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Oral Pathology

Authors: Dr. Shruti Nayak, Dr.B. SivapathaSundharam, Dr. B. Kavitha.


ABSTRACT:
Intra muscular Haemangiomas (IMH) are relatively uncommon benign vascular tumors which account for less than 1% of all haemangiomas. Diagnosis of this lesion is important not only because of its rarity but also due to dangers posed by misdiagnosis and mismanagement. A rare case of a 17 year old male patient with a complaint of a recurrent swelling in the cheek region was subsequently diagnosed as an intra-muscular haemangioma and has been reported in this article.

INTRODUCTION:
Haemangioma are uncommon benign vascular neoplasms1 and they are abnormal proliferation of blood vessels that may occur in any vascularised tissue 2. It is a benign soft tissue tumor occurring most often in the skin and sub-cutaneous tissues. Haemangiomas are hamartomas rather than true neoplasms as they are generally composed of vascular spaces arising from endothelial cells and not by incorporation of nearby vascular channels. They are mostly congenital with 85 % seen in newborns and first year infants and are not commonly seen in adults 3as they regress with age.
It predominantly affects the skin and subcutaneous tissues of the trunk, followed by extremities and head and neck region (13.5-21%). In the head and neck region, it occurs very rarely in oro-facial muscles (<1%) 4,5. Surgical intervention is needed only when a symptom arises and it carries excellent prognosis.
Unlike infantile cutaneous haemangiomas, Intramuscular Haemangiomas do not regress spontaneously, and are usually detected in the second or third decade of life. Their location and unfamiliar presentation may require sonography, magnetic resonance imaging and sometimes angiography for accurate diagnosis. 6,7 The exact cause of intramuscular haemangioma has been an enigma. 8 The most accepted nomenclature for classifying intramuscular haemangioma is based on histological appearance. Allen and Enzinger in 1972 formed a classification system based on vessel size; capillary, cavernous or mixed small/large vessel types. This classification correlates well with location and prognosis. 9
Till recently only five cases of intramuscular haemangiomas have been reported in the head and neck region. It includes diagastric muscle 2, massetor muscle 2, and myolohyoid muscle1,5. We hereby report a rare case of intra–massetric haemangioma in a 17 yr old male patient in this article.

CASE REPORT:
A 17 yr old male patient presented with a complaint of swelling in the right cheek region for the past 10 years. History revealed that it started as a small painless swelling over the right side of angle of the mandible at an age of 8 years, and over a period of 10 years it has gradually increased and attained the present size of 4 x 4 cm. This solitary swelling is elliptical and located antero-inferior to the ear lobe and 1cm above the inferior border of the mandible, (Figure 1) with normal overlying skin colour without any sinus involvement. On palpation, swelling is soft, non-tender, lobulated, but becomes firm and increases in size on clenching with no evidence of any nerve involvement. Intra Orally no swelling was observed and the buccal mucosa showed normal colour and texture when compared to the opposite side.
Ultrasonic investigation of the soft tissue swelling showed, non-homogeneous, ill-defined, mixed echogenic lobulated mass with fluid collection over right masseter muscle at its insertion. The mass measured 2.9cm x 2.2cm, and a dense calcification measuring 5 x 3cm was seen within the lesion.
Previous surgical history (2 biopsies) done under local anesthesia reveal the lesion as myoma of right masseteric muscle. Due to recurrence of the lesion a detailed surgery was planned, patient was admitted and the lesion was excised under general anesthesia. After submandibular incision, platysma was identified and subplatysmal dissection was done upto the lower border of mandible. After the facial vessels were identified and ligated, superior fibers of masseter muscle was identified, excised and sent for histopathological examination.
The histopathologic examination of the H and E stained soft tissue section showed muscle fibers in both cross section and longitudinal sections, with areas of prominent vascularity with numerous endothelial lined blood capillaries, arterioles and venules of variable sizes and shapes (Figure 2A, 2B, and 3). Sections were highly vascular with extravasated RBCs and extensive areas of hemorrhage (Figure 4). Areas of muscular degeneration in varying stages were also evident (Figure 5). Based on these findings, the lesion was diagnosed as intramuscular haemangioma.

Figure-1--Lobulated-swellin Figure-2-A-and-2-B--H-and-E
Figure1:Lobulated swelling located antero-inferior to the ear lobe and 1cm above the inferior border of the mandible.

Figure 2A and 2B: H and E, 10X showing numerous arterioles and venules of variable sizes and shapes.


Figure-3--H-and-E-40X-show Figure-4--H-and-E-10X-show Figure-5--H-and-E-10X-show
Figure 3: H and E, 40X showing a large sized vein in high power.

Figure 4: H and E, 10X showing extravasated RBC`s and extensive areas of hemorrhage.

Figure 5: H and E, 10X showing areas of muscle degeneration in varying stages.


DISCUSSION:
Haemangiomas are benign proliferative vascular lesions characterized by increased endothelial turn over. These tumors usually appear after birth, grow rapidly and involute over the years within the spectrum of vascular lesions. Intra muscular haemangioma are very rare, accounting for less than 1% of all Haemangiomas, and less than 20% of these are found in the head and neck area. The masseter muscle is most frequent muscle accounting for 5% of all intramuscular haemangioma. The trapezius, periorbital, sternocleidomastoid and temporalis muscle follow the masseter muscle in frequency. The tongue extra ocular and posterior neck muscles have also been reported to be involved with haemangioma with less frequency. 10 Due to their fibrovascular nature intra muscular haemangiomas have a rubbery, firm texture and vascular bruits or thrills are infrequent. The lesion can be moved from side to side but not along the direction of the muscle fibres and there is rarely any overlying skin discolouration as seen in other forms of haemangiomas. 1
The first case of intra muscular haemangioma of masseter muscle was reported by Listen in 1843, subsequently other reports have accumulated to a total of 457, of these 63(13.8%) involved head and neck and 23(5.0%) involved masseter muscle. 11,12 Intramuscular haemangioma of masseter muscle shows a definite male predominance among the reported cases. In contrast Scott’s review of intramuscular haemangioma in general revealed an equal sex distribution. The tumor develops before the age of 30 years in approximately 90% of cases, so due to this the etiological role of either congenital origin or possibility of trauma is playing a role remains unresolved. 11 The cause of intramuscular haemangioma is unknown. Several theories have been proposed, but the most likely explanation is that the intramuscular hemangioma is a congenital mass, arising by abnormal embryonic sequestrations, similar to congenital arteriovenous malformations. 12
Intra muscular haemangiomas are non metastasizing benign congenital tumors that, after remaining unrecognized for long periods, may suddenly start to grow in second or third decade of life .5 More than 90% of Intra muscular haemangiomas are detected before the fourth decade.7 A possible hormonal role in growth of intramuscular haemangioma was speculated, but no specific data was available to prove this hypothesis. They are usually asymptomatic until a growth spurt occurs at which time pain occurs in about 50% of cases. A palpable fluctuant swelling or firm mass is present in up to 98% of cases. 10
Traditionally the haemangiomas has been classified into capillary and cavernous types. Now recently Allen and Enzinger examined 89 intra muscular haemangiomas and found that they can be divided according to the size of the vessels predominantly into small- vessels or capillary type which comprised vessels of less than 140 μ diameter, large- vessel or cavernous type which comprised vessels of more than 140 μ diameter and mixed type which consists of both small and large vessels. 1,11 About 30% of small- vessel intra muscular haemangiomas are found in head and neck and tend to have short clinical history compared to 19% of large-vessel and 5% of mixed types.1 The mixed type shows greater tendency for local recurrence (28%) and the large vessel type, the least (9%). To date no reasons have been given for the recurrence rate of mixed type. 11
The diagnosis of intra muscular haemangioma requires a high index of suspicion whenever a mass of soft tissue density is encountered in the region of skeletal muscle in a young adult, haemangioma should be considered in the differential diagnosis. 10 Magnetic resonance imaging (MRI) has been shown to provide better detection and delineation of the extent of IMH than CT. The MRI is superior because of its multiplanar capabilities and the distinct contrast between normal muscle and the IMH. Intramuscular haemangiomas are characteristically much brighter on T2- than on T1-weighted images because of the increased free water present within the stagnant blood in the larger vessels.
A clear distinction, however, between capillary and cavernous types is often not possible. Magnetic resonance imaging findings suggestive of IMH include: (i) high-signal intensity on both T1- and T2-weighted images; (ii) serpinginous pattern, septated-striated high-signal channels and curvilinear areas of low intensity consistent with vascular spaces; (iii) focal heterogeneities representing areas of thrombosis, fibrosis or calcification; and (iv) adjacent focal muscular atrophy. Arteriography is useful as a preoperative embolisation of feeding vessels which enhances homeostasis. Fine-needle aspiration cytology in most reported series is frequently non-diagnostic. 9
The differential diagnosis of mass in masseter includes congenital cysts, muscle fibre herniation, lymphadenopathy, cystic hygroma, sialocele of parotid duct and various parotid and muscle neoplasm. Several forms of therapy have been advocated for the various types of haemangiomas, including cutaneous, cavernous or capillary, subglottic and intramuscular types. The management has ranged from steroids to injection of sclerosing agents, radiation therapy, and surgical excision. Recently there have been reports of pre operative embolisation of haemangiomas with muscle fragments as a technique to decrease intraoperative blood loss. It is unlikely that these procedures will be indicated before removal in cases of small muscle haemangiomas such as those presenting because of small caliber of feeding vessels. The accepted optimal treatment of a muscle haemangioma is its total excision with a surrounding cuff of normal muscle. Masseter muscle haemangioma have been approached by an intra oral route, by a preauricular incision with a superficial parotidectomy, and by a pre auricular incision with development of flap lateral to it and preserving parotid gland. But certain factors must be considered before selecting the surgical approach. The intraoral route affords relatively poor exposure for adequate tumor removal and risks injury to the facial nerve. 12 Local recurrences occur in approximately 18% of IMH, usually as a result of incomplete surgical resection. Regional and distant metastasis has not been reported. 9

CONCLUSION:
Intra muscular haemangioma may start to grow in childhood and should be considered in the differential diagnosis of isolated muscle enlargement. MRI and colour Doppler Sonography are very helpful in diagnostic workup and the treatment of choice should be individualized in view of the clinical status of the patient.

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