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CORRESPONDENCE
Year : 2015  |  Volume : 11  |  Issue : 4  |  Page : 946-949

Enriched vascularity in ameloblastomas, an indeterminate entity: Report of two cases


1 Department of Oral Pathology and Microbiology, JSS Dental College and Hospital, Mysore, Karnataka, India
2 Department of Oral Maxillofacial Surgery, JSS Dental College and Hospital, Mysore, Karnataka, India
3 Department of Oral Medicine and Radiology, JSS Dental College and Hospital, Mysore, Karnataka, India

Date of Web Publication15-Feb-2016

Correspondence Address:
Usha Hegde
Department of Oral Pathology and Microbiology, JSS Dental College and Hospital, Mysore, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.163737

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 > Abstract 

Vascularity is a highly essential element that is required for the growth, development, and functioning of the body and variations in it can cause pathologies. It is one of the prime features of a proliferating lesion, where it aids in the growth of the lesion through its nutrition supply. Highly increased vascularity in a disease can itself affect the prognosis of the lesion, and in malignancies, it can induce tumor seeding and secondaries. Most of the pathologies including tumors, related to blood vessels, and vascularity are well established. There are some conditions, wherein altered vascularity is one of the prime components along with other diagnostic components of an established disease. In such cases, these lesions are diagnosed with special names, with varying biological behavior and prognosis in comparison to that of established entity. However, there still are few similar conditions whose nature is uncertain due to the rarity of the lesion and the insufficient scientific evidence which eludes the diagnostician. Here is the report of two cases of ameloblastoma, an established entity, with significant vascularity whose nature is indeterminate.

Keywords: Ameloblastoma, hemangioameloblastoma, odontogenic, unicystic, vascularity


How to cite this article:
Hegde U, Sheshanna SH, Chandan S N, Mahima V G. Enriched vascularity in ameloblastomas, an indeterminate entity: Report of two cases. J Can Res Ther 2015;11:946-9

How to cite this URL:
Hegde U, Sheshanna SH, Chandan S N, Mahima V G. Enriched vascularity in ameloblastomas, an indeterminate entity: Report of two cases. J Can Res Ther [serial online] 2015 [cited 2019 Sep 20];11:946-9. Available from: http://www.cancerjournal.net/text.asp?2015/11/4/946/163737


 > Introduction Top


Ameloblastomas are benign epithelial tumors of odontogenic origin known for about more than 180 years. Yet the variations seen in its presentation is intriguing even today. Though the different variants such as solid, peripheral, desmoplastic, and unicystic ameloblastomas have been established well, the reports of these lesions showing a multitude of histopathologic features continue to tickle our thoughts. [1],[2] In case, such variations have any impact on treatment planning, and prognosis for the patient is not clear, since not many cases with long-term follow-up of such lesions have been reported. Here are reports of two such cases, with a lot of vascular areas mimicking hemangioameloblastoma. Hemangiomatous ameloblastoma is a solid ameloblastoma with a lot of extravasated red blood cells (RBCs) and large endothelial lined capillaries. [2] Although, the present cases were plexiform ameloblastoma and unicystic ameloblastoma of subgroup 1.2.3, they showed a lot of vascular components mimicking the hemangiomatous ameloblastoma, which prompted us to report and discuss them. Hemangioameloblastoma in a unicystic ameloblastoma is not been reported in the literature so far.


 > Case reports Top


Case 1

An 18-year-old female patient came with the complaint of painless swelling over the right side of the face since 3 months. On examination, a diffuse swelling was noticed over the right side of the face extending from angle to parasymphysis region of the mandible. Intraorally, swelling was seen extending from the right lower third molar to the first premolar region, with vestibular obliteration. The expansion of both buccal and lingual cortices was noted. The lower second premolar on that side was congenitally missing. The other teeth in the vicinity were unaffected. Orthopantomograph (OPG) showed multilocular radiolucent lesion with a radio-opaque tooth-like substance at the center [Figure 1]. Expansion and thinning of buccal and lingual cortices was seen on the computed tomography (CT) scan [Figure 2]. An incision biopsy was performed, and specimen sent for histopathological examination.
Figure 1: Orthopantomograph showing multilocular radiolucent lesion with a radiopaque tooth like substance at the center

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Figure 2: Computed tomography scan showing expansion and thinning of buccal and lingual cortices

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Histopathologically, the H and E stained sections showed connective tissue stroma with odontogenic tumor islands resembling ameloblastoma, arranged in a plexiform pattern [Figure 3]. The stroma also showed the presence of numerous vascular areas. The increase in vascularity was seen as dilated endothelial lined vessels and as areas of extravasation of RBCs in the stroma [Figure 4]. Few of the vessel walls showed eosinophilic thickening around them [Figure 5]. Based on these findings a diagnosis of hemangioameloblastoma was given. The patient was treated surgically with partial resection of the mandible and a continuous follow-up for 18 months has not shown any recurrence.
Figure 3: Connective tissue stroma with odontogenic tumor islands in plexiform pattern (H and E, ×100)

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Figure 4: Increased vascularity with RBC's in prominent endothelial lined blood vessels and extravasated red blood cells in stroma (H and E, ×40)

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Figure 5: Prominent dilated endothelial lined blood vessel with eosinophilic cuffing around them (H and E, ×100)

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Case 2

A 24-year-old male patient reported with the history of pain and swelling in lower left back teeth region since 2 months. On examination, a diffuse swelling was seen over the left preauricular and ramus region. It was firm to hard in consistency, without any pulsations or bruit. Intraorally, the buccal vestibule was obliterated.

OPG showed a well-defined multilocular radiolucency with thin cortical margins extending from the left angle of the mandible to the coronoid and condylar process. The left third molar was impacted within the radiolucency [Figure 6]. An incisional biopsy was done, and the histopathology revealed a cystic ameloblastoma like lining and a connective tissue capsule. There were no areas of solid ameloblastoma. Since the gross specimen was that of an incisional biopsy, diagnosis of cystic ameloblastoma was given rather than unicystic ameloblastoma. The left partial mandibulectomy was done and sent for histopathologic examination. Multiple soft-tissue tumor masses and bony margins were taken for processing and histopathological examination.
Figure 6: Orthopantomograph shows a well-defined multilocular radiolucency on the left side of the mandible with impacted third molar within the radiolucency

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The microscopic examination revealed a cystic ameloblastoma lining showing both mural and luminal proliferations. The epithelium lining the cystic cavity showed all cytomorphologic features suggestive of unicystic ameloblastoma with intraluminal and intramural proliferations in a plexiform pattern [Figure 7]. The stroma in these proliferations showed a lot of vascularity. The vascular component consisted of numerous endothelial lined channels and large blood-filled spaces not lined by the endothelium [Figure 8]. The connective tissue capsule of the cyst also showed islands of infiltrating ameloblastoma, with few areas of cystic degeneration. The bony margins were clear of the tumor. Based on all the above excisional biopsy findings, it was diagnosed as unicystic ameloblastoma subgroup 1.2.3. The patient has been followed up since then for the past 1-year now without any recurrence.
Figure 7: H and E stained section showing cytomorphologic features of unicystic ameloblastoma with intraluminal and intramural proliferations

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Figure 8: Plexiform proliferations in the stroma with lot of vascularity (H and E, ×100)

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 > Discussion Top


Not much is known regarding the etiopathogenesis or biologic behavior of hemangioameloblastoma, as there is a lack of report of many such cases. Different schools of thought exist in explaining the pathogenesis of the vascular component in ameloblastomas. It is said that the blood vessels necessary for enamel completion might get abnormally induced and become part of the tumor or any traumatic incident such as tooth extraction might also provide stimulus for its proliferation. [3] Further, it is opined that the disturbance in the repair of neoplastic odontogenic tissue may result in the excessive abnormal vascular component. [3] Hemangiomatous ameloblastoma could be a collision of two tumors, a vascular tumor and ameloblastoma. [4] The vascular component may simply represent a variation in the blood supply to the tumor while the other factors play an important role in tumor proliferation and progression. [5] Many other tumors with prominent vascular features such as angiomatoid malignant fibrous histiocytoma [6] and telangiectatic osteosarcoma [7] have been accepted as histologic entities. The angiomatous nature of this tumor is also clearly appreciated in the CT scan and magnetic resonance imaging findings. [8] Hence, it is opined that ameloblastoma with prominent vascularity to be considered as a separate histologic entity and be termed as hemangioameloblastoma. [9]

The present cases were clearly diagnosed as hemangiomatous ameloblastoma (Case 1) and unicystic ameloblastoma subgroup 1.2.3. (Case 2). They were treated surgically by partial hemimandibulectomy. The follow-up of more than a year in both cases has been uneventful. However, the striking vascular component here cannot be ignored. The term hemangioameloblastoma refers to solid ameloblastoma with a very prominent vascular component. Hence, the Case 1 was diagnosed as hemangioameloblastoma. However, we noticed the same in a unicystic ameloblastoma; should this be categorized as unicystic hemangioameloblastoma is questionable. The most accepted mode of treatment for conventional ameloblastomas is surgical resection of the involved area and at present, it remains the same even for hemangioameloblastomas. Since, there are not many cases of hemangioameloblastoma being reported and especially in unicystic ameloblastoma; it is not possible to predict its prognosis. Only a long-term follow-up of the present case and report of many such cases will throw more light on its behavior.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Reichart PA, Philipsen HP, Sonner S. Ameloblastoma: Biological profile of 3677 cases. Eur J Cancer B Oral Oncol 1995;31B: 86-99.  Back to cited text no. 1
    
2.
Reichart PA, Philipsen HP. Odontogenic Tumors and Allied Lesions. London: Quintessence Publishing Co., Ltd.; 2004. p. 41-84.  Back to cited text no. 2
    
3.
Aisenberg MS. Adamantinohemangioma. Oral Surg Oral Med Oral Pathol 1950;3:798-801.  Back to cited text no. 3
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4.
Oliver RT, McKenna WF, Shafer WG. Hemangio-ameloblastoma: Report of a case. J Oral Surg Anesth Hosp Dent Serv 1961;19:245-8.  Back to cited text no. 4
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5.
Smith JF. The controversial ameloblastoma. Oral Surg Oral Med Oral Pathol 1968;26:45-75.  Back to cited text no. 5
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6.
Enzinger FM. Angiomatoid malignant fibrous histiocytoma: A distinct fibrohistiocytic tumor of children and young adults simulating a vascular neoplasm. Cancer 1979;44:2147-57.  Back to cited text no. 6
    
7.
Matsuno T, Unni KK, McLeod RA, Dahlin DC. Telangiectatic osteogenic sarcoma. Cancer 1976;38:2538-47.  Back to cited text no. 7
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8.
Donnelly LF, Adams DM, Bisset GS 3 rd . Vascular malformations and hemangiomas: A practical approach in a multidisciplinary clinic. AJR Am J Roentgenol 2000;174:597-608.  Back to cited text no. 8
    
9.
van Rensburg LJ, Thompson IO, Kruger HE, Norval EJ. Hemangiomatous ameloblastoma: Clinical, radiologic, and pathologic features. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:374-80.  Back to cited text no. 9
    


    Figures

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



 

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