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Table of Contents   
SHORT COMMUNICATION  
Year : 2012  |  Volume : 23  |  Issue : 1  |  Page : 116-119
A nodular growth on maxillary gingiva


Department of Oral Medicine and Radiology, Bharati Vidyapeeth University Dental College and Hospital, Pune, India

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Date of Submission26-Jul-2010
Date of Decision25-Sep-2010
Date of Acceptance13-Oct-2011
Date of Web Publication26-Jul-2012
 

   Abstract 

A case of angiosarcoma of maxilla is presented. The occurrence of angiosarcoma in the oral cavity is a rare incidence and maxilla is one of the rarest sites to be involved. The purpose of this article is also to emphasize the fact that sometimes small, innocent-looking masses in the oral cavity might actually turn out to be a highly destructive malignant tumor. Hence, a complete radiographic and histopathologic examination is mandatory.

Keywords: Angiosarcoma, Factor VIII related antigen, immunoreactivity

How to cite this article:
Aditya A, Lele S. A nodular growth on maxillary gingiva. Indian J Dent Res 2012;23:116-9

How to cite this URL:
Aditya A, Lele S. A nodular growth on maxillary gingiva. Indian J Dent Res [serial online] 2012 [cited 2021 Apr 20];23:116-9. Available from: https://www.ijdr.in/text.asp?2012/23/1/116/99054
Sarcomas of the soft tissues are uncommon malignancies, accounting for <1% of all cancers. Angiosarcomas are malignant vascular tumors that comprise only approximately 2% of all soft tissue sarcomas. They can occur in any region of the body, but 60% arise in the skin or superficial tissue. [1] In head and neck area, most of these lesions affect the scalp and facial soft tissue and only uncommonly, the oral cavity is the site of primary location. Most of the intraoral tumors are located in the mandible and occurrence in maxilla and maxillary sinus is rare. [1] Due to the rarity and varied clinical appearance, intraoral angiosarcoma can pose a challenge for the diagnosticians.

As skin tumors, they appear as red plaques and nodules, which may be extensive and ulcerate, rarely in the form of bruise, a pyoderma or resembling infected conditions. In the oral cavity, angiosarcoma generally occurs as red, blue or purple nodular tumors. [2],[3]

Because these tumors infiltrate substantially beyond their visible borders, prognosis is generally poor. Even well-differentiated angiosarcoma metastasizes widely and can be difficult to control locally.

The microscopic appearance of angiosarcomas is varied. They show irregular anastomosing vascular channels and immature endothelial cells arranged in the shape of threads or forming capillaries at places. There are three main patterns of growth: an angiomatous pattern with epithelioid feature, spindle cell pattern and an undifferentiated or solid pattern.

Factor VIII related antigen and lectins and Antigen CD31 have been used as a specific diagnostic markers for this type of malignancy. [4]


   Case Report Top


A 75-year-old woman reported to our department with pain and a gingival growth in the right maxillary region. Pain was present for the last 15 days; however, she could not indicate the onset and duration of the growth.

Extraorally, a diffuse, soft, and slightly tender swelling was present involving infraorbital and buccal spaces. A solitary, non-tender right submandibular lymph node was palpable, which was firm to hard in consistency.

Intraorally, a multilobulated, sessile mass was seen involving the gingival and alveolar mucosa in the maxillary right molar region. Maximum diameter of the mass was about 3 cm. Color of the overlying mucosa ranged from pale pink to red in one lobule and was bluish in another lobule. A few petechiae were also noticed. On lifting the mass, erosion of the buccal cortex and another lobule could be seen arising from deeper tissues [Figure 1]. A small amount of pus could be seen on the inner aspect of the mass. A slight swelling and redness could also be seen on the palatal gingival of the first molar. On palpation, the mass was soft to firm in consistency and was slightly tender. Moderate bleeding could be elicited on probing. The periodontal status of the teeth in the region was poor. The first molar showed gross destruction of distal half of the tooth and grade II mobility. Grade I mobility was elicited in the second premolar and the second molar. Resorption of the floor of the maxillary sinus and its opacification seen on the periapical radiograph [Figure 2] warranted further investigations. A Paranasal sinus view was obtained, which showed opacification of the right maxillary sinus and resorption of its lateral wall, and of zygoma [Figure 3]. Computed tomographic examination revealed a large, soft tissue density lesion in the maxillary and ethmoidal sinuses, and opacification of frontal sinus on the right side of the face. Extending medially, the lesion had partially obliterated the right nasal cavity and obscured the turbinates. Destroying the postero-lateral wall of the maxillary sinus, the lesion had extended into the infratemporal and zygomatic region [Figure 4]. On post-contrast study, it showed heterogeneous enhancement with non-enhancing areas of necrosis within. Superiorly, the inferior wall of the right orbit was destroyed and the lesion extended into the extraconal compartment [Figure 5].
Figure 1: Intraoral view shows erosion of buccal cortex

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Figure 2: Intraoral periapical radiograph shows resorption of the floor of the maxillary sinus

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Figure 3: PNS view shows opacification of the right maxillary sinus, resorption of its lateral wall, and of zygoma

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Figure 4: CT scan shows soft tissue density lesion involving maxillary sinus and obscuration of nasal turbinates

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Figure 5: Post-contrast CT scan shows opacification of maxillary and ethmoidal sinuses, obliteration of right nasal cavity, obscuration of the nasal turbinates, and destruction of inferior wall of the right orbit

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Based on the clinical presentation of the lesion, our initial list of differential diagnosis included irritational hyperplasia, peripheral giant cell granuloma, peripheral fibroma, and pyogenic granuloma.

All these lesions considered in the differential diagnosis pointed toward a reactive lesion of the gingiva. However, on lifting the nodular growth, another growth could be appreciated which appeared to be protruding out of the alveolus. Hence, the probability of a growth extending from the deeper tissues could not be ruled out. Also, the radiographic presentation of the lesion did not correlate with the innocuous clinical presentation of the growth. The intraoral periapical radiograph, showing advanced periodontal bone loss, resorption of the floor of the maxillary sinus and its opacification, indicated toward a relatively more destructive lesion. At this point, we considered the possibility of a benign, or even a malignant tumor in the list of our differential diagnosis.

For a clearer picture, a PNS view was obtained, which, along with opacification and resorption of lateral wall of maxillary sinus, also showed resorption of zygoma. This directed us toward considering a malignancy arising in maxillary sinus as a probable diagnosis, which could be either a carcinoma or a connective tissue tumor like fibrosarcoma. Computed tomography report added to our knowledge about the extent of the lesion and helped us in ruling out any osseous origin. Considering the advanced age of the patient and the relative prevalence of different malignancies in the maxillary sinus, squamous cell carcinoma was considered as a more likely diagnosis than a sarcoma of the maxillary sinus.

One of the lobules was excised and its histopathologic examination revealed a tumor mass underneath normal or mildly hyperplastic, ulcerated epithelium. The mass comprised anaplastic endothelial cells arranged in sheets and alveolar pattern, with rampant mitotic activity. The neoplastic cells showed diffusely infiltrative and perithelial distribution, with anastomosing channels [Figure 6]. The histopathologic diagnosis of high-grade anaplastic angiosarcoma was made, which was confirmed by immunoreactivity to Factor VIII related antigen [Figure 7].
Figure 6: Anaplastic endothelial cells with rampant mitotic activity and perithelial distribution (hematoxylin and eosin; original magnification, ×40)

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Figure 7: Tumor cells positive for factor VIII (peroxidase-anti-peroxidase; original magnification, ×40)

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Extensive involvement of the facial structures and advanced age precluded surgical treatment. Screening for metastasis and palliative radiotherapy were advised. The patient, however, declined any further investigations and therapy.


   Discussion Top


Angiosarcoma, also known as malignant hemangioendothelioma, is a rare and aggressive malignant vascular tumor arising from endothelial cells, which accounts for approximately 10% of soft tissue sarcomas in the head and neck. [5] Trauma, longstanding lymphedema, and irradiation of benign vascular lesions appear to be contributory factors in the onset of some cases, but most cases present with no obvious etiology.

Angiosarcoma of maxilla has a predilection for elderly individuals. [4] This is in contrast to angiosarcoma elsewhere in the body where the tumor seems to occur more frequently in children and young adults. [6] There is no apparent gender predilection. Clinically, the rare intraoral angiosarcoma has been described as round or ovoid bluish nodule. [2] The surface can be ulcerated, but the lesion is generally painless, firm on palpation and capable of spontaneous bleeding. [2] Our case showed almost identical findings.

The clinical differential diagnosis list may include pyogenic granuloma, hemangioma, Kaposi's sarcoma, melanoma and metastatic lesions. Occasionally, these masses mimic periodontal disease closely and may be misdiagnosed as reactive lesions such as pyogenic granuloma. The occurrence of primary angiosarcoma in periodontal tissue is particularly rare. [3] This perplexity often leads to difficulty in clinical diagnosis of intraoral angiosarcoma; as in the present case.

Microscopically, the list of differential diagnosis may again be very long because of its resemblance to pyogenic granuloma, hemangioma, hemangioendothelioma, papillary endothelial hyperplasia, angiolymphoid hyperplasia with eosinophilia, Kaposi's sarcoma, and malignant melanoma. Hence, various criteria have been proposed to assist surgical pathologists in distinguishing angiosarcoma from other neoplasms. These criteria include presence of atypia and nuclear pleomorphism of endothelial cells, endothelial multilayering, and an infiltrative growth pattern. [4],[6] The lesion in our patient showed typical histological pattern of angiosarcoma.

One in four angiosarcomas is immunoreactive for Factor VIII associated antigen. CD34 is also expressed by most angiosarcomas. In our case, immunoreactivity was present for Factor VIII associated antigen but not for CD34 antigen. Though not used in our case, perhaps the most reliable and specific immunohistochemistry analysis is to use CD31 antibody, a platelet-endothelial cell adhesion molecule. [4]

Only a few cases of angiosarcoma of the maxilla and maxillary sinus have been reported that it is difficult to draw conclusions regarding the efficacy of various treatment regimens on the prognosis of the tumor at this site. [1] However, most authors believe that surgery in combination with radiotherapy and chemotherapy offers the best chance of survival.

The overall prognosis of angiosarcoma presents a very gloomy picture. It is an aggressive tumor with high propensity for local recurrence and distant metastases. [7] Tumor grade, site and size are prognostic. [8]

The relative rarity of its occurrence in the oral cavity, as well as resemblance of its clinical, radiographic and histopathologic features to other benign and malignant conditions, poses a diagnostic challenge to the clinicians. Immunohistochemistry, however, may help in accurate diagnosis.

 
   References Top

1.Mark RJ, Poen JC, Tran LM, Fu YS, Julliard GF. Angiosarcoma: a report of 67 patients and a review of literature. Cancer 1996;77:2400-6.  Back to cited text no. 1
    
2.Triantafillidou K, Lazardis N, Zaramboukas T. Epithelioid angiosarcoma of the maxillary sinus and the maxilla: A case report and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:333-7.  Back to cited text no. 2
    
3.Abdullah BH, Yahya HI, Talabani NA, Alash NI, Mirza KB. Gingival and cutaneous angiosarcoma. J Oral Pathol Med 2000;29:410-2.  Back to cited text no. 3
[PUBMED]    
4.Florescu M, Simionescu C, M¢arga¢ritescu CL, Georgescu CV. Gingival angiosarcoma: histopathologic and immunohistochemical study. Rom J Morphol Embryol 2005;46:57-61.  Back to cited text no. 4
    
5.Yang XJ, Zheng JW, Zhou Q, Ye WM, Wang YA, Zhu HG, et al. Angiosarcomas of the head and neck: A clinico-immunohistochemical study of 8 consecutive patients. Int J Oral Maxillofac Surg 2010;39:568-72.  Back to cited text no. 5
[PUBMED]    
6.Loudon JA, Billy ML, De Young BR, Allen CM. Angiosarcomas of the mandible: A case report and review of the literature. Oral surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:471-6.  Back to cited text no. 6
    
7.Mücke T, Deppe H, Wolff KD, Kesting MR. Gingival angiosarcoma mimicking necrotizing gingivitis. Int J Oral Maxillofac Surg 2010;39:827-30.  Back to cited text no. 7
    
8.Abdullah BH, Yahya HI, Talabani NA, Alash NI, Mirza KB. Gingival and cutaneous angiosarcoma. J Oral Pathol Med 2000;29:410-2.  Back to cited text no. 8
    

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Correspondence Address:
Amita Aditya
Department of Oral Medicine and Radiology, Bharati Vidyapeeth University Dental College and Hospital, Pune
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.99054

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    Figures

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

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