

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Wound healing was uneventful and the sutures were removed on the 7th postoperative day. Patient was reviewed after 6 months for clinical and radiographic assessment. During follow-up, there were no signs of inflammation, infection or recurrence of the tumor clinically. Radiographically, there was a large radiolucent area in the surgical site representing a surgical scar [Figure 11].
Figure 11: Follow-up orthopantomograph

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   Discussion Top


Ameloblastoma is the most frequent odontogenic tumor arising from dental epithelium. It is characterized by its histological resemblance to the enamel organ of the developing tooth germ; but enamel formation is not observed.[5] Ameloblastoma accounts for about 1% of all tumors of the jaw mainly encountered during third to fifth decades of life.[6] About 80% of all cases occur in mandible, of which 70% cases are seen in the ramus.[7]

Ameloblastomas are defined as benign, locally invasive, clinically malignant, slow-growing tumors, rarely associated with paresthesia and pain. However, many lesions are detected incidentally on radiographic studies in asymptomatic patients.[5],[8] The potential sources for this tumor are the cell rests of the enamel organ (cell rests of Malassez and cell rests of Serres), epithelial odontogenic cysts (dentigerous cysts), basal cells of the surface epithelium of the jaws and heterotrophic epithelium in other parts of the body.[9]

Literatures reveals that follicular ameloblastoma is the most prevalent histological variant (64.9%), followed by the plexiform (13.0%), desmoplastic (5.2%) and acanthomatous (3.9%) varieties.[1] In acanthomatous ameloblastoma, the cells occupying the position of the stellate reticulum undergo squamous metaplasia and keratin formation within the island of tumor.[2] Moreover, recurrent tumors are more aggressive and anaplastic than the primary tumor, and have a biological behavior similar to that of squamous cell carcinoma.[1]

No single standard type of therapy can be advocated. Rather, each case should be judged on its own merits. The stronghold of treatment is surgery with wide resection. If possible, conservative treatment such as enucleation and curettage can be done.[8] Many authors suggest removal of margins 1.5–2 cm beyond the radiological limit to eliminate all microcysts. The present case was treated conservatively by enucleation along with curettage. Many studies suggest that treatment is an important prognostic factor, implicating the cause for recurrence. Follow-up is essential as most recurrences present within the first 5 years; however, some have been observed more than 10 years after the initial treatment.[5]


   Conclusion Top


Ameloblastomas are uncommon benign odontogenic neoplasms that rarely become malignant. To conclude, the definitive diagnosis is based on its histopathological variants which cannot be distinguished clinically and appropriate treatment planning should be provided.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Adebiyi KE, Ugboko VI, Omoniyi-Esan GO, Ndukwe KC, Oginni FO. Clinicopathological analysis of histological variants of ameloblastoma in a suburban Nigerian population. Head Face Med 2006;2:42.  Back to cited text no. 1
[PUBMED]    
2.
Shafer WG, Hine MK, Levy BM, Tomich CE. Textbook of Oral Pathology. Ectodermal Tumours of Odontogenic Origin. 6th ed. Philadelphia: JB Saunders; 1983. p. 276-92.  Back to cited text no. 2
    
3.
Selvamani M, Yamunadevi A, Basandi PS, Madhushankari GS. Analysis of prevalence and clinical features of multicystic ameloblastoma and its histological subtypes in South Indian sample population: A retrospective study over 13 years. J Pharm Bioallied Sci 2014;6(Suppl 1):S131-4.  Back to cited text no. 3
    
4.
Reichart PA, Philipsen HP. Odontogenic Tumors and Allied Lesions. London: Quintessence; 2004. p. 35-55.  Back to cited text no. 4
    
5.
Bhargava A, Saigal S, Chalishazar M. Acanthomatous ameloblastoma of mandible. J Dent Sci Res 2011;2:74-7.  Back to cited text no. 5
    
6.
Bansal M, Chaturvedi TP, Bansal R, Kumar M. Acanthomatous ameloblastoma of anterior maxilla. J Indian Soc Pedod Prev Dent 2010;28:209-11.  Back to cited text no. 6
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7.
Walke VA, Munshi MM, Raut WK, Bobahate SK. Cytological diagnosis of acanthomatous ameloblastoma. J Cytol 2008;25:62-4.  Back to cited text no. 7
  Medknow Journal  
8.
Singh G, Agarwal R, Kumar V, Passi D. Acanthomatous ameloblastoma - A case report. J Int Oral Health 2013;5:54-8.  Back to cited text no. 8
[PUBMED]    
9.
Nadeem J, Jeyapradha T, Ananthalakshmi R, Jeeva S, Saikrishna P, Lakshmipathy P. The molecular and genetic aspects in the pathogenesis and treatment of ameloblastoma. JNTR Univ Health Sci 2013;2:157-61.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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