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Table of Contents   
CASE REPORT  
Year : 2020  |  Volume : 31  |  Issue : 1  |  Page : 164-166
Arteriovenous malformation of the parotid gland: Diagnostic perspective- A case report


1 Department of Oral Medicine and Radiology, Annoor Dental College and Hospital Muvattupuzha, Ernakulam, Kerala, India
2 Department of Oral Medicine and Radiology, Vydehi Institute of Dental Sciences, White Field, Bengaluru, Karnataka, India
3 Consultant Oral and Maxillofacial Radiologist, Pune, Maharashtra, India

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Date of Submission03-Apr-2018
Date of Decision22-Mar-2019
Date of Acceptance15-Aug-2019
Date of Web Publication02-Apr-2020
 

   Abstract 


Arteriovenous malformation (AVM) of the parotid gland is an extremely rare condition with very few cases reported in the literature. Majority of the swellings in the parotid region are usually due to sialadenitis, tumour etc., therefore making the initial diagnosis of vascular malformation may be difficult if there is no evident Turkey-wattle sign. Here, we present a case of AVM of parotid gland with its clinical features, radiographic features and its management. The lesion, being a high flow one, was managed with sclerosing agents to regress its size.

Keywords: Arteriovenous malformation, sclerosing agent, turkey-wattle sign

How to cite this article:
John H, Padmashree S, Pandeshwar P, Innus SZ. Arteriovenous malformation of the parotid gland: Diagnostic perspective- A case report. Indian J Dent Res 2020;31:164-6

How to cite this URL:
John H, Padmashree S, Pandeshwar P, Innus SZ. Arteriovenous malformation of the parotid gland: Diagnostic perspective- A case report. Indian J Dent Res [serial online] 2020 [cited 2021 May 11];31:164-6. Available from: https://www.ijdr.in/text.asp?2020/31/1/164/281806



   Introduction Top


All vascular lesions until 1980's were called as haemangiomas. In 1982, Mullikin and Glowacki, classified vascular lesions into haemangiomas and vascular malformation based upon their respective endothelial characteristics. Based upon their flow, these lesions can be classified into low-flow like capillary malformations, lymphatic malformations and venous malformations and high-flow lesions like arteriovenous malformations (AVMs) and arteriovenous fistulae.[1]

Vascular malformations of the parotid region are extremely rare with just around 50 cases being reported so far (mostly venous malformations). It is very important to arrive at a proper clinical diagnosis first because irrational advocation of surgical procedures may lead to exsanguination and fatalities.[2] Vascular malformations are usually malformations: 1) which are structural with a normal rate of endothelial cell turnover, 2) which are present at birth and are relatively static in nature, 3) changing in size when they sustain trauma, infection or endocrine changes.[3] With this background, we present the case of AVM of parotid gland.


   Case Report Top


A 47-year-old female patient presented with the chief complaint of pain and swelling in the region in front of the left ear since four months. Pain appeared first which was intermittent, throbbing type, aggravated on having food and also while bending the head down and got relieved by itself. Swelling appeared almost simultaneously which was initially small in size and then gradually increased and reached the present size. She has a history of a similar problem around 13 years ago for which she underwent surgery but presently not carrying any reports pertaining to the same.

On extra oral inspection, diffuse swelling was present in the left preauricular region roughly measuring 4 cm × 6 cm. Anteriorly extending 2 cm posterior to a line dropped from the outer cathus of the eye posteriorly till a line dropped from the tragus. Superiorly, along the ala tragal line and inferiorly till about 1 cm inferior to the inferior border of mandible. Surface skin appeared shiny and stretched in the superior portion. Surgical scar was noted in the same region [Figure 1].
Figure 1: Swelling present in the left parotid region with the presence of a surgical scar

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The striking feature was the positive Turkey-wattle sign, i.e. increase in the size of the swelling while bending the head down [Figure 2]. On palpation, mild local rise of temperature was noted. Swelling was soft in consistency and slightly tender. Pulsations or bruit was not felt. Compressibility was present. Based on the above features, a provisional diagnosis of vascular malformation of the parotid gland region was made. Differential diagnosis of chronic sialadenitis and benign salivary gland tumour was given.
Figure 2: Prominence of the swelling in left parotid region on bending the head forward (Turkey wattle sign) which can be seen in Figure 2b

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OPG revealed small radiopaque flecks, i.e. phleboliths (about 5 in number) noted in the left ramus and posterior to left ramus each measuring not more than 3 mm in the greatest diameter [Figure 3]. Ultrasonography of left parotid showed evidence of dilated and tortuous vascular channels in the left parotid with high flow blood supply from the left facial artery. Few calcifications were noted in the dilated vascular channels. Evidence of echogenic content suggestive of thrombosis were also noted [Figure 4]. MRI revealed evidence of mass lesion arising from the superficial lobe of parotid. Iso-intense on T1 weighted images and hyperintense on T2 weighted images. MRA showed the extensive involvement of the left facial artery which was identified as the feeder vessel [Figure 5]. Therefore, based on the above features, a radiographic diagnosis of AVM left parotid gland was made. Surgical intervention was not done due to the high vascularity of the lesion and close proximity to the left facial nerve. Sclerotherapy of the lesion with 1 mL N- Butyl cyanoacrylate with 1 mL xylocaine, one injection weekly, for a duration of four weeks were given. Patient is currently under follow-up.
Figure 3: OPG Revealed small radiopaque flecks about 5 in number (depicted with the arrow) noted in the left ramus and posterior to left ramus each measuring not more than 3 mm in the greatest diameter

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Figure 4: USG of left parotid gland shows evidence of blood supply from left facial artery, phleboliths and also echogenic content suggestive of thrombosis

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Figure 5: (a) MRI showing Hyperintense on T2 weighted images, (b) MRA showing the foci of AVM in the Parotid Gland

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   Discussion Top


AVMs are the most common high-flow lesions, frequently identified in the head, neck, limbs and lungs. However, lesions of the oro-facial regions are very rare. AVMs have been reported in the maxilla, but they are more common in the mandible.[4] Vascular malformation of the parotid gland is an extremely rare condition with a female preponderance with no apparent side predominance. Only the superficial lobe of the parotid was involved in majority of the cases.[5] Vascular malformations are usually seen as painless, slow growing soft tissue enlargement, but in our case, the patient had slight throbbing pain. Patients normally seek cosmetic correction of the same.

'Turkey wattle sign' may be seen on clinical examination. The enlargement of a facial mass on dependency of the head is called the Turkey-wattle sign, which when present is pathognomonic of a vascular malformation or haemangioma.[6] The red vascular structure in the neck of the male turkey is called as Turkey-wattle, that can increase in size when filled with blood.

Imaging is important to assess the extent of disease and for diagnosis. MRI, magnetic resonance angiography (MRA), or arteriogram are the best imaging studies which will give a clearcut view of the extension, feeder vessels, etc., An arteriogram will demonstrate a rapid filling lesion with extravasation of dye in the lesion, which would appear as a nidus and rapid filling veins draining it. The nidus is where the abnormality is and the site of AV shunting. A large number of tumors may be considered as the differential diagnosis for these type of swellings like benign and malignant salivary gland tumors, but the tell-tale signs of positive Turkey-wattle sign and imaging investigations help us at arriving at a diagnosis.[7]

Various treatment modalities are present like surgical resection and/or embolization. Sclerotherapy is also one of the treatment modalities for management of AVMs, which will help in reducing the size of the lesion and plan out a surgery at a later date.[8],[9] Sclerotherapy involves injection of a special chemical into the AVM to ultimately shrink it and relieve the symptoms. Commonly used agents are sodium tetradecylsulphate, N-Butyl cyanoacrylate, etc., In our case, we used N- Butyl cyanoacrylate. Complications of sclerotherapy include skin and mucosal injury, anaphylaxis, infection, and nerve injury. Arteriovenous malformations are very destructive, locally invasive and can be life threatening. They must be treated as early as possible and complete removal or control should be the goal.[10],[11]


   Conclusion Top


Salivary glands involved by these vascular lesions are uncommon but presents a major dilemma. Understanding the classification and natural history is crucial to their management. Treatment of vascular anomalies is complex and often involves multiple disciplines and therapeutic options. As oral diagnosticians, our role in proper diagnosis and referral to the vascular anomalies therapeutic team is quite significant.

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.



 
   References Top

1.
Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: A classification based on endothelial eharacteristics. Plas Reconstr Surg 1982;69:412-22.  Back to cited text no. 1
    
2.
Achache M, Fakhry N, Varoquaux A, Coulibaly B, Michel J, Lagier A, et al. Management of vascular malformations of the parotid area. Eur Ann Otorhinolaryngol Head Neck Dis 2013;130:55-60.  Back to cited text no. 2
    
3.
Abramowicz S, Marshall CJ, Dolwick MF, Cohen D. Vascular malformation of the temporomandibular joint: Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:203-6.  Back to cited text no. 3
    
4.
Wakoh M, Harada T, Yamamoto K, Hashimoto S, Noma H, Kaneko Y. Three-dimensional volumetric visualization of arteriovenous malformation of the maxilla. Dentomaxillofac Radiol 2003;32:63-6.  Back to cited text no. 4
    
5.
Bhatia C, Dalal S, Sattibabu V, Beniwal JP. Vascular malformation of the parotid gland: A rare case report. Int Surg J 2017;4:2081-3.  Back to cited text no. 5
    
6.
Saeed WR, Kolhe PS, Smith FW, Murray GI. The 'turkey wattle' sign revisited: Diagnosing parotid vascular malformations in the adult. Br J Plast Surg 1997;50:43-6.  Back to cited text no. 6
    
7.
Anesti K, Moaveni Z, Wu HY. Management of extensive intraparotid vascular malformation: A case report. Plast Aesthet Res 2014;1:33-6.  Back to cited text no. 7
  [Full text]  
8.
Konas E, Canter HI, Cil B, Peynircioglu B, Karabulut E, Tuncbilek G, et al. Volumetric assessment of results of treatment of vascular malformations of the head and neck regions treated with a minimally invasive surgical technique after embolization procedure. J Craniofac Surg 2009;20:402-5.  Back to cited text no. 8
    
9.
Shailaja SR, Manika, Manjula M, Kumar LV. Arteriovenous malformation of the mandible and parotid gland. Dentomaxillofac Radiol 2012;41:609-14.  Back to cited text no. 9
    
10.
Mishra M, Singh G, Gaur A, Tandon S, Singh A. Role of sclerotherapy in management of vascular malformation in the maxillofacial region: Our experience. Natl J Maxillofac Surg 2017;8:64-9.  Back to cited text no. 10
[PUBMED]  [Full text]  
11.
Veräjänkorva E, Rautio R, Giordano S, Koskivuo I, Savolainen O. The Efficiency of Sclerotherapy in the Treatment of Vascular Malformations: A Retrospective Study of 63 Patients. Plast Surg Int 2016;2016:2809152. doi:10.1155/2016/2809152  Back to cited text no. 11
    

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Correspondence Address:
Hima John
Department of Oral Medicine and Radiology, Annoor Dental College and Hospital, Muvattupuzha, Ernakulam - 686 673, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_296_18

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    Figures

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



 

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