AIJOC 10.5005/jp-journals-10003-1138 CASE REPORT Traumatic Arteriovenous Malformation of Cheek: A Case Report and Review of Literature Traumatic Arteriovenous Malformation of Cheek: A Case Report and Review of Literature Vadisha Srinivas Bhat, Rajeshwary Aroor, B Satheesh Kumar Bhandary, Shama Shetty ABSTRACT CASE REPORT Arteriovenous malformations (AVM) are congenital vascular A 31-year-old woman presented with swelling on right anomalies but are usually first noticed in childhood or adulthood. cheek of 6 months duration, which was insidious in onset Head and neck is the most common location for AVM. Extracranial lesions are rare compared to intracranial lesions. and gradually increasing in size. She had a history of The rapid enlargement of the malformation leading to symptoms undergoing dental treatment on the right upper premolar is usually triggered by trauma or hormonal changes of puberty tooth 1 month before the onset of swelling. There was no or pregnancy. Traumatic AVM of the head and neck are very history of any other trauma to the face. There were no rare. Here we report a case of AVM of cheek in an adult woman developed following a dental treatment. The diagnosis was symptoms of nasal disease. On examination, her general confirmed by imaging and was treated surgically after health state was good. There was a swelling measuring angiography and embolization. 3 × 2 cm on the right cheek near the nasolabial grove which Keywords: Arteriovenous malformation, Cheek, Dental was soft, compressible and nontender. The swelling was procedure, Angiography, Embolization. pulsatile (Figs 1 and 2). Nasal cavity and paranasal sinuses How to cite this article: Bhat VS, Aroor R, Bhandary BSK, were within normal limits. Oral cavity was normal. Shetty S. Traumatic Arteriovenous Malformation of Cheek: A Ultrasonography showed a hypoechoic area with central Case Report and Review of Literature. Int J Otorhinolaryngol necrosis measuring 15 × 10 mm in right premaxillary region. Clin 2013;5(3):173-177. On color Doppler, extensive vascularity noted consisting Source of support: Nil of arterial and venous channels. Adequate vascular channels Conflict of interest: None declared suggested feeders from branch of maxillary artery. The sonological features were suggestive of AV malformation. INTRODUCTION Contrast enhanced CT scan of paranasal sinus showed Arteriovenous malformations are congenital vascular intensely enhancing mass in right premaxillary soft tissue, anomalies but are usually first noticed several years after measuring 2.2 × 2.0 × 2.0 cm, with prominent vessels birth. The rapid enlargement of the malformation is usually surrounding it, suggesting vascular malformation (Figs 3 triggered by trauma or hormonal changes during puberty or to 5). MRI of the maxillofacial region showed a well defined pregnancy. Enlargement of the lesion is due to the changes rounded lesion in right premaxillary and nasolabial region in pressure and flow, dilatation of vascular channels, measuring 3.5 × 2 cm in size. Multiple enlarged flow voids shunting and collateral proliferation, rather than cellular seen within it suggesting high vascularity of malformation proliferation.1 The delay is diagnosis is either due to the (Fig. 6). Imaging studies were diagnostic of an arteriovenous dormant lesion expanding later, or due to the misdiagnosis malformation. of the malformation as hemangioma, especially in small Right external carotid angiogram showed moderate size vessel rich AVM, where even imaging may not differentiate (3-4 cm), compact, moderate flow right premaxillary and the lesion from hemangioma.2 Head and neck is the most nasolabial arteriovenous malformation supplied by common location for arteriovenous malformations. hypertrophied angular and anterior jugular branch of facial Extracranial lesions are rare compared to intracranial artery, infraorbital and buccal branches of distal internal lesions.3 Clinical diagnosis of AVM can be confirmed by maxillary, jugular branches of transverse facial artery. imaging- Doppler ultrasonography, CT scan with contrast Venous drainage was inferiorly into facial vein and and magnetic resonance imaging (MRI) will give a definite superiorly through angular vein into contralateral angular diagnosis.4 Angiogram shows dilated tortuous arteries, vein and also into right superior ophthalmic vein (Fig. 7). arteriovenous shunting and dilated draining veins, but this Selective embolization of the vessels was done with gel is useful only if embolization is planned. Surgical excision foam, attaining more than 50% reduction in vascularity. following selective embolization is the treatment of choice.1 Complete excision of the mass with ligation of the vessels, Very few cases of traumatic AVM are reported in the was done by lateral rhinotomy incision, elevating the facial literature. We report a case of AVM of cheek in an adult skin flap off the mass, under general anesthesia, the woman, following dental procedure. following day of embolization (Fig. 8). There was no Otorhinolaryngology Clinics: An International Journal, September-December 2013;5(3):173-177 173 Vadisha Srinivas Bhat et al Fig. 1: Clinical photograph (anterior view) Fig. 4: CT scan paranasal sinus—axial view post-contrast showing intensely enhancing region in right premaxillary soft tissue Fig. 2: Clinical photograph (right lateral view) Fig. 5: CT scan of paranasal sinus (coronal view post-contrast) Fig. 3: CT scan paranasal sinus (axial view plain) Fig. 6: MRI showing a well-defined rounded lesion in right premaxillary and nasolabial region 174 AIJOC Traumatic Arteriovenous Malformation of Cheek: A Case Report and Review of Literature Fig. 7: Right external carotid angiogram showing right premaxillary and nasolabial arteriovenous malformation Fig. 8: Excision of mass by lateral rhinotomy incision Fig. 9: H&E stain, low power showing vascular channels of various size significant bleeding during surgery. Patient withstood the procedure well. Postoperative period was uneventful. She was discharged from the hospital 6 days after surgery, after suture removal. Histopathological examination showed large dilated vascular channels with thrombi. Surrounding these large vascular channels is proliferation of capillaries in lobules with stasis of blood. Histological features were confirmatory of arteriovenous malformation (Figs 9 and 10). As the patient hails from a distance place, she could not visit us after surgery; however she is in touch with us on telephone. She is not having any complaints pertaining to the disease 1 year after surgery. Fig. 10: H&E high power showing vascular channels with thrombus DISCUSSION Vascular malformations are a form of vascular anomaly during embryogenesis and in fetal life.4 These vascular caused by abnormal development of vascular elements malformations do not produce increased endothelial cell Otorhinolaryngology Clinics: An International Journal, September-December 2013;5(3):173-177 175 Vadisha Srinivas Bhat et al turnover as seen in vascular tumors like hemangioma.4 Stage IV (decompensation): Features of cardiac failure These malformations can be of a single vessel form or in caused by an extensive AVM causing increased cardiac combination of arterial, venous, capillary or lymphatic output. malformation. Vascular malformations can be a slow flow The AVM can infiltrate causing local tissue destruction malformation like capillary, lymphatic or venous; or it can and often lead to life-threatening hemorrhage. Our patient be a fast flow like arterial or arteriovenous malformation.3 presented with a swelling over the cheek which was Arteriovenous malformations (AVM) are fast-flow pulsatile, and the AVM was demonstrated by imaging. vascular malformations, consisting anomalous capillary Hence she was considered in stage II. beds between the arterial and venous system, thus causing History and clinical examination can distinguish shunting of blood. AVMs are the most aggressive form of vascular tumors and vascular malformations in over 90% vascular malformations, which can lead to significant of cases.4 However types of malformation can be confirmed deformity and functional impairment.2 They are usually with ultrasound with color Doppler examination. CT scan noted at birth, but are commonly misdiagnosed as other with iodinated contrast identifies AVMs as a highly vascular anomalies leading to delay in diagnosis.3 Fast flow enhancing lesion and can demonstrate soft tissue becomes evident during childhood and puberty.4 Kohout involvement, as well as dilated feeding and draining MP et al in their study of AVM of head and neck, found in vessels.11 Magnetic resonance imaging of an AVM 59% of cases, AVM was present at birth. Ten percent noted demonstrates a collection of vascular flow voids corres- in childhood, 10% in adolescent and 21% in adulthood.5 ponding to fast-flow vessels and it can be considered the AVM grow synchronously with the growth of the child. investigation of choice due to its ability to delineate the Puberty and trauma are found to have triggered the rapid extent of the lesion, provide multiplane images and growth of the lesion leading to manifestation of the difference between high and low flow lesions. MRI is 3 symptoms. Enlargement is the result of changes in the superior to magnetic resonance angiography (MRA) and pressure and flow within the malformation, ectasia, shunting conventional cerebral angiography CCA in showing the and collateral proliferation, rather than cellular prolife- nidus and the involved brain structures,