1. Lingual Artery Pseudoaneurysms – A Report of 2 Cases Rashid Ahmed MD1, Husitha R. Vanguru MBBS1,Carlos Ynigo D. Lopez MD1, Neil Suryadevara MD1, Thanh Nguyen MD2, Hesham Masoud MD1 1SUNY Upstate Medical University 2Boston University School of Medicine
Introduction
Case 1
Discussion
A 65-year-old man with a history of oral squamous cell carcinoma presented to the hospital with acute blood loss anemia secondary to refractory oral bleeding, 3 weeks after a right partial glossectomy. Imaging revealed foci of contrast enhancement in continuity with a right external carotid artery branch. Cerebral angiogram revealed a 7 mm x 6mm pseudoaneurysm of the right lingual artery. Cannulation, angiography of the facio-lingual trunk was then performed using a 5F Envoy MPC guide. Glidewire and roadmap guidance were utilized. A Headway-17 with co-axial 014 Synchro2 Soft microwire (later swapped for 012 Headliner) was used for micro-catheter cannulation. Micro-angiography demonstrated position of the micro-catheter tip in the lingual artery pseudoaneurysm, and coil embolization was performed of the fundus and sacrifice of the distal parent vessel. Control angiography of the facio-lingual artery showed occlusion of the pseudoaneurysm and distal lingual artery.
Aneurysms are rarely noted in the external carotid artery and it’s branches. A review of cervical carotid aneurysms by Schechter revealed a total of 853 aneurysms. Only 2.2% primarily affected the external carotid artery. Lingual artery pseudoaneurysms are usually secondary to trauma, or iatrogenic causes.
Pseudoaneurysms differ from true aneurysms in that a true aneurysm involves all three arterial walls: the intima, media and adventitia. Pseudoaneurysms result from a tear in the vessel wall with subsequent development of peri-arterial hematoma separated from the lumen by a thin fibrous lined pseudo-intima. This leads to their predisposition to leakage and rupture.
We report two cases of lingual artery pseudoaneurysm occurring from two different mechanisms, both presenting as oral bleeding, and managed with endovascular techniques.
Treatment modalities have historically included open surgery. However surgery usually involves higher rates of morbidity and mortality when compared to non-invasive modalities.Non-invasive modalities have included ultrasound guided thrombin injection,Liquid embolic agents and endovascular repair.
Although both cases were treated with coil embolization, the parent vessel in the first case was sacrificed while in the second it was preserved . This was done because, in the first case the parent vessel served no purpose with patient having already undergone ipsilateral glossectomy. It was preserved in the second case to avoid tongue necrosis as a complication of vessel sacrifice. Tongue necrosis may occur due to inconsistent collateral supply from the contralateral lingual artery.
Figure 4 – Lateral projection angiogram of the facio-lingual trunk showing 7mm pseudoaneurysm of the lingual artery (A) treated with coil embolization and parent vessel sacrifice (B).
Figure 1 – Lingual artery (7)
Arrow heads depict potential anastomosis between branches representing the facial, ophthalmic, internal maxillary, and transverse facial artery
Case 2
A 27-year-old man presented with oral bleeding after a gunshot wound to the face. Imaging revealed a right lingual artery pseudoaneurysm measuring 11mm. He was successfully treated with coil embolization without parent vessel sacrifice, to preserve supply to the tongue.
Conclusion
Anatomy
Lingual artery pseudoaneurysms are rarely encountered in the Interventional Neurology practice , and are usually related to trauma or surgical procedures involving the floor of the mouth. The cases presented demonstrate successful treatment with coil embolization and cessation of active bleeding.
The lingual artery is the second anterior ECA branch. It initially courses superiorly, medial to the pharyngeal muscles, and the hypoglossus muscle. It then loops down and forward before curving upward towards the tongue.
Variations in origin do occur. Zumre et al. recorded that 20% of the human fetuses showed linguofacial trunks, and Lohan et al. using high-spatial-resolution MR angiography at 3T, reported that about 13.05% of their cases revealed common origin of the facial and lingual arteries. A rare combination branch of the external carotid is a thyro-linguo-facial trunk.
References
Thomas J. Chirichella et al. 2011,Lingual Artery Pseudoaneurysm with Arteriovenous Fistula Formation Following a Gun Shot Wound, Journal of surgical Radiology, Vol. 2 ,
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Zumre O, Salbacak A, et al Investigation of the bifurcation level of the common carotid artery and variations of the branches of the external carotid artery in human fetuses. Ann. Anat., 187:
Bergman RA, Afifi AK, et al. 2009,llustrated encyclopedia of human anatomic variation. Opus II. Cardiovascular system: head, neck, and thorax.
Lohan D, Barkhordarian F, et al. (2007) MR angiography at 3 T for assessment of the external carotid artery system. AJR, 189: A B
Figure 2 – Lateral (A) and anteroposterior views (B) of a left common carotid angiogram. Lingual artery (2) with its U-shaped curve
Figure 3: Sagittal CTA showing pseudoaneurysm of the lingual artery (A) and increase in size on follow up imaging 1 day later (B).
Figure 5 – Lateral projection demonstrating traumatic pseudo aneurysm of the lingual artery before coiling (A) and (B) after coil.