Abstract

Traumatic arteriovenous (AV) fistulas are the vascular lesions which usually develop due to penetrating or blunt injuries or rarely due to an iatrogenic cause. Gunshot wounds, penetrating injuries, or bone fractures which are located in an adjacent artery and vein constitute the major etiology of the traumatic AV fistulas. Arteriovenous fistulas may not be diagnosed for years and may require surgical intervention long after the injury. When a traumatic AV fistula is not treated immediately and appropriately, it may cause subacute and chronic complications. Traumatic AV fistulas may progress to rarely encountered aneurysmatic dilatations in chronic periods. In this article, we report a 33-year-old male case with a neglected traumatic AV fistula which progressed to a facial AV aneurysm and received endovascular treatment successfully using transcatheter embolization technique.

Acquired arteriovenous (AV) fistulas usually develop following a penetrating injury.[1] These fistulas are called as traumatic AV fistulas, as the major etiology is injury. Malignancy or infections are also among the uncommon causes. Acquired AV fistulas usually involve the medium and large vessels and tend to present as single. Gunshot wounds, penetrating injuries, or surgical interventions are the main causes of the etiology.[1] Repeated arterial or venous interventions may also cause iatrogenic AV fistulas. In these cases, clinical signs may be detected from a few hours to years after injury, revealing a delayed formation of a pulsatile mass.[1,2] Clinical diagnosis of the traumatic AV fistulas depends on the history of injury and careful physical examination consisting of palpation and auscultation. Thrill and bruit over a pulsatile mass are the main characteristic features. Distal pulses are usually palpable; however a large AV fistula may shunt away blood from the extremity, causing peripheral ischemia.[3] The Nicoladoni-Israel-Branham’s sign may be also positive which means manual compression of the AV fistula causes reduced heart rate.[4]

The treatment of acquired AV fistulas is primarily surgical. Interruption of the connection between the artery and vein is sufficient for recovery. Existing vascular defects should be also repaired. Compression with the Doppler ultrasound (DUS) probe for 30 to 45 minutes over the AV fistulas was reported to be useful for the catheterization-induced iatrogenic cases. In addition, the graft coated stents may be used in certain regions. Therapeutic embolization may be used in pseudoaneurysms or in cases who are not candidate for surgery.[1] Insisted effort to perform open surgery in inappropriate patients may result in exposure difficulties, intraoperative hemorrhage, tissue damage, and postoperative scar development.

Regarding the treatment of extracranial and peripheral vascular pathologies, progressions in endovascular methods have revealed a more promising pathway. In this article, we report as uccessful endovascular treatment of a rare case with a facial AV fistula and aneurysm using transcatheter embolization technique. Emphasis is particularly given to the success of the endovascular methods in the treatment of AV fistulas which are located in the face and neck which are important visible parts of the body.

Case Presentation

A 33-year-old male patient was admitted to our outpatient clinic with a complaint of a painful swelling on the left side of the face in front of the left ear and the left neck region. Swelling was augmenting and spreading through the chin down to the neck during exertion. His medical history was otherwise normal except the history of penetrating injury to the left side of the neck under the chin with a sharp steel object seven years ago. Swelling grown out soon after the injury and showed progression over time. It became painful and non-reducible for the past year, since the reduction was initially possible with a gentle external manual compression.

There was a strongly pulsatile mass in front of his left ear on physical examination. Sharp thrill and bruit were auscultated along the left face up to the upper margin of the ear level. External jugular vein was engorged and weak thrill all the way its trace along the neck was obtained (Figure 1). External compression of the mass reduced the size and thrill of the aneurysm and the jugular vein. Doppler ultrasound revealed an AV aneurysmatic structure located in the related region. Initially, contrasted computed tomography (CT) angiography scan (1 mm/slice) and digital substraction angiography (DSA) were performed. An AV fistula and aneurysm with a size of 28x29 mm were detected in CT scans (Figures 2, 3). However, the distribution and quantity of feeding branches were unable to be detected clearly in either modalities. As a result, standard arteriography was further planned for the accurate diagnosis in combination with the treatment in a single session.

Figure 1: The image of the external morphology of the arteriovenous fistula (engorged jugular vein predominantly visualized)./ffigure1>

Figure 2: Computed tomography angiography (a threedimensional reconstruction image) showing the vasculature (upper arrow pointing the facial aneurysm, lower arrow pointing the congested jugular vein).

Figure 3: Computed tomography (contrasted) showing transverse view of the facial aneurysm.

The intervention was performed in an angiography unit with Artis Imaging System (Siemens Healthcare, Erlangen, Germany) under general anesthesia. A 7 F sheath was inserted into the right femoral artery with the Seldinger technique. A 0.035 inch x 150 cm hydrophilic guide-wire (Terumo Interventional Systems, Somerset, NJ, USA) was used within the supporting catheters. The Cobra-1 (Glidecath) hydrophilic (Terumo Interventional Systems, Somerset, NJ, USA) and Simmons-1 (Surefire) hydrophilic (Surefire Medical Inc., The Hague, The Netherlands) supporting catheters were used. The catheters were introduced through the left common carotid artery of the arcus aorta. Arteriography revealed an AV fistula with multiple feeders originating from the facial branches of the external carotid artery (Figure 4a). Azur Peripheral Hydrocoil System (Terumo Interventional Systems, Somerset, NJ, USA) was selectively placed at the distal part of the AV fistula through the facial artery, a branch of the external carotid artery. Distal obliteration stopped the retrograde filling of the artery. N-butyl-2-cyanoacrylate (NBCA; Histoacryl; B. Braun, Melsungen, Germany) and Lipiodol (Guerbet SA, France) mixture was selectively injected into five feeders in different locations via micro-catheter to prevent recurrences. The nidus was completely obliterated by the injection of 2.5 mL of NBCA. After the obliteration of the nidus, repeated angiogram revealed re-filling of the AV fistula by different multiple millimetric multi-feeders. Then, the main trunk of the external carotid artery was totally obliterated with NBCA (3.5 mL)-Lipiodol (10 mL) mixture including all the millimetric feeders. Repeated angiograms revealed no residual AV fistula (Figure 4b). No neurological deficit was seen after the procedure.

Following the procedure, the jugular venous engorgement disappeared and the thrill over the left face became non-palpable. He was discharged on the postoperative second day without any complication. Calcium dobesilate (1000 mg/day) was prescribed to increase and regain the venous tonus of the formerly overinflated jugular vein. Diclofenac potassium (100 mg/day) was also given for analgesia.

Figure 4: Arteriography before (a) and after (b) the endovascular treatment (arrow pointing the initial facial aneurysm) (images were taken in cranial-18 degree of angle).