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Case Report

Hemangiomas of the Oral and Maxillofacial Region: A Series of

Five Cases with Literature Review
Sooraj Soman, T. Ajay Das, Leslie Sara Mathew Kalathil, Sachin Aslam, Tom Thomas, Vijayakumar Depesh1
Department of Oral and Maxillofacial Surgery, MES Dental College and Hospital, Perinthalmanna, Kerala, 1Department of Oral and Maxillofacial Surgery, KMCT Dental
College, Mukkam, Kerala, India

Abstract
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

Introduction are a histological variant of pyogenic granuloma; these

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)
Date of Submission: 30 October 2020,
Date of Acceptance: 22 November 2020,
Date of Web Publication: 29 December 2020
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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]
Address for correspondence: Dr. Sooraj Soman,
Department of Oral and Maxillofacial Surgery, MES Dental
College and Hospital, Perinthalmanna, Malappuram 679321, Kerala, India.
E-mail: drsoorajsoman@gmail.com
This is an open access journal, and articles are distributed under the terms of the
Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows
others to remix, tweak, and build upon the work non-commercially, as long as
appropriate credit is given and the new creations are licensed under the identical terms.
For reprints contact: reprints@medknow.com

How to cite this article: Soman S, Das TA, Kalathil LS, Aslam S,
DOI: Thomas T, Depesh V. Hemangiomas of the oral and maxillofacial
10.4103/INJO.INJO_46_20 region: A series of five cases with literature review. Int J Oral Care Res
2020;8:97-102.

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Soman, et al.: Hemangiomas of the oral and maxillofacial region
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
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].
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
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
98 International Journal of Oral Care and Research ¦ Volume 8 ¦ Issue 4 ¦ October-December 2020
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].
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
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].
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
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].
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
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
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Soman, et al.: Hemangiomas of the oral and maxillofacial region

was not associated with pain and did not blanch on Clinically, hemangiomas can be classified as: peripheral
compression. It was diagnosed as capillary hemangioma. and central type. Peripheral hemangiomas arise from the
The patient was under regular follow-up, and treatment vessel in the periosteum with secondary bone involvement.
was planned after the involution phase of the lesion Central hemangiomas destroy the bony cortex by initially
[Figure 5]. developing in the spogiosum.[10] According to the size
of the blood vessels, they are classified as capillary and
cavernous hemangiomas. Cavernous hemangiomas are
Discussion characterized by large blood vessels with an epithelial
Hemangiomas are considered as hamartomas. The term lining. Capillary hemangiomas are those with small
“hamartoma” refers to the excessive overgrowth of cells blood vessels with fattened epithelial cells.[11] There are
and tissues localized to the respective organ in which various theories such as the placental theory, estrogen
it occurs. They constitute about 7% of all the benign signaling theory, hypoxia theory, theory of angiogenesis,
tumors arising in infancy and childhood.[9] The WHO has and genetic theory explaining the pathogenesis and origin
classified hemangiomas into five histological types: (1) of hemangiomas. The placental theory put forward by
cavernous, (2) capillary, (3) epithelioid, (4) histiocytoid, North et al. explained that hemangiomas occur in infants
and (5) sclerosing. whose mothers have placental abnormalities such as
preeclampsia and placenta previa with the expression of
The International Society for the Study of Vascular GLUT1, Lewis Y antigen, and type III iodothyronine.[12]
Anomalies (ISSVA) has classified vascular anomalies
into vascular tumors and vascular malformations on The estrogen signaling theory states that the increased levels of
the basis of radiological, clinical, pathological, and estrogen during the perinatal period stimulate the endothelium
behavioral characteristics and it revised its classification to induce hemangioma.[13] The hypoxic theory explains that
hemangiomas develop as a result of the proliferation of the
in May 2018.
endothelial progenitor cells due to the hypoxic environment.[14]
Vascular tumors Vascular malformations According to the genetic theory, hemangiomas are considered
Infantile hemangiomas Slow (low) flow to have an autosomal dominant trait whereby the gene
 Focal pericenter is probably at chromosome 5q, and this may be
 Segmental passed from mother to child.[15] Hemangiomas can appear to
 Indeterminate be red to blue in color depending on their depth. Superficial
Congenital hemangiomas Capillary malformations (CM) ones will be red and will blanch on pressure, whereas the
 Rapidly involuting congenital   Port-wine stain deeper ones will have a bluish tint.[16]
Hemangioma (RICH)
 Noninvoluting congenital  Telangiectasia Syndromes associated with hemangioma[4]
Hemangioma (NICH) Rendu–Osler–Weber syndrome
 Angiokeratoma Sturge–Weber–Dimitri syndrome
Tufted angioma Venous malformations (VM) Kasabach–Merritt syndrome
  Common sporadic VM Maffucci syndrome
  Bean syndrome von Hippel–Lindau syndrome
 Familial cutaneous and mucosal Klippel–Trenaunay–Weber syndrome
VM (VMCM) PHACES syndrome
 Glomuvenous malformation
(GVM) or glomangioma Cavernous hemangiomas are mostly seen in adulthood.
  Maffucci syndrome They are less common than the capillary hemangiomas.
Pyogenic granuloma Lymphatic malformation (LM) They are congenital and are characterized by large
 Lymphedema blood vessels with dilated vascular spaces.[15] Capillary
  Lymphangioma circumscriptum hemangiomas of the oral cavity account for only 0.5%
  Lymphangioma cavernosum to 1%. They gradually increase in size initially and later
  Lymphangioma cysticum regress. Buccal mucosa is a frequent site for the intraoral
Dermatologic acquired vascular Fast (high) flow capillary hemangiomas.[17] Capillary hemangiomas
tumors show a female predilection, with increased incidence
  Arterial malformation (AM) among Caucasians.[17] The clinical features include facial
  Arteriovenous fistula (AVF) asymmetry, pain, bleeding, paresthesia, tooth mobility,
 Arteriovenous malformation tissue blanching, root resorption, and missing teeth.
(AVM)
Clinically, capillary hemangiomas mimic pyogenic
Kaposiform Complex combined vascular
hemangioendothelioma malformations
granulomas.[18]
Spindle cell According to ISSVA classification, pyogenic granulomas
hemangioendothelioma are classified as vascular tumors. There are two histological

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Soman, et al.: Hemangiomas of the oral and maxillofacial region

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
Drugs associated with LCH[22]
• ANTI-NEOPLASTICS: e.g. 5 Fluorouracil, docetaxel, ramucirumab
• IMMUNOSUPPRESSANTS: e.g. cyclosporine, etanercept, mTOR
inhibitors
• ANTI-RETROVIRALS: e.g. Indinavir
• RETINOIDS
•EPIDERMAL GROWTH FACTOR RECEPTOR INHIBITORS
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/

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.
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 Figure 1: CT scan showing lesion involving the left zygomatic bone

      
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Soman, et al.: Hemangiomas of the oral and maxillofacial region

Figure 2: Intraoral view of the lesion on the right buccal mucosa Figure 4: Intraoral view of the lesion on the left side of buccal mucosa

Figure 3: Lesion on the lip Figure 5: Lesion above the upper lip

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

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Soman, et al.: Hemangiomas of the oral and maxillofacial region
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
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.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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