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Table of Contents
Year : 2020  |  Volume : 8  |  Issue : 4  |  Page : 97-102

Hemangiomas of the oral and maxillofacial region: A series of five cases with literature review

1 Department of Oral and Maxillofacial Surgery, MES Dental College and Hospital, Perinthalmanna, Kerala, India
2 Department of Oral and Maxillofacial Surgery, KMCT Dental College, Mukkam, Kerala, India

Date of Submission30-Oct-2020
Date of Acceptance22-Nov-2020
Date of Web Publication29-Dec-2020

Correspondence Address:
Dr. Sooraj Soman
Department of Oral and Maxillofacial Surgery, MES Dental College and Hospital, Perinthalmanna, Malappuram 679321, Kerala.
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/INJO.INJO_46_20

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Most of the vascular tumors in the maxillofacial region are hemangiomas. Hemangiomas are basically benign lesions of a vascular origin. This article describes a series of five cases on hemangioma recorded on the lip, buccal mucosa, and zygoma. Here, we present four variants of hemangioma, namely cavernous hemangioma, capillary hemangioma, lobular capillary hemangioma, and intraosseous hemangioma, which were reported to our Maxillofacial Unit.

Keywords: Capillary hemangioma, cavernous hemangioma, intraosseous hemangioma, lobular capillary hemangioma

How to cite this article:
Soman S, Das T A, Kalathil LS, Aslam S, Thomas T, Depesh V. Hemangiomas of the oral and maxillofacial region: A series of five cases with literature review. Int J Oral Care Res 2020;8:97-102

How to cite this URL:
Soman S, Das T A, Kalathil LS, Aslam S, Thomas T, Depesh V. Hemangiomas of the oral and maxillofacial region: A series of five cases with literature review. Int J Oral Care Res [serial online] 2020 [cited 2021 Jan 24];8:97-102. Available from: https://www.ijocr.org/text.asp?2020/8/4/97/305360

  Introduction Top

Hemangiomas are defined as the proliferation of blood vessels creating a mass, connected to the main vascular system. They are fundamentally classified as superficial, deep, or compound and as congenital or infantile. Even though hemangiomas of the head and neck region are common, they are a rare entity in the oral cavity.[1] They can be cutaneous (the lips, skin), mucosal, intramuscular (masseter and other perioral muscles), and intraosseous.[2],[3] There is a high incidence of soft tissue hemangiomas noticed among children, whereas hemangiomas of the bones are seen in the elderly. The likelihood for developing hemangiomas in preterm babies increases by 23%.[4] Hemangiomas usually manifest since birth or at early childhood and they comprise a proliferative phase, an early involution phase, and a late involution phase (10–12 years).

Hemangiomas may appear soft, pedunculated, smooth, lobulated, or sessile. They can vary from a few millimeters to several centimeters in size. Hemangiomas of the oral mucous membrane appear slightly raised and are usually blue in color.[5] Lobular capillary hemangiomas (LCHs) are a histological variant of pyogenic granuloma; these have blood vessels in lobules. The most common site for occurrence of the LCH is the lip, with the least being the tongue.[6]

Intraosseous hemangioma occurs in the calvaria and vertebral column. Vertebral and skull hemangiomas are usually asymptomatic, whereas facial intraosseous hemangiomas are commonly symptomatic.[7] Among the calvarial bones, the parietal bones are commonly affected whereas the occipital and temporal bones are least affected.[8] Intraosseous hemangiomas of the facial bones are rare. The sites of occurrence observed in literature are the maxilla, mandible, and nasal bones.[9] Intraosseous hemangiomas of the zygomatic bone are unusual, with an incidence of only about 8% to 12%.[8] If they do occur, they involve the orbit that results in ocular complications such as proptosis, diplopia, and visual loss.[7] A routine biopsy that is usually done to establish the type of lesion is dehorted in hemangiomas due to the risk of hemorrhage, and needle aspiration is mostly preferred.[9] The other investigations performed are computed tomography, magnetic resonance imaging, color Doppler, ultrasonography, and angiography.

The treatment modalities for hemangiomas involve medical and surgical management. The medical management involves the use of corticosteroids, beta blockers, alpha interferon, and sclerosing agents. The surgical management involves laser photocoagulation, curettage, embolization, and surgical excision. In intraosseous hemangioma, the preferred curative treatment modality is complete resection with safe margins. Here, we present a series of five cases describing four different variants of hemangiomas.

  Case 1 Top

A 54-year-old female patient reported to our division of Oral and Maxillofacial Surgery with the chief complaint of painful swelling on the left side of the face for three months. Pain was gradual at onset, of the moderate throbbing type, radiating toward the forehead that aggravated at nighttime without other associated symptoms. The swelling was initially small in size that later progressed to 4x4 cm over a period of three months. The skin over the swelling appeared to be normal. The mass was found to be hard, tender, immobile, and fixed to the underlying bone over the left zygoma region [Figure 1].
Figure 1: CT scan showing lesion involving the left zygomatic bone

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Our clinical impression favored “Osteoma,” for which CT of the facial bones was advised. The CT image showed an expansile lesion with a sclerotic border and fine radiopaque spots in the radiolucent background internally, resembling a “ground glass” appearance. The lesion was seen involving the left zygomatic bone, infraorbital and lateral orbital walls. In accordance with the CT, we came up with the following differential diagnosis: fibrous dysplasia, ossifying fibroma, or chondrosarcoma. An incisional biopsy was performed, and the report revealed it as a “Hemangioma.”

Complete resection of the hemangioma with safe margins was performed under general anesthesia. Bleeding was encountered and was controlled by cauterization of the vessels involved. Reconstruction of the defect was done by contouring the titanium mesh. The void was filled by harvesting the abdominal fat to eliminate any dead space. The final HP report confirmed it as an “Intra-osseous Hemangioma.”

  Case 2 Top

A 13-year-old male patient presented with the chief complaint of swelling on the right side of the mouth for six years that was not associated with pain. Initially, the swelling was small in size and it gradually progressed to the current size over a period of six years. Intraoral examination revealed a solitary sessile smooth irregular swelling on the right side of the buccal mucosa with a size of approximately 3 × 2 cm. The swelling was dark blue in color, and the lesion showed blanching on pressure [Figure 2].
Figure 2: Intraoral view of the lesion on the right buccal mucosa

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Based on the history and clinical evaluation, the lesion was provisionally diagnosed as a hemangioma. Thus, an ultrasonography was advised. The ultrasonography described the lesion as an ill-defined heterogenous mass with no arterial component within. The impression was that of a cavernous hemangioma. The treatment protocol involved the administration of sclerosing agent 2% tetradecyl sulfate in three sittings, which showed a reduction in the size and appearance of the lesion. Healing was uneventful.

  Case 3 Top

A 26-year-old male patient presented with the complaint of growth on the right side of the lower lip since four months. It was not associated with pain. The patient reported it as being aesthetically displeasing. Clinically, a solitary erythematous lobulated exophytic growth was noted on the right side of the lower lip with a size of approximately 0.6 × 0.6 cm: It was red in color, firm in consistency, fixed to the underlying structures and was nontender on palpation. Based on the clinical features, a provisional diagnosis of squamous cell carcinoma was made [Figure 3].
Figure 3: Lesion on the lip

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Excisional biopsy with safe margins and primary closure was done under local anesthesia. The excised lesion was sent for histopathological examination. Histopathological examination revealed a polypoidal neoplasm composed of closely packed dilated capillary-sized vessels arranged into lobules by thin fibrous septae. These are suggestive of LCH.

  Case 4 Top

A 42-year-old female patient reported to our OPD with complaint of swelling on the left side of the buccal mucosa since two months. Extraorally, no facial asymmetry was noted and lymph nodes were not palpable [Figure 4].
Figure 4: Intraoral view of the lesion on the left side of buccal mucosa

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Intraoral examination revealed a duplet of mildly raised dark blue swelling on the left side of the buccal mucosa, each of a size approximately 1 × 1cm. Swelling was not associated with pain. On palpation, the lesion was soft, blanched on application of pressure, and bled on provocation. The lesion was diagnosed as cavernous hemangioma. The lesion was treated by using sclerosing agent in two sittings, after which there was a complete resolution.

  Case 5 Top

A four-year-old female patient presented to the department with the complaint of reddish discoloration on the right side of the face above the upper lip since childhood that was not associated with pain and did not blanch on compression. It was diagnosed as capillary hemangioma. The patient was under regular follow-up, and treatment was planned after the involution phase of the lesion [Figure 5].
Figure 5: Lesion above the upper lip

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  Discussion Top

Hemangiomas are considered as hamartomas. The term “hamartoma” refers to the excessive overgrowth of cells and tissues localized to the respective organ in which it occurs. They constitute about 7% of all the benign tumors arising in infancy and childhood.[9] The WHO has classified hemangiomas into five histological types: (1) cavernous, (2) capillary, (3) epithelioid, (4) histiocytoid, and (5) sclerosing.

The International Society for the Study of Vascular Anomalies (ISSVA) has classified vascular anomalies into vascular tumors and vascular malformations on the basis of radiological, clinical, pathological, and behavioral characteristics and it revised its classification in May 2018.

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Clinically, hemangiomas can be classified as: peripheral and central type. Peripheral hemangiomas arise from the vessel in the periosteum with secondary bone involvement. Central hemangiomas destroy the bony cortex by initially developing in the spogiosum.[10] According to the size of the blood vessels, they are classified as capillary and cavernous hemangiomas. Cavernous hemangiomas are characterized by large blood vessels with an epithelial lining. Capillary hemangiomas are those with small blood vessels with fattened epithelial cells.[11] There are various theories such as the placental theory, estrogen signaling theory, hypoxia theory, theory of angiogenesis, and genetic theory explaining the pathogenesis and origin of hemangiomas. The placental theory put forward by North et al. explained that hemangiomas occur in infants whose mothers have placental abnormalities such as preeclampsia and placenta previa with the expression of GLUT1, Lewis Y antigen, and type III iodothyronine.[12]

The estrogen signaling theory states that the increased levels of estrogen during the perinatal period stimulate the endothelium to induce hemangioma.[13] The hypoxic theory explains that hemangiomas develop as a result of the proliferation of the endothelial progenitor cells due to the hypoxic environment.[14] According to the genetic theory, hemangiomas are considered to have an autosomal dominant trait whereby the gene pericenter is probably at chromosome 5q, and this may be passed from mother to child.[15] Hemangiomas can appear to be red to blue in color depending on their depth. Superficial ones will be red and will blanch on pressure, whereas the deeper ones will have a bluish tint.[16]

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Cavernous hemangiomas are mostly seen in adulthood. They are less common than the capillary hemangiomas. They are congenital and are characterized by large blood vessels with dilated vascular spaces.[15] Capillary hemangiomas of the oral cavity account for only 0.5% to 1%. They gradually increase in size initially and later regress. Buccal mucosa is a frequent site for the intraoral capillary hemangiomas.[17] Capillary hemangiomas show a female predilection, with increased incidence among Caucasians.[17] The clinical features include facial asymmetry, pain, bleeding, paresthesia, tooth mobility, tissue blanching, root resorption, and missing teeth. Clinically, capillary hemangiomas mimic pyogenic granulomas.[18]

According to ISSVA classification, pyogenic granulomas are classified as vascular tumors. There are two histological variants of pyogenic granuloma: lobular capillary hemangioma and non-lobular capillary hemangioma. The different forms of LCH described in the literature are oral mucosal, satellite, intravenous, dermal, and subcutaneous.[19] The LCH usually occurs in the second or third decades of life with a female predominance.[20] However, Bhaskar and Jacoway state that there is no sex predilection.

LCH are often seen on the gingiva. Extragingival sites include the lips, tongue, buccal mucosa, and palate. The etiology for LCH is believed to be due to staphylococcal, streptococcal infections or botrymycosis. Other reasons for LCH are believed to be as a result of minor trauma from foreign bodies, root remnants, cheek biting, bony spicules, and overhanging restorations calculus, all of which can act as a pavement for microorganisms. Such microtrauma can also interrupt the vascular system in the affected area, causing immense proliferation of the vascularity.[21] In the initial stage of LCH, they are highly vascular and are red, reddish purple in color. The older lesions are pink in appearance, as they become more collagenized over time.

The use of certain drugs has been to found to be associated with LCH.

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Intraosseous hemangiomas usually occur in the fourth and fifth decades of life, with female predominance in the ratio of 3:1. The cause can be congenital but can occur due to any prior trauma at the site of occurrence. They usually present as small, growing, bony hard lesions.[11] The differential diagnosis of hemangiomas are epulis, varicosities, OSCC, Kaposi’s sarcoma, peripheral giant cell granuloma, telangiectasia, angiosarcoma, and ossifying fibroma.[23]

The diagnosis of hemangiomas is based on clinical, radiographic, laboratory, and histopathological findings. CT scanning is the most important imaging modality as stated by Moore et al. because of better characterization and assessment of the bony cortex and periosteum. It also helps in assessing the extent of the lesion and the surrounding region.[8] MRI and ultrasound are helpful to determine any soft tissue lesion or the flow of the respective vascular malformation.[22] Other investigations that are used to diagnose hemangioma are color doppler, dynamic gadolinium-enhanced MRI, RBC-tagged Tc-99m, and SPECT. Radiographically, intraosseous hemangiomas can present as multilocular radiolucency, resulting in “honey-comb,” “soap bubble,” or “sunburst” appearance depending on the size of loculations and the expansile nature of the lesion.[24],[25]

Laboratory tests are done to evaluate the presence of glucose transporter-1 (GLUT-1), vascular endothelial growth factor, insulin-like growth factor, and tissue growth factor-beta to differentiate hemangioma from other vascular lesions.[25] Various factors such as the age of the patient, size, site, and extent of the lesion determine the type of treatment to be adopted.

Different treatment modalities include sclerotherapy, cryotherapy, radiotherapy, steroids, interferon alpha, beta blockers, laser photocoagulation, and surgery.[26]

Local and systemic corticosteroids can be used to treat hemangiomas conservatively. Prednisolone is the paradigm for the systemic corticosteroids. Locally, steroids can be used by intralesional injection at the site in the form triamcinolone acetonide. They act by inhibiting the development of VEGF and also by causing vasoconstriction of the capillaries.[27]

Interferon alpha is indicated in lesions that do not respond to corticosteroid therapy. They are known to prevent the development of new blood vessels from the existing vessels. They are given at a dosage of 3 million U/m2 subcutaneously. However, they are neurotoxic and can cause neutropenia and spastic diplegia.[28] Propanolol, a selective beta blocker, is known to reduce the size and cause changes in color within 24–28 hours of administration. Sclerotherapy helps in the obliteration of the vessels but in high-flow lesions, the sclerosing agents can be displaced, making it ineffective.[8] The different sclerosing agents used are sodium tetra decylsulphate, 5% phenol, sodium morrhuate, nitrogen mustard, boiling water, sodium psylliate, sodium citrate, invert sugar, absolute alcohol, hypertonic saline, and hypertonic dextrose.[29] The disadvantages are the necrosis, sloughing, and anaphylaxis in response to the sclerosants.

Laser photocoagulation with argon, carbon dioxide, and neodymium: yttrium aluminum-garnet (Nd:YAG) laser are helpful for excision due to less chances for hemorrhage. Lasers are indicated for superficial lesions that are at the involuting stage. Lasers can be used in conjugation with other drugs.[30] However, the main disadvantages are the formation of the superficial ulcerations and scarring.[31] Radiotherapy is mainly used in lesions; access is difficult and has been found to cause scar formation. Malignant transformation of the lesion after radiotherapy is unusually possible.

The most recommended treatment modality is surgery. It is usually indicated whenever there is an increasing lesion and for esthetic purposes.[31] Complete surgical excision is the backbone for the treatment of hemangiomas followed by embolization or ligation. Two types of excision that are often paced are the circular and lenticular excisions. Lenticular excisions result in an enlarged linear scar. Circular excisions produce a less distorted scar. In case of intraosseous type of hemangiomas, complete excision along with the resection of the adjacent normal bone is advocated. Reconstruction to repair the defect is done for better cosmetic results and for functional rehabilitation. Reconstruction of the facial region is challenging, and it is difficult to maintain the contour of the facial region. To overcome such difficulties, patient-specific implants can utilize rapid prototyping and stereolithographic models. Reconstruction can be done by using autogenous grafts such as the calvarium, iliac, ribs, or fatty tissue. The main drawback was the difficulty in convolution of the implants. Alloplastic materials such as hydroxyapatite and silicon can be used.

  Conclusion Top

Since hemangiomas often occur in the head and neck region, maxillofacial surgeons should be accustomed and acquainted with the different variants, clinical and radiological features of hemangioma to arrive at a prompt diagnosis to adopt and perform a finer and superlative respective therapy. Hemangiomas should be included in the differential diagnosis of kindred lesions in the oral and maxillofacial region.

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.

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Conflicts of interest

There are no conflicts of interest.

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

  [Table 1], [Table 2], [Table 3]


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