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Table of Contents   
CASE REPORT  
Year : 2015  |  Volume : 26  |  Issue : 1  |  Page : 96-100
Nonsurgical management of vascular malformation of masseter


1 Department of Oral Medicine and Radiology, Rajah Muthiah Dental College and Hospital, Annamalai University, Chidambaram, Tamil Nadu, India
2 Consultant Radiologist, Saravana Scans, Chennai, India

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Date of Submission20-Dec-2013
Date of Decision25-Dec-2013
Date of Acceptance19-Feb-2015
Date of Web Publication11-May-2015
 

   Abstract 

Intramuscular vascular anomalies are rare congenital hamartomatous lesions. Less than 1% of these occur in skeletal muscle out of which 15% arise in head and neck musculature. In the head and neck region, masseter muscle is the most common site. It accounts for about 5% of intramuscular vascular malformations. They are present from birth but are clinically apparent during infancy and childhood and occasionally during adulthood. Due to its location it is often mistaken for a parotid swelling. The usual treatment of choice is surgical excision with a margin. This is associated with loss of motor function, hemorrhage, nerve damage. Intralesional sclerotherapy, embolization are nonsurgical alternatives for treatment of slow flow venous malformations. Sclerotherapy can be used solely in multiple sittings or as an adjunct to surgery. This article presents a case report of a 28-year-old male with recurrent intramuscular vascular malformation in the masseter muscle, which was successfully treated by ethanol sclerotherapy.

Keywords: Ethanol sclerotherapy, intramuscular vascular malformation, masseter muscle

How to cite this article:
Jayaraman V, Austin RD, Kannan S. Nonsurgical management of vascular malformation of masseter. Indian J Dent Res 2015;26:96-100

How to cite this URL:
Jayaraman V, Austin RD, Kannan S. Nonsurgical management of vascular malformation of masseter. Indian J Dent Res [serial online] 2015 [cited 2019 Nov 13];26:96-100. Available from: http://www.ijdr.in/text.asp?2015/26/1/96/156824
Vascular malformations are often mistaken for tumors because of their similar clinical presentation and dubious nomenclature. [1] Hemangiomas are tumors of infancy characterized by endothelial proliferation and regression over time. Vascular malformations have normal endothelial growth cycle, and they do not regress spontaneously. Intramuscular vascular malformations of the skeletal muscle occur commonly in the trunk and extremities probably due to the bulk of muscle in these areas. They are uncommon in the head and neck accounting for only 15%. When occurring in the head and neck, masseter muscle is the commonest site, which accounts for approximately 5% of intramuscular vascular lesions of head and neck. [2] They are congenital lesions present from birth but manifest later as enlarging soft tissue mass with or without pain. They are often mistaken for parotid or muscle related tumors. The rarity of the lesion, deep-seated location and unfamiliar presentation make it mandatory to include investigatory methods such as color Doppler ultrasound, contrast enhanced computed tomography (CT), magnetic resonance imaging (MRI) for accurate preoperative diagnosis and for delineation of extent. [3] Treatment of choice varies from nonsurgical method to surgical excision. Surgical excision is associated with complications such as flattening, facial nerve injury but nonsurgical methods such as sclerotherapy, laser therapy, electrocoagulation, radiation, etc., are effective alternatives to surgery. [4] The results are variable with recurrence rates ranging from 18% to 61%. [5] There are limited reports in the literature on nonsurgical management of intra-masseteric vascular malformation. Intralesional sclerotherapy is a safe, simple and effective alternative to surgery in the management of intra-masseteric venous malformation (VM). Though the success of the treatment is not adequate in literature, it is indeed one of the hopeful alternative for surgery. Hence, this case is presented to demonstrate effective treatment of a case of intra-massteric VM. The presented case has a successful follow-up for 2 years and still the patient shows no signs of recurrence.


   Case report Top


A 28-year-old male patient came to our outpatient department with a chief complaint of painful swelling in the right side of the face for past 2 months. The size of the swelling increased after clenching and after sleep and the size reduced on relaxing the jaws. The swelling was prominent and painful on clenching his teeth and on doing stressful work. There was no history of fever, trauma, dental complaints or trismus. Patient gave a history of similar swelling in the same site before 1-year, which was sudden in onset without history of trauma or coagulation disorder. The swelling was surgically excised, and histopathological examination revealed organized hematoma surrounded by fibroblasts and inflammatory cells. Hence, it was diagnosed as intramuscular hematoma. Patient doesn't have any other relevant medical or family history. Patient was apparently normal for next 10 months then he again developed a similar swelling in the same site without any obvious causative history.

On extra oral examination, there was a single diffuse dome shaped swelling present on the right side of the face near the angle of the mandible region. The swelling was 1 cm × 1 cm in size. The size of the swelling increased and became more prominent on clenching [Figure 1]. The skin over the swelling showed previous surgical scar. There was no discoloration, inflammation or ulceration. On palpation, the swelling was not warm, mildly tender. The swelling without clenching was diffuse, soft in consistency freely mobile and partially compressible. On clenching the swelling was more defined, circular in shape, with well-defined borders, rubbery to firm in consistency and was fixed to the masseter muscle. Pulsation was felt at the center of the mass. No bruit or thrill was noted. Intraoral findings were not contributory. The buccal mucosa appeared to be normal. Regional lymph nodes were not enlarged. No cranial nerve deficit was noted. Based on the previous history and clinical features the case was provisionally diagnosed as intramuscular hematoma. Differential diagnosis like intramuscular vascular malformation and lymphangioma were also considered.
Figure 1: Lateral view showing prominence of the swelling on clenching the teeth

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His blood count and coagulation profile were within normal limits. Orthopantomogram was not contributory. Ultrasound imaging of the swelling showed a well-defined hypoechoic lesion within the right masseter muscle and increase in muscle thickness on right masseter when compared with the left side. Color Doppler examination showed venous blood flow suggesting a venous vascular lesion [Figure 2]. CT scan showed an increase in the size of right masseter with signal voids. Lateral border of the masseter muscle was hyperintense on contrast enhancement. MRI examination showed a heterogenous mass within the right masseter with multiple hypointense areas in the center suggestive of venous vascular malformation [Figure 3] and [Figure 4].
Figure 2: Color Doppler ultrasound showing venous flow

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Figure 3: Magnetic resonance imaging T1-weighted image showing heterogenous hypointense mass in right masseter

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Figure 4: Magnetic resonance imaging T2-weighted image showing heterogenous hyperintense mass in right masseter

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The patient was referred to an interventional radiologist for sclerotherapy. He underwent intralesional injection of 10 ml of 95% ethanol in a single session. This was, followed by complete resolution of the lesion [Figure 5]. The patient is under follow-up for the past 2 years with no evidence of recurrence [Figure 6].
Figure 5: Peroperative angiogram

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Figure 6: Postoperative picture after 2 years

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   Discussion Top


Vascular anomalies are a heterogeneous group of congenital blood vessel disorders more typically referred to as birthmarks. Vascular anomalies were broadly classified by Hein et al. into hemangiomas and vascular malformations based on clinical presentation, histopathologic features and biological behavior. [1] Hemangiomas are benign congenital neoplasms of the childhood. Infantile hemangiomas are the most common tumor in infancy and occur in approximately 10% of the population. Infantile haemangiomas are rarely apparent at birth but present shortly after birth most often as well-demarcated, flat, and erythematous red patches. Hemangiomas follow a predictable course with three distinct developmental phases: Proliferation, quiescence, and involution. They grow rapidly during the first 6 months of life by cellular hyperplasia and are characterized by increased endothelial cell turnover. Following proliferation, hemangiomas enter a slower or no growth phase, known as quiescence. This phase typically lasts from 9 to 12 months of age. The final and unique phase of the hemangioma life cycle is involution. This phase is marked by graying of the overlying skin and shrinking of the deeper components. At the final stages of involution, a fibrofatty protuberance may remain. Hemangiomas are managed with close observation over their lifecycle. Medical and surgical options are available for the treatment of "problematic" hemangiomas. Systemic therapy with corticosteroids, interferon, and vincristine have been successful for massive and life-threatening disease. Surgical management involves excision, laser treatment or both.

Vascular malformations are irregular vascular networks defined by their particular blood vessel type. Intravascular malformations are often mislabeled as hemangiomas due to a similar presentation. They are usually present at birth and are slow growing, infiltrative, and destructive. Developmentally they are localized or diffuse errors of embryonic development characterized by normal endothelial cell turnover, altered vessel architecture. Vascular malformations are classified rheologically into slow flow lesions (capillary, lymphatic, venous) and fast flow lesions (arterial lesions such as aneurysm, ectasia, stenosis, fistula, arteriovenous malformation [AVMs]). Histologically they are classified into a small vessel (capillary), large vessel (cavernous) or mixed vessel lesions.

Lymphatic malformations (LMs) are composed of dilated lymphatic vessels with inappropriate communication, lined by endothelial cells and filled with lymphatic fluid. LMs may be macrocystic, microcystic, or mixed. Gradual growth and expansion are typical. Most LMs are found in the cervicofacial region.

Capillary malformations (CMs) consist of dilated capillary-like channels. They are mostly found in the cervicofacial region. They present at birth as flat, red or purple, cutaneous patches with irregular borders. Although they are mostly solitary lesions, CM may exist as a part of a syndrome. The most common of these is the  Sturge- Weber syndrome More Details More Details and is characterized by a CM in the region of the ophthalmic branch of the trigeminal nerve, leptomeningeal angiomatosis, and choroid angioma. The mainstay of treatment for CM is laser therapy. Surgical excision is also an option in lesions not amenable to laser therapy. This is especially true in advanced lesions, which have become nodular.

Arteriovenous malformations are congenital high-flow vascular malformations composed of anomalous capillary beds shunting blood from the arterial system to the venous system. The natural course of AVM is early quiescence, late expansion, and ultimately infiltration and destruction of local soft tissue and bone. Common sites for occurrence are the midface, oral cavity, and limbs.

Venous malformations are slow-flow vascular anomalies composed of ectatic venous channels. VMs are often visible at birth but may present as a deep mass. Protrusion may be the only presenting symptom. They are known to grow proportionately with the child with a sudden expansion in adulthood. Rapid growth may occur during puberty, pregnancy, or traumatic injury.

Venous malformations of the head and neck present in a wide spectrum from isolated skin varicosities or deep spongy masses to complex lesions that permeate across tissue planes to involve airway, pharynx and mediastinum. About 50% of craniofacial VMs are noted at birth reminder appear in childhood. They are soft, decompressible, non-pulsatile lesions that expand with valsalva maneuver and when the patient bends over with the head in the dependent position. There is little tendency to enlarge rapidly unless thrombosed. In older lesions, phlebolith can be palpated or can be seen on the radiograph.

Intramuscular VMs previously incorrectly termed intramuscular hemangioma constitute a rare subgroup of VMs. The age at presentation in this subgroup is usually 20-30 years. [6] They occur most often in the trunk and extremities, perhaps because of the larger muscle volume in those areas. In head and neck, common locations are masseter, trapezius, and sternocleidomastoid, etc. [7] Patients usually present with a growing palpable mass with or without pain. [6] The swelling is usually soft, diffuse and compressible. Increase in size with procedures that increase venous pressure such as dependent position, valsalva maneuver, compression of jugular vein can be demonstrated in venous lesions. Arterial thrill or bruit is rare in venous lesions.

Lesions of the masseter are often mistaken for parotid swelling. [8] Differential diagnoses include salivary gland tumors, masseter muscle hypertrophy, lymphangioma, angiosarcoma, haemangiopericytoma, rhabdomyosarcoma, myositis ossificans. Due to deep-seated location and rarity of signs suggesting a vascular etiology, an accurate preoperative diagnosis needs a high degree of suspicion. Ultrasonogram coupled with color Doppler can be used to distinguish hemangioma from vascular malformation and is often used to guide sclerotherapy. [9] MRI plays an important role in diagnosis. It characterizes type of lesion, extent of the lesion, involvement of tissue layers, flow pattern to guide treatment towards transarterial or percutaneous embolization. [10] Phlebography plays an important role in identifying venous drainage pattern that is, cavitary, spongy or dysmorphic. [3]

Management of vascular malformation needs to be individualized based on location, extent of the lesion, flow characteristics, accessibility, cosmetic considerations. Surgical removal is the treatment of choice in symptomatic well circumscribed lesions. If the muscle can be sacrificed without compromising function, then resection of that muscle can lead to cure. However, the surgery is associated with the risk of bleeding, loss of motor function, nerve damage, etc. [11] The intra-masseteric location also poses a special problem in terms of proximity to the facial nerve and the postoperative flattening following excision of the masseter muscle. Recurrence rates vary depending on size and margin. Nonsurgical methods of treatment include sclerotherapy, laser therapy, embolization and cryotherapy. Sclerotherapy is a simple and feasible alternative to surgery used as a sole modality or in combination with surgery. [11] When used alone multiple sessions are often necessary. Sclerotherapy uses sclerosant agents to destroy vascular endothelium. Various sclerosants used are chemical agents like ethanol or iodine, osmotic agents such as salicylates, hypertonic saline, detergents such as sodium tetradecyl sulfate, polidocanol, diatrizoate sodium. Sclerotherapy yields good to excellent results in the majority of cases especially in diffuse lesions. Contraindications are multiple intralesional thrombosis, neurologic impairment, signs and symptoms of compression. Complications are accidental injection into the systemic circulation, hemoglobinuria, renal toxicity, skin necrosis, ulceration, nerve injury and infection. [6] Laser therapy uses the principle of selective photohemolysis. Carbon dioxide laser can be used to treat small oral mucosal lesions. Photocoagulation with argon or neodymium doped: yttrium aluminum garnet laser can be effective for small superficial venous or capillary, venous lesions. Laser photocoagulation causes dermal/submucosal fibrosis, thickening. Embolization using absorbable or nonabsorbable material prior to surgery to reduce the size has also been demonstrated.


   Conclusion Top


Presented above is a case report of intramuscular VMs treated by intralesional sclerotherapy, a nonsurgical procedure in the management of these lesions. This paper also emphasizes the importance of sclerotherapy in its management.



 
   References Top

1.
Hein KD, Mulliken JB, Kozakewich HP, Upton J, Burrows PE. Venous malformations of skeletal muscle. Plast Reconstr Surg 2002;110:1625-35.  Back to cited text no. 1
    
2.
Rai P, Setia S, Kalra N, Upreti L. Intramuscular vascular malformation of the masseter muscle presenting with turkey wattle sign. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:618.  Back to cited text no. 2
    
3.
Dubois J, Soulez G, Oliva VL, Berthiaume MJ, Lapierre C, Therasse E. Soft-tissue venous malformations in adult patients: Imaging and therapeutic issues. Radiographics 2001;21:1519-31.  Back to cited text no. 3
    
4.
Zheng JW, Mai HM, Zhang L, Wang YA, Fan XD, Su LX, et al. Guidelines for the treatment of head and neck venous malformations. Int J Clin Exp Med 2013;6:377-89.  Back to cited text no. 4
    
5.
Bella GP, Manivel JC, Thomoson RC, Cbhisy D, Cheng EY. Intramuscular haemangioma: Recurrence risk related to surgical margins. Curr Orthop Pract 2007;459:186-91.  Back to cited text no. 5
    
6.
Burrows PE, Mason KP. Percutaneous treatment of low flow vascular malformations. J Vasc Interv Radiol 2004;15:431-45.  Back to cited text no. 6
    
7.
Mulliken JB. Vascular malformations of the head and neck. In: Mulliken JB, Burrows PE, Fishman SJ, editors. Vascular Anomalies: Hemangiomas and Malformations. 2 nd ed. New York: Oxford University Press; 2013. p. 839-71.  Back to cited text no. 7
    
8.
Lee SK, Kwon SY. Intramuscular cavernous hemangioma arising from masseter muscle: A diagnostic dilemma (2006:12b). Eur Radiol 2007;17:854-7.  Back to cited text no. 8
    
9.
Paltiel HJ, Burrows PE, Kozakewich HP, Zurakowski D, Mulliken JB. Soft-tissue vascular anomalies: Utility of US for diagnosis. Radiology 2000;214:747-54.  Back to cited text no. 9
    
10.
Fayad LM, Hazirolan T, Bluemke D, Mitchell S. Vascular malformations in the extremities: Emphasis on MR imaging features that guide treatment options. Skeletal Radiol 2006;35:127-37.  Back to cited text no. 10
    
11.
Yakes WF. Diagnosis and management of venous malformations. In: Savader SJ, Trerotola SO, editors. Venous Interventional Radiology with Clinical Perspectives. 2 nd ed. New York: Thieme Medical Publishers; 2000. p. 190-3.  Back to cited text no. 11
    

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Correspondence Address:
Venkatesh Jayaraman
Department of Oral Medicine and Radiology, Rajah Muthiah Dental College and Hospital, Annamalai University, Chidambaram, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.156824

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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