| Abstract|| |
Intramuscular hemangiomas are rare benign neoplasms accounting for <1% of all hemangiomas and <20% are found in head and neck area. The muscle most frequently involved is the masseter muscle and very few cases have been reported for the occurrence of these hemangiomas in the buccinator muscle. Here, we are presenting a case report of intramuscular hemangioma occurring in the buccinator muscle in a 22-year-old girl.
Keywords: Buccinator, hemangioma, intramuscular, vascular malformation
|How to cite this article:|
Doddanna SJ, Dawar G, Rallan NS, Agarwal M. Intramuscular cavernous hemangioma: A rare entity in the buccinator muscle. Indian J Dent Res 2014;25:813-5
Vascular lesions have been known by a number of terms which are classified as hemangiomas or vascular malformations.  They are benign vascular tumors occurring commonly in infancy and childhood, but few may also be present since birth or even develop in adults.  They show a notorious higher prevalence in females than males (3:1-7:1).  Head and neck are the common sites,  accounting 60% of all the cases. Hemangiomas of the oral cavity are not common pathologic entities. In the oral cavity, the most common sites are the lips, buccal mucosa, tongue, and palate. About 80% hemangiomas occur as single lesions and multiple tumors are seen in only 20% of affected tumors. 
|How to cite this URL:|
Doddanna SJ, Dawar G, Rallan NS, Agarwal M. Intramuscular cavernous hemangioma: A rare entity in the buccinator muscle. Indian J Dent Res [serial online] 2014 [cited 2019 Oct 19];25:813-5. Available from: http://www.ijdr.in/text.asp?2014/25/6/813/152211
The term hemangioma describes many lesions that bear little relationship to each other, except the fact being involved with vessels.  One special form of hemangioma is intramuscular hemangioma, which is a rare benign congenital neoplasm accounting for <1% of all haemangioma, and <20% of those found in the head and neck area.  The masseter muscle is the most frequently involved site in the head and neck area accounting for 5% of all intramuscular haemangioma.  Here, We are reporting a rare case of intramuscular hemangioma of the buccal mucosa in a 22-year-old girl.
| Case report|| |
A 22-year-old female patient reported with a chief complaint of swelling on left side of her face since 8 months. She was apparently asymptomatic 8 months back when she noticed a small swelling on her left side of face, which has gradually increased up to the present size. Initially there was no pain associated with it but she has experienced mild pain on pressure since 1 month. There was neither a relevant history of trauma nor any significant medical and dental history was found. Family history was also noncontributory.
Extraoral examination revealed facial asymmetry with diffuse indistinct swelling on the left middle facial region with normal overlying skin [Figure 1]. A comprehensive intraoral examination revealed an absolutely normal buccal mucosa on inspection. However, on palpation a solitary, diffuse swelling measuring approximately 1 cm × 2 cm was found on the middle third area of left buccal mucosa. The swelling was mildly tender and soft in consistency. Left submandibular lymph nodes were palpable, mobile, tender and soft in consistency. A provisional diagnosis of fibroma was made.
|Figure 1: Extraoral photograph of the patient showing diffuse swelling on the left mid facial region|
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Complete blood count was within normal ranges. No lesion was found on panoramic radiograph. Ultrasonography and Color Doppler imaging revealed a small well defined hypoechoeic, mobile, oval solid lesion measuring 1.9 cm × 1.6 cm within the muscular plane in the region of left cheek with no significant vascularity within and around the lesion and the impression of a benign lesion was given. Fine needle aspiration cytology of the lesion revealed the sea of red blood cells (RBCs) and few inflammatory cells with the impression of noninflammatory lesion. The lesion was surgically excised under local anesthesia. Gross specimen revealed a solitary soft tissue, bluish white in color, irregular in shape measuring about 1.9 cm × 1.2 cm × 1.0 cm [Figure 2]. But to our surprise, histopathological examination revealed numerous large dilated endothelial lined blood sinuses filled with RBCs [Figure 3]. Correlating with the clinical features, definite diagnosis of intramuscular cavernous hemangioma of the left buccal mucosa was given.
|Figure 3: Photomicrograph of the H and E, stained section showing numerous large dilated endothelial lined blood sinuses filled with red blood cells|
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| Discussion|| |
Hemangioma are benign proliferative vascular lesions characterized by increased endothelial cell turnover that usually appears after birth, grow rapidly, and then involute over the years. Within the wide spectrum of vascular lesions, intramuscular hemangioma are very rare, accounting for <1% of all hemangiomas. The masseter muscle is the most frequent muscle, accounting for 5% of all intramuscular hemangioma. The trapezius, periorbital, sternocleidomastoid, and temporalis muscles follow the masseter muscle in frequency.  Very few cases of the cavernous hemangioma of the buccinator muscle has been reported.  Here, we are also adding our case of intramuscular hemangioma occurring in the buccinator muscle.
In 1843, Liston was the first to report a case of intramuscular cavernous hemangioma naming it as an "erectile tumor." Intra muscular hemangiomas are considered hamartomatous lesions and thought to arise from abnormal embryonic rests. 
Developmentally, intramuscular hemangiomas represent congenital vascular malformations. They generally develop in patients during the first three decades of life with no gender predispositions. 
Most hemangioma are recognized clinically and do not require any investigation. The predominant complaint is presence of a slowly enlarging mass.  Intramuscular hemangiomas rarely display any clinical symptoms or signs that reveal their vascular nature. They usually presents with a normal overlying skin, although there may be occasional reddish-blue discoloration. Thrills, bruits, compressibility, and pulsation are usually absent; however, pain can be present.  These features were also consistent with the present case.
Intramuscular hemangiomas represent a challenge on diagnosis because they exhibit few signs on clinical examination. Often times the extent of the lesion is not clinically apparent on examination and imaging techniques frequently define more extensive lesions than suspected.  Definitive preoperative diagnosis has been reported in <8% of cases. 
Sonography is the first-line imaging procedure for patients with soft tissue swellings. Color Doppler sonography is especially useful to demonstrate the vascular structures in and around the masseter muscle and has the potential to evaluate the pathological changes. Hemangioma could be distinguished from other soft tissue lesions by the features of abundant vascularity and high blood flow velocity. When performing sonography on soft tissue masses in the head and neck, the presence of a color Doppler signal in a well-defined hypoechoic mass with heterogeneous echotexture should raise the possibility of hemangioma.  However in the present case, no significant vascularity was found within and around the lesion on Ultrasonography and Color Doppler imaging and the impression of a benign lesion was given.
Arteriography is diagnostic in most cases, but may fail to show the feeding arteries of an intramuscular hemangioma if it is supplied by small arteries with slow blood Flow. Although some authors believe that fine needle aspiration is commonly nondiagnostic, showing the presence of a purely bloody specimen may be considered strongly suggestive for intramuscular hemangioma as seen in our case. Fine needle aspiration should also be performed to aid in the exclusion of other soft tissue tumors. 
Allen and Enzinger classified them as large vessel (>140 mm in diameter) small vessel (<140 mm in diameter) and mixed vessel types. They correspond to cavernous, capillary, and mixed type of hemangiomas respectively. The above classification is useful and correlates well with clinical presentation and recurrence rates. 
Management of intramuscular haemangioma should be individualized according to its size, growth rate, anatomic accessibility of the tumor, age of the patient and cosmetic and functional considerations. Complete surgical resection is preferred as a treatment of choice, but local recurrence rates have been reported as 18% and 19% in two different studies. 
| Conclusion|| |
Cavernous hemangioma of the muscle although rare, should be considered in the differential diagnosis of soft tissue tumors of the orofacial region.
| References|| |
Dilsiz A, Aydin T, Gursan N. Capillary hemangioma as a rare benign tumor of the oral cavity: A case report. Cases J 2009;2:8622.
Jeddy N, Duraiyan JP, Palanivelu S, Ramamoorthy A. A giant compound hemangioma of lower lip. J Oral Maxillofac Pathol 2012;16:438-40.
Avila ED, Molon RS, Conte Neto N, Gabrielli MA, Hochuli-Vieira E. Lip cavernous hemangioma in a young child. Braz Dent J 2010;21:370-4.
Shafer W.G, Hine M.K, Levy B.M. Shafer's Textbook of Oral Pathology. 7 th
ed . New Delhi: Elsevier Publication; 2012.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofaical Pathology. 3 rd
ed. Philadelphia, Toronto: W.B. Saunders; 2008.
Narwal A, Sen R, Singh V, Gupta A. Masson's hemangioma: A rare intraoral presentation. Contemp Clin Dent 2013;4:397-401.
Afsar FS, Oziz E, Hamdioglu Y, Karasoy I, Uguz B. Intramuscular haemangioma of the masseter muscle in a 9-year-old girl. Acta Angiol 2007;13:42-6.
Kushraj T, Chatra L, Shenai P, Rao PK, Shetty SR. Cavernous hemangioma of the buccinator muscle-MRI features. Pac J Med Sci 2013;12:65-70.
Zengin AZ, Celenk P, Sumer AP. Intramuscular hemangioma presenting with multiple phleboliths: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;115:e32-6.
Narayanan CD, Prakash P, Dhanasekaran CK. Intramuscular hemangioma of the masseter muscle: A case report. Cases J 2009;2:7459.
Sunitha Jagalur Doddanna
Department of Oral Pathology and Microbiology, Teerthanker Mahaveer Dental College and Research Centre, Teerthanker Mahaveer University, Bagarpur, Moradabad, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3]