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C A S E R E P O R T
Oral intramuscular hemangioma: Report of three cases
Carlos M. Lescura1 | Bruno A. B. de Andrade2 | Kelly T. Bezerra2 |
Michelle Agostini2 | Milagros V. A. Ankha1 | Felix de Castro3 | Yasmin R. Carvalho1 |
Mário J. Romañach2 | Ana Lia Anbinder1
1Department of Bioscience and Oral
Diagnosis, Institute of Science and
Technology, São Paulo State University
(UNESP), São José dos Campos, Brazil
2Department of Oral Diagnosis and Pathology,
School of Dentistry, Federal University of Rio
de Janeiro (UFRJ), Rio de Janeiro, Brazil
3Policlin Hospital, São José dos Campos, Brazil
Correspondence
Ana Lia Anbinder, Avenida Engenheiro
Francisco José Longo, 777, 12245-000,
São José dos Campos, SP, Brazil.
Email: ana.anbinder@unesp.br
Abstract
Intramuscular hemangioma (IMH) represents less than 1% of all hemangiomas. In the
head and neck region, it occurs mostly in the masseter, temporalis and
sternocleidomastoid muscles. Despite its infiltrative growth pattern and several wor-
risome histological features, such as increased mitotic activity, plumpness of the
nuclei, intraluminal papillary projections or perineural infiltration, the lesion is benign,
and complete surgical excision is the preferred treatment for such oral lesions.
Herein, we report three rare cases of IMH in the tongue and lip, discuss the clinical
and histological aspects, and review the literature regarding this lesion.
K E YWORD S
intramuscular hemangioma, vascular malformations, vascular tumors
1 | INTRODUCTION
Intramuscular hemangioma (IMH) is a vascular benign proliferation that
can occur within any muscle, particularly those of the lower limbs.1,2
IMH is considered an uncommon lesion, accounting for less than 1% of
all hemangiomas. In the head and neck region, the masseter is the most
commonly affected muscle, followed by the temporalis and
sternocleidomastoid.3 Clinically, it presents as a mass with slow, infiltra-
tive growth, of variable size and coloration.1 Histologically, it consists of
a mixture of capillary and cavernous spaces infiltrating skeletal muscle
fibers, with variable amount of fatty tissue.2 Magnetic resonance imag-
ing (MRI) seems to be the best imaging modality to evaluate IMH, which
is usually treated by surgical excision with clear margins because of its
infiltrative nature and possible recurrent behavior.
After an electronic search performed in January 2019 in the
PubMed/Medline, Scopus, Web of Science and Google Scholar data-
bases using the key words “hemangioma,” “intramuscular,” “tongue,”
“oral,” “orbicularis oris muscle” and “lip,” 31 cases of intraoral IMH
were identified in the English-language literature to date, 21 of them
located in the tongue and lips (Table 1). The aim of this study is to
report three additional cases of IMH in the oral cavity, one in the
tongue and two in the lips.
2 | CASES REPORT
The first patient was a 59-year-old woman referred to a dentist for
evaluation of an asymptomatic slow-growing swelling in the dorsum
of the tongue present for 1 year, causing difficulties in phonation.
The intra-oral examination showed a submucosal nodule in the
middle-third dorsal tongue, measuring approximately 2 cm, slightly
paler than the adjacent mucosa and firm on palpation. The past med-
ical history and extra-oral examination were unremarkable and the
patient denied any prior trauma. MRI, T1-weighted, depicted a nod-
ule on the left side of the tongue, ranging from an intermediate sig-
nal to a hyposignal, measuring 14 × 26 × 13 mm (Figure 1A, B).
Considering the diagnostic hypotheses of leiomyoma or solitary
fibrous tumor, an incisional biopsy was performed. Microscopic eval-
uation showed numerous small and thin-walled blood vessels, mainly
infiltrating skeletal muscle fibers, but sometimes also into perineural
sheaths. Adipose tissue was also observed between blood vessels
and muscle fibers. The final diagnosis was of IMH and complete exci-
sion of the lesion was performed under general anesthesia, through
partial glossectomy with a surgical diode laser to control bleeding.
The gross appearance was of a well-defined solid mass of brownish
coloration and smooth surface (Figure 1C). Immunohistochemical
Received: 10 February 2019 Revised: 11 April 2019 Accepted: 18 April 2019
DOI: 10.1111/cup.13482
© 2019 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.
J Cutan Pathol. 2019;46:603–608. wileyonlinelibrary.com/journal/cup 603
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analysis was also performed for illustrative purpose, and positivity
for CD34 (QBend 10, 1:50, Dako) and smooth muscle actin (SMA,
1A4, 1:200, Dako) were found in the endothelial and muscular layer
of blood vessels, respectively (Figure 1D-G). Positivity for CD105
(SN6h, 1:500, Dako) was found in some small vessels of the sub-
epithelial region of the lamina propria and antibody D2-40 (D240,
1:50, Dako) highlighted lymphatic vessels within the lesion and in
the lamina propria. No recurrence has been detected after follow-up
of 1 year and 4 months.
The second patient was a 67-year-old man presenting an asymp-
tomatic upper lip swelling of approximately 20 years of evolution. Intra-
oral examination showed an ill-defined, normal-colored submucosal
nodule measuring 2.5 cm in the upper lip close to the right commissure
(Figure 2A). With the hypotheses of canalicular adenoma or leiomyoma,
an excisional biopsy was performed, under local anesthesia, with signifi-
cant trans-surgical hemorrhage. The gross examination showed a well-
defined solid mass with homogenous cut surface of brownish coloration.
Microscopic analysis showed numerous blood vessels of different sizes
and diameters, most of them congested, intermingled with skeletal mus-
cle fibers and adipose tissue (Figure 2B-D). The final diagnosis was of
IMH. The patient has no sign of recurrence after 6 months of follow-up.
The third patient was a 57-year-old male presenting a swelling in
the right side of lower lip semi-mucosa, with discomfort and occa-
sional bleeding only when biting the lesion. Patient reported history
of previous local trauma 5 years earlier, when he suffered an auto
accident. During intra-oral examination, a 3 cm reddish nodule with
central superficial ulcer was observed in the lower lip (Figure 3A). An
excisional biopsy was performed under general anesthesia and hemo-
stasis by electrocoagulation. Microscopical features were similar to
cases 1 and 2, and the diagnosis of IMH was established (Figure 3B-D).
The patient was lost to follow-up.
3 | DISCUSSION
The term “hemangioma” has been widely used for benign vascular prolif-
erations of different clinical behaviors and microscopic features, causing
TABLE 1 Published cases of intramuscular hemangioma located in tongue and lips
Author, year Age Gender Site Size (cm)
Tongue
Small and Nathan, 19854 48 M Ventral 4 × 2
Sund and Bang, 19905 16 M Diffuse to tongue, lower lip and cheek N/A
Gillett et al., 20036 32 F Right lateral border 1.5
Chien et al., 20057 16 M Base 3 to 4
Elston and Kobayashi, 20058 60 M Diffuse-dorsal N/A
Løes et al., 20099 43 M Right lateral border N/A
Kucuk et al., 201410 32 F Right ventral and dorsal 7 × 5 × 3
Kamatani et al., 201411 51 M Right lateral border, lower surface and base 1
Babu et al., 201412 60 F Dorsum 4 × 3
Petrovic et al., 201513 51 F Ventral and base 3.2 × 2.3 × 2.9
Kamra et al., 201614 29 M Dorsal 3 × 3
Case 01 59 F Dorsal 1.4 × 2.6 × 1.3
Lips
Ivey et al., 198015 44 M Upper lip 1.5 × 1.25
Kinni et al., 198116 72 M Upper lip 3 × 2 × 1
Sund and Bang, 19905 50 M Upper lip 1.6 × 0.9
Chan et al., 199217 9 months N/A Upper lip 1.5
Rossiter et al., 199318 12 M N/A 4 × 4
Nam and Hwang, 200719 5 M Lower lip 3 × 1.5 × 1
Silva et al., 201120 48 F Upper lip N/A
Jamshidi et al.,201421 54 M Upper lip 2 × 3 × 5
Konda et al., 201622 20 M Lower lip 2 × 2.5
Kim et al., 201723 58 F Upper lip 2.7
Case 02 67 M Upper lip 2.5
Case 03 57 M Lower lip 2
F, female; M, male, N/A, not available.
604 LESCURA ET AL.
difficulty in proper nomenclature of such lesions.24 The International
Society for the Study of Vascular Anomalies (ISSVA) classification of
2014 divides the vascular anomalies between tumor lesions and vascular
malformations.24 Hemangiomas (infantile, congenital, tufted, spindle-cell
and epithelioid) are benign tumors, unlike vascular malformations (capil-
lary, lymphatic, venous, arteriovenous or combined). The term IMH is
proposed by the World Health Organization, and only appeared in the
updated 2018 classification of ISSVA, as a provisionally unclassified vas-
cular anomaly. Despite the denomination of hemangioma, most IMH
probably represent malformations and not truly a vascular neoplasm.1,25
Oral IMH is uncommon, the tongue being a rather uncommon
head and neck site, as well as the lips.3 Including our first case, the
mean age of patients with IMH of the tongue was 41.42
± 16.10 years, with a slight preference for males (2:1.4). For IMH of
the lips, the mean age was 40.65 ± 24.65 years with male preference
(4.5:1), occurring more frequently in the upper lip. Interestingly, our
three patients were older, with a mean age of 61 at the time of diag-
nosis. IMHs usually remain asymptomatic for up to two or three
decades, when they become palpable after a sudden growth, and this
swelling is the most common initial presentation.18 Due to the
F IGURE 1 Case 1. A, Axial T1-magnetic resonance imaging (MRI) after contrast, showing lesion in left side of tongue, with intermediate
signal, more intense than the surrounding tissue, resulting from the impregnation of the contrast; B, MRI sagittal T1-weighted post-clear; lesion
with hyposignal in the tongue, with a differential aspect in relation to the surrounding tissue, which presents comparative hypersignal. C,
Macroscopic aspect of the surgical specimen, showing a solid lesion with no hemorrhagic appearance in the center. D, Numerous predominantly
capillary blood vessels that infiltrate the skeletal muscle fibers of the tongue. Hematoxylin and eosin (H&E) staining. Bar = 200 μm. E, Adipose
tissue between the blood vessels and muscle fibers, blood vessels in the perineurium (arrow). H&E staining. Bar = 90 μm. F, Immunohistochemical
reaction for the CD34 antibody, evidencing strong positivity in endothelial cells, showing numerous blood vessels between the skeletal muscle
bundles. Bar = 200 μm. G, Immunohistochemical reaction for the SMA antibody, evidencing strong positivity of smooth muscle cells in vascular
walls. Bar = 200 μm
LESCURA ET AL. 605
presence of surrounding muscle fibers, pulsations and bruits are fre-
quently absent, and mucosal coloration might remain unchanged,
making the pre-operative diagnosis difficult. Because a vascular lesion
may not be clinically suspected, there is an increased risk of bleeding
complications during biopsy procedures.4 The clinical differential diag-
nosis includes several lesions, such as salivary gland and benign mes-
enchymal neoplasms, mainly because of its non-specific clinical
features.12 The clinical diagnostic hypotheses of the published IMH
cases in tongue and lip varied from cysts (epidermoid and dermoid) to
salivary gland or mesenchymal neoplasms, as also observed in the pre-
sent cases.
The most accepted classification of IMH was made by Allen and
Enzinger in 1972, who described three histological subtypes: small
vessel (capillary), large-vessel (cavernous) and mixed subtype (cavern-
ous and capillary).2 Phleboliths or metaplastic calcification may be pre-
sent in some cases.1,11
The histological differential diagnosis of IMH includes mainly
angiosarcoma, Kaposi sarcoma, angiomatosis and infiltrating
angiolipoma.2,5 In the differentiation of angiosarcoma and IMH, some
important features present in malignancy, such as pleomorphism,
nuclear hyperchromatism, the presence of multinucleated endothelial
cells, as well as necrosis and solid growth of malignant cells, are use-
ful.2 The absence of the most common risk factors for angiosarcoma,
chronic lymphoedema and history of radiation, can also aid in the
diagnosis. Due to is similarity to some cases of angiosarcoma,
acantholytic squamous cell carcinoma may rarely be considered in the
differential diagnosis. The initial lesions of Kaposi sarcoma are com-
posed of the proliferation of numerous capillaries, but they differ from
IMH, because there is no muscular infiltration. HHV-8 immunohisto-
chemistry is also helpful in this differentiation, associated with clinical
correlations, such as endemic settings. With a histological appearance
similar to IMH, angiomatosis is a rare condition, usually presenting
during childhood, in which several segments of the body are involved
by vessels that are proliferating.1 The infiltrating angiolipoma is now
considered as an IMH with abundant adipose tissue.1,5 Because oral
lesions are often subjected to local trauma, worrisome inflammatory
reactive changes can be present, and should be also taken into consid-
eration in the differential diagnosis. Pyogenic granuloma or lobular
capillary hemangioma, a very common oral vascular lesion, is easily
differentiated from IMH, because it does not infiltrate skeletal muscle.
F IGURE 2 Case 2. A, Clinical appearance of the lesion, leading to enlargement of the upper lip. B, Macroscopic aspect of the surgical
specimen. C, Numerous blood vessels and adipose tissue infiltrate the muscle fibers. Hematoxylin and eosin (H&E) staining. Original magnification
40×. D, Numerous small blood vessels between the muscle fibers. H&E staining. Original magnification 400×
606 LESCURA ET AL.
To aid diagnosis, ancillary tests may be used. In IMH, MRI presents
better detection and delimitation of lesion extension than computed
tomography. IMH presents a hyposignal at T1 and greater signal
intensity in T2-weighted images, as well as poorly defined T1 margins
and well limited T2-weighted margins. It may also present rounded
hyposignal foci that can represent a cross-section of fibrofatty septa,
smooth musculature, ossification, or vascular channels with thrombus
formation.12
Several treatment options have already been studied, such as
cryotherapy, sclerotherapy, radiation, laser and corticosteroids, each
with their advantages and disadvantages, but the main treatment is
surgical excision.10 Local recurrence, ranging from 9% to 28%, has
been reported and is usually associated with incomplete surgical
resection.2
In conclusion, IMH is an uncommon benign vascular prolifera-
tion in the oral cavity. The clinical appearance may be non-spe-
cific, leading to unexpected bleeding complications during a
biopsy procedure. IMH may show microscopic features similar to
those observed in infiltrative vascular malignant tumors. Oral
IMH should be considered in the differential diagnosis of
swellings of the tongue and lips of older patients with history of
local trauma.
ACKNOWLEDGEMENT
The authors would like to thank Dr. Vinicius Machado and Dr. Daniel
Caetano for providing clinical pictures of patients of case 2 and
3, respectively.
ORCID
Ana Lia Anbinder https://orcid.org/0000-0003-3930-4274
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https://orcid.org/0000-0003-3930-4274
https://orcid.org/0000-0003-3930-4274
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How to cite this article: Lescura CM, de Andrade BAB,
Bezerra KT, et al. Oral intramuscular hemangioma: Report of
three cases. J Cutan Pathol. 2019;46:603–608. https://doi.
org/10.1111/cup.13482
608 LESCURA ET AL.
http://www.issva.org/UserFiles/file/ISSVA-Classification-2018.pdf
http://www.issva.org/UserFiles/file/ISSVA-Classification-2018.pdf
https://doi.org/10.1111/cup.13482
https://doi.org/10.1111/cup.13482
	Oral intramuscular hemangioma: Report of three cases
	1 INTRODUCTION
	2 CASES REPORT
	3 DISCUSSION
	ACKNOWLEDGEMENT
	REFERENCES