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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 12  |  Issue : 1  |  Page : 52-56

Cavernous hemangioma of the tongue


Department of Oral and Maxillofacial Pathology, SRM Dental College, Chennai, Tamil Nadu, India

Date of Submission29-Dec-2020
Date of Decision23-Jan-2021
Date of Acceptance10-Feb-2021
Date of Web Publication30-Mar-2021

Correspondence Address:
Dr. Madhu Narayan
Department of Oral and Maxillofacial Pathology, SRM Dental College, Ramapuram, Chennai - 600 089, Tamil Nadu
India
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DOI: 10.4103/srmjrds.srmjrds_138_20

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  Abstract 

Hemangiomas are benign tumors of the oral cavity. Some consider them as hamartomas or developmental anomaly. There are multiple ways of classification of hemangiomas. The most common being as capillary and cavernous ones. In this article, we present a case report of cavernous hemangioma of the tongue reported at our institution. Histopathological and immunohistochemical analyses were done to arrive at the diagnosis.

Keywords: Cavernous hemangioma, hemangioma, histopathology, immunohistochemistry


How to cite this article:
Sivaranjani S, Ulaganathan P, Ruth Chacko RS, Narayan M. Cavernous hemangioma of the tongue. SRM J Res Dent Sci 2021;12:52-6

How to cite this URL:
Sivaranjani S, Ulaganathan P, Ruth Chacko RS, Narayan M. Cavernous hemangioma of the tongue. SRM J Res Dent Sci [serial online] 2021 [cited 2021 Jun 25];12:52-6. Available from: https://www.srmjrds.in/text.asp?2021/12/1/52/312475


  Introduction Top


Hemangioma is known to be a benign, fairly common oral cavity tumor.[1] It is characterized by blood vessel proliferation.[1] It is also known as port-wine stain, strawberry hemangioma, and Salmon patch.[1],[2] Instead of a true neoplasm, some consider congenital hemangiomas present from birth to be hamartomas or a developmental anomaly, as they seldom display penetration into adjacent tissues.[1] In fact, hemangiomas, between hamartomatous malformations and true neoplasms, appear to occupy a gray zone.[3] [Table 1] for the differences between hemangiomas and vascular malformation.[4] The absence of chromosomal alterations does not, however, endorse a genuine neoplastic character.[3] Hemangioma has been graded into eight major categories by Watson and McCarthy. They are: (i) capillary hemangioma; (ii) cavernous hemangioma; (iii) angioblastic or hypertrophic hemangioma; (iv) racemose hemangioma; (v) diffuse systemic hemangioma; (vi) metastatic hemangioma; (vii) nevus vinosus; and (viii) inherited hemorrhagic telangiectasis.[1]
Table 1: Differences between Hemangioma and vascular malformation[4]

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The classification of hemangiomas as capillary and cavernous is essentially based on the vascularization pathway lines found in the lesion.[4] Capillary hemangiomas are made of small capillaries arranged in a lobular pattern.[4] Cavernous hemangiomas, on the other hand, consist of deep and dilated vessels and are capable of reaching large dimensions.[4]

Infantile and congenital hemangiomas are other ways of classifying hemangiomas.[5] Congenital hemangiomas are less common and occur at birth.[5] They consist of two types: Rapidly involuting congenital hemangiomas and noninvoluting congenital hemangiomas.[5]

Infantile hemangioma, the more common type, may either arise from disrupted placental tissue inserted during pregnancy or birth in the soft tissue of the fetus or may arise from circulating endothelial progenitor cells and stem cells.[5]

According to North et al., positive GLUTI staining is known to be highly specific and diagnostic for hemangioma and also assists in the differential diagnosis between hemangioma and other clinically associated vascular lesions.[5]

The involvement of hematopoietic progenitor cells (placenta or stem cells), the conducive environment, genetic alterations, and cytokines play a key role in the occurrence of hemangioma.[5] Proangiogenic factors such as vascular endothelial growth factor, transforming growth factor-beta-1 and basic fibroblast growth factor, abnormal matrix metalloproteinase-9 levels, and gene errors associated with growth factor receptors also lead to hemangioma pathogenesis.[5]

The irregular and focal proliferation of endothelial cells induces hemangioma.[3] More recently, hemangioma development has been associated with somatic mutational events in the gene involved in angiogenesis.[3]

Another hemangioma classification categorizes hemangiomas into focal (more localized) and segmental (more diffuse) types.[5]

The current classification of hemangioma and vascular malformations:[4]

  • Hemangiomas


    • Superficial (capillary hemangioma)
    • Deep (cavernous hemangioma)
    • Compound (capillary cavernous hemangioma).


  • Vascular malformations


    • Simple lesions
    • Low-flow lesions


      • Capillary malformation (capillary hemangioma, port-wine stain)
      • Venous malformation (cavernous hemangioma)
      • Lymphatic malformation (lymphangioma, cystic hygroma).


  • High-flow lesions


    • Arterial malformation.


  • Combined lesions


    • Arteriovenous malformations
    • Lymphovenous malformations
    • Other combinations.


Hemangiomas and vascular malformations present a diagnostic challenge but there are etiological and behavioral differences between both the lesions.[4]


  Case Report Top


A 43-year-old male patient reported to the department of oral surgery at our institution with the chief complaint of a growth on the right side of the tongue for the past 3 months. The patient gave the history of a blister-like swelling on the right side of the tongue present since 3 months following a traumatic bite of the tongue with no associated pain or change in size. The patient was a known hypertensive and was on anti-hypertensive drugs (Bisoprolol-2.5 mg daily).

On clinical examination, the lesion was present on the right posterolateral border of the tongue, was deep reddish-blue in color, round, everted lesion measuring about 0.5 mm in diameter. No bleeding or ulceration was present. Prominent veins of the tongue were evident near the lesion.

Excisional biopsy was done and the provisional diagnosis was given as hemangioma. The specimen was submitted in 10% formalin to the department of oral and maxillofacial pathology for histopathological analysis.

Macroscopically, the specimen was grayish-brown in color on the superior surface and whitish-gray in color on the inferior surface. It was firm in consistency, irregular in shape with dimensions measuring 1.0 cm × 0.7 cm.

Microscopically, the specimen showed a parakeratinized stratified squamous epithelium with an underlying fibrovascular connective tissue stroma. The epithelium was of varied thickness exhibiting hyperplasia to atrophy in focal areas. The underlying connective tissue displayed numerous red blood cells (RBCs), dilated blood vessels with flattened endothelial cells and extravasated RBCs, few adipocytes, large number of inflammatory cells, chiefly lymphocytes. Muscle tissue and salivary acini were evident in the deeper section [Figure 1], [Figure 2], [Figure 3], [Figure 4].
Figure 1: Hyperplastic parakeratinized epithelium

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Figure 2: Chronic inflammatory cell infiltration

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Figure 3: Dilated blood vessels with RBCs and flattened endothelial cells

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Figure 4: Muscle and salivary acini

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Immunohistochemical analysis was done with CK-10 to rule out epithelial anomaly which revealed no abnormal findings [Figure 5].
Figure 5: CK-10-immunohistochemistry

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The histopathological diagnosis was suggestive of “Cavernous Hemangioma”.

The lesion was excised and the patient was followed up for 6 months. No recurrence was noted. The excised site had healed uneventfully.


  Discussion Top


Face, oral mucosa, lips, tongue, buccal mucosa, gingiva, palatal mucosa, salivary gland, alveolar ridge, and jaw bones are the usual sites of hemangiomas of the head and neck.[2],[3] In contrast to males, hemangiomas show a higher predilection in females.[3] Tangles of thin-walled cavernous vessels or sinusoids separated by a scanty stroma of connective tissue are seen.[6]

The development of hemangioma may be related to reproductive age in females, gestational hypertension, and infant birth weight.[3]

Hemangiomas appear to have a known pattern of three distinct developmental phases: Proliferation, quiescence, or rapid growth and involution.[5],[7] The clinical presentation of hemangiomas is generally that of soft, smooth or lobulated, sessile or pedunculated, varying from a few millimeters to several centimeters in size.[6] The color ranges from pink to reddish-purple.[6] Pressure whitening also occurs, and either spontaneously or after minor trauma, hemorrhage can occur.[6] Bi-digital palpation of the area and identification of the absence of blood by the use of finger pressure and the presence of blood by the removal of finger pressure are the key signs for the diagnosis of cavernous hemangiomas.[4] The lesion is of arterial origin if the pulse can be obtained by applying finger pressure.[4]

Hemangiomas may be superficial, deep, or compound, with both superficial and deep components of the latter.[5] In fact, the names of superficial, deep, and compound hemangiomas have been replaced by capillary, cavernous, and capillary-cavernous hemangiomas in the current nomenclature scheme.[5]

Hemangiomas are quite uncommon in the oral cavity.[6] The oral mucosa and skin are most affected in the oral cavity, followed by bones and muscle.[8] Owing to the sensitivity of the tongue to regular, but often-mild trauma, hemangiomas of the tongue are of particular importance.[2]

Compared to other areas, the posterolateral surface of the tongue is more vulnerable to trauma from teeth), so it seems safe to conclude that tumors arising in these regions are caused by such injuries.[9] Such trauma is likely to cause irregular arteriovenous contact.[9] This method of contact would force tiny vessels to withstand pressure suddenly and hold a volume of blood they are not used to.[9] In a way, the sudden stretching and dilation of their walls will mimic a chronic phase of irritation.[9] In order to protect the vessel, the endothelium might be propelled to proliferate, and when this proliferation goes beyond reach, it results in an abnormally differentiated and proliferating network of mitotic endothelial cells.[9] Thus, trauma may have acted as a provocative stimulus in the case discussed above too.

Radiographically, with cortical extension, central hemangiomas have a classical characteristic of “polka-dot” appearance.[10] There is a honeycombed or sunburst appearance with spindles radiating to the periphery, which is a varying characteristic of hemangioma from ameloblastoma.[10] For volumetric analysis, diagnostic differentiation, lesion size analysis, extension, position, follow-up of the lesion under systemic therapy, for identification and ligation of the feeding vessel to the lesion, various imaging maneuvers such as CT, MRI, and Doppler ultrasound may be used.[6]

Hemangioma is commonly seen as a tissue-density tumor with a poor pattern of enhancement due to intratumoral nonenhanced thrombosis.[7] Hemangioma in MRI is isointense in T1-weighted images, hyperintense in T2-weighted images, and typically displays bright enhancement with varying degrees of homogeneity after injection of gadolinium.[7]

Histologically, hemangioma demonstrates a progression from densely cellular endothelial cell proliferation in the early stages to a lobular mass of well-formed capillaries in the mature stage, frequently resembling pyogenic granuloma without inflammatory properties.[9] Established lesions consist of capillary channels of different sizes-well formed, flattened, and endothelium-lined, in a lobular configuration.[2] This standard histopathology was found in the case mentioned in this paper. Involving lesions reveal increased fibrosis and hyalinization with luminal occlusion of capillary walls.[2] There are large dilated blood sinuses in cavernous hemangioma, each with thin walls showing an endothelial lining, as was seen in the case mentioned in this article.[2] Sinusoidal spaces are normally filled with blood, while lymphatic vessels may be present.[2]

Pyogenic granuloma, chronic inflammatory gingival hyperplasia, epulis granulomatosa, thyroglossal or ectopic thyroid relics, telangiectasia, lymphangioma, neurofibroma, angiosarcoma, squamous cell carcinoma, and other vascular lesions of the face or oral cavity, such as Sturge Weber Syndrome, are included in the differential diagnosis.[3],[7],[9] As was done in our case, immunohistochemical analysis will help in shortlisting the differential diagnosis.

Although hemangiomas have conventionally been treated with mere observation and close follow-up, it has been found that about 40% of children are in need of greater intervention because of complications such as bleeding, ulceration, visual axis obstruction, airway obstruction, high-output cardiac failure, or risk for permanent disfigurement.[5]

Treatment modalities for hemangiomas range from no treatment for true hemangiomas to larger lesions requiring either wait and watch policy, intralesional or systemic corticosteroids, embolization, excision, electrolysis, and thermocautery, interferon-alpha-2a immunomodulatory therapy, and photocoagulation laser therapy.[6] Amazing results have been achieved with sodium morrhuate, sodium sulfate tetradecyl, polidocanol and ethanolamine oleate, and hypertonic glucose solution, among the sclerosing agents available.[6] However, sclerotherapy is unsafe for major venous malformations and must be carried out by a trained and experienced interventional radiologist.[8] Laser energy with transcutaneous and contact applications has been studied with argon and Nd:YAG lasers and the pulsed dye laser 585 nm flashlamp can be used in cutaneous and subcutaneous hemangiomas.[6]

However, some of the treatment modalities referred to above have significant side effects, such as excessive bleeding, fibrosis, scarring, esthetic and functional disability due to long-term steroid and radiation effects.[4]

Propranolol has been shown to have a high rate of efficacy in children with hemangioma.[5] It works by controlling hemodynamic cytokines and vascular growth factors.[5] However, strict caution must be exercised before administration to tackle the side effects of this treatment, such as hypoglycemia, lethargy, preexisting cardiovascular disorders such as heart block or reactive airway disease.[5]

While tongue hemangiomas occur rarely, they can cause difficult problems for patients when they do occur, such as frequent bleeding, functional problems with voice, chewing and swallowing, and esthetic deformity.[6] Radiotherapy and chemotherapy are not recommended as a treatment alternative for tongue hemangiomas due to their side effects, such as trouble in chewing, swallowing, and communicating, with CO2 lasers.[6]

Usually, simple excision with or without plastic surgery is indicated when systemic procedures fail or when cosmetic corrections are performed.[6] Conservative surgery can result in recurrence and functional loss or deformity may be caused by radical measures.[6] Cryotherapy tackles this problem and has been successfully used in the treatment of lesions of the tongue.[6]


  Conclusion Top


Thus, it is evident that an accurate diagnosis of the vascular lesions is essential as it can have a profound effect on the choice of the treatment modality and the thence the prognosis. Histopathology serves as the gold standard in the diagnosis of such lesions. Further research is needed in the study of biomarkers delineating such lesions from frank malignancies and improvements in therapeutics, particularly to prevent a recurrence.

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.
Sivapathasundaram B. Shafer's Textbook of Oral pathology. 8th ed. RELX India Private Limited: Reed Elsevier India Private Ltd.; 2016.  Back to cited text no. 1
    
2.
Kripal K, Rajan S, Ropak B, Jayanti I. Cavernous hemangioma of the tongue. Case Rep Dent 2013;2013:898692.  Back to cited text no. 2
    
3.
Rashmi G, Dilip SS, Siddharatha AV, Wasim K. A review of hemangiomas of the oral cavity. UJMDS 2015;03:4-6.  Back to cited text no. 3
    
4.
Newadkar UR. Oral hemangioma or vascular malformation: Different entities! J Indian Acad Oral Med Radiol 2015;27:497-9.  Back to cited text no. 4
    
5.
Richter GT, Friedman AB. Hemangiomas and vascular malformations: Current theory and management. Int J Pediatr 2012;2012:645678.  Back to cited text no. 5
    
6.
Kamala KA, Ashok L, Sujatha GP. Cavernous hemangioma of the tongue: A rare case report. Contemp Clin Dent 2014;5:95-8.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Beghdad DM, Choukry DK, Mkhatri DA, El Krimi DZ, Oukessou PY, Rouadi PS, et al. A cavernous hemangioma of the tongue base presenting as an ectopic thyroid: A case report. Ann Med Surg (Lond) 2020;60:115-20.  Back to cited text no. 7
    
8.
Trivedi K, Soni A, Meshack R, Kulthya RS. Intraoral hemangioma: An overview of the clinical entity. J Int Clin Dent Res Organ 2015;7:79-81.  Back to cited text no. 8
  [Full text]  
9.
Jawanda MK, Narula R, Sharma V, Gupta P. Reddishblue lesion of the tongue. Indian J Dent Sci 2019;11:159-63.  Back to cited text no. 9
  [Full text]  
10.
Dhiman NK. Cavernous hemangioma of mandible: A rare case report. J Oral Maxillofac Radiol 2015;3:83-7.  Back to cited text no. 10
  [Full text]  


    Figures

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