Traumatic Pseudoaneurysm in Maxillary Sinus Presenting Intractable Epistaxis: a Case Report of Transarterial Embolization

Online December 15, 2016 Journal of Neuroendovascular Therapy 2017; 11: 76–80 DOI: 10.5797/jnet.cr.2016-0033

Traumatic Pseudoaneurysm in Maxillary Sinus Presenting Intractable Epistaxis: A Case Report of Transarterial Embolization

Kentaro Hayashi, Yoshitaka Matsuo, Yukishige Hayashi, Kiyoshi Shirakawa, and Makio Kaminogo

Objective: Craniofacial injury with fracture may lead to pseudoaneurysm formation in the external carotid artery system, causing massive epistaxis. In this study, we report a patient with intermittent epistaxis related to a traumatic aneurysm following right blow-out fracture, and review the literature with respect to the pathogenesis and treatment of traumatic aneurysms. Case Presentation: The patient is a 91-year-old male. He had undergone conservative treatment for right blow-out fracture because he fell down while walking. Subsequently, he had received medication with an iron preparation to control intermittent massive epistaxis. Six months after the head injury, he consulted the Department of Otorhinolaryngology with persistent epistaxis. In the right nasal cavity, a pulsatile mass was detected, suggesting a vascular lesion. He was referred to our department. Neurologically, there were no abnormalities, but marked anemia was noted, and blood transfusion was performed. Head CT revealed a mass occupying the right maxillary sinus. The mass was partially enhanced on contrast-enhanced CT. Angiography showed a pseudoaneurysm, measuring 8 × 10 × 15 mm, originating from a branch of the right internal maxillary artery. Subsequently, transarterial embolization was performed. A microcatheter was inserted to reach the aneurysm, and it was embolized using a liquid embolic material. After surgery, there was no epistaxis, and an improvement of anemia was achieved. Conclusion: In epistaxis patients with a history of craniofacial injury, it is necessary to differentiate traumatic aneurysms. Endovascular treatment is effective for traumatic aneurysms, and should be selected as a first-choice treatment.

Keywords▶ head injury, blow-out fracture, pseudoaneurysm, epistaxis

Introduction symptoms, and review the literature with respect to the pathogenesis and treatment of traumatic aneurysms. Craniofacial injury with fracture may lead to pseudoaneurysm formation in the external carotid artery system, caus- Case Presentation ing massive epistaxis. In this study, we report a patient with intermittent epistaxis, related to a pseudoaneurysm follow- Patient: A 91-year-old male. ing right blow-out fracture, in whom endovascular treat- Complaint: Intermittent massive epistaxis. ment (embolization of the pseudoaneurysm) reduced the Medical history: Hypertension. Mild dementia was noted, but independence in daily living at home was maintained. Family history: Not contributory. Department of Neurosurgery, Sasebo City General Hospital, Present illness: While walking, he fell down, leading to a Sasebo, Nagasaki, Japan right blow-out fracture. At a local clinic, conservative Received: March 8, 2016; Accepted: September 11, 2016 treatment was performed. Epistaxis appeared 3 months Corresponding author: Kentaro Hayashi. Department of Neuro- after the head injury, and intermittently persisted thereafter. surgery, Sasebo City General Hospital, 9-3 Hirase-machi, Sasebo, In the Department of Internal Medicine, an iron preparation Nagasaki 857-8511, Japan was prescribed, but anemia deteriorated. He consulted the Email: [email protected] ©2017 The Editorial Committee of Journal of Neuroendovascular Department of Otorhinolaryngology 6 months after the Therapy. All rights reserved. head injury. Nasal endoscopy showed a pulsatile clot in

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Medical Co., Ltd., Japan). An aneurysm originating from a branch of the external carotid artery was visualized. Subse- quently, transarterial embolization was performed. The 4 Fr sheath was exchanged for a 6 Fr sheath, and heparin- ization was conducted. A 6 Fr guiding catheter (Envoy; Codman & Shurtleff, Inc., Raynham, MA, USA) was inserted into the right external carotid artery. A microcatheter (Renegade; Stryker, Natic, MA, USA) was inserted into the right maxillary artery using a Guidewire (0.016-inch GT wire double angle; Terumo, Tokyo, Japan). Angiogra- phy through the maxillary artery revealed a pseudoaneurysm (Figs. 4A and 4B). The infraorbital artery was suspected to be the responsible blood vessel. The microcatheter was inserted into the infraorbital artery. On selective Fig. 1 Otolaryngological endoscopy. A pulsatile mass covered by clot (arrow) is seen at the right nasal cavity. angiography, no blood vessel peripheral to the pseudoaneurysm was visualized, and there was no anastomosis with the internal carotid artery system. Therefore, an provoca- the right nasal cavity and hemorrhage involving the larynx tion test was not conducted. Embolization with 25% (Fig. 1). He was referred to our department. n-butyl 2-cyanoacrylate (NBCA) was performed. When Physical examination: Blood pressure, 140/70 mmHg; the pseudoaneurysm was embolized, NBCA outflow was pulse, 70 beats/min; height, 160 cm; and body weight, 50 kg. noted on the peripheral side of the responsible blood vessel Neurological findings: Mild dementia was noted, but there (Figs. 4C and 4D). When NBCA retrograded to the prox- were no other abnormalities. imal side, infusion was stopped, and the microcatheter was Hematological data: The erythrocyte count, hemoglobin removed. The periphery became clear with NBCA outflow, level, hematocrit, platelet count, and leukocyte count were and the infraorbital artery could be identified as the respon- 282 × 104 µL, 6.1 g/dL, 20.4%, 261 × 103 µL, and 6530 µL, sible blood vessel. On postoperative angiography of the respectively. external carotid artery, there was no pseudoaneurysm visu- Neuroradiological findings: Plain CT revealed a mass alized (Figs. 4E and 4F). After confirming the absence of occupying the right maxillary sinus. Bone-conditioning CT a pseudoaneurysm using angiography of the right and left showed right blow-out fracture (old) and osteolysis of the common carotid arteries, the procedure was completed. lateral wall of the maxillary sinus. Contrast-enhanced sin- Postoperative course: After surgery, there were no neuro- gle-phase CT revealed a lesion measuring 8 mm × 10 mm logical abnormalities. Epistaxis disappeared, and anemia × 15 mm, which was enhanced above the mass, suggesting was improved. Plain CT showed a high-attenuation area, a pseudoaneurysm (Fig. 2). On MRI T2* images, a high- suggestive of NBCA, in the right maxillary sinus (Fig. 5). signal-intensity mass with a low-signal intensity at the The mass in the right maxillary sinus had slightly reduced. margin was detected (Fig. 3). Neither original nor maxi- The patient was referred to another hospital for rehabilita- mum intensity projection (MIP) MRA images showed any tion 12 days after surgery. abnormalities in the right internal carotid artery, suggesting the involvement of a branch of the right external carotid Discussion artery. However, there was no pseudoaneurysm or responsible blood vessel visualized. Craniofacial injury-related traumatic aneurysms are classi- Angiography and endovascular treatment: Four units of fied into intracranial and extracranial aneurysms. Intracra- packed red blood cells were transfused for systemic man- nial aneurysms frequently develop in fixed sites of blood agement. Subsequently, angiography was performed to vessels or around the cerebral falx. Their rupture leads to make a definitive diagnosis. A 4 Fr sheath was inserted into subarachnoid or intracerebral hemorrhage. Extracranial the right common femoral artery, and angiography of the aneurysms are detected in the internal/external carotid and right common carotid artery was conducted using a vertebral arteries. Many internal carotid artery aneurysms HANACO excellent EN catheter (4.2 Fr/100 cm, Hanaco are related to fracture of the skull base, and most vertebral

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Fig. 2 Postcontrast CT shows the enhancement of a part of the mass with contrast medium (arrow). (A) axial image; (B) coronal image; and (C) sagittal image.

a few months after injury, possibly because it depends on the strength of the vascular wall or peripheral osteolysis.5) In the present case, a mass occupying the right maxillary sinus was detected, and contrast-enhanced CT revealed a portion of the pseudoaneurysm. However, the periphery was not enhanced, and MRI showed hemosiderin deposition and granulomatous changes. Usually, epistaxis grad- ually becomes severe, but initial hemorrhage is fatal in some cases.6) For the diagnosis of epistaxis, otorhinolaryngological examination is initially performed. In the present case, endoscopy revealed a pulsatile mass, and a clot was adhered. Hemorrhage involved the larynx, and persisted. CT showed a mass occupying the right maxillary sinus and osteolysis of the maxillary sinus wall in addition to old blow-out fracture, suggesting the influence of chronic pseudoaneurysm-/ clot-related compression. Based on contrast-enhanced CT findings, a pseudoaneurysm was suspected. MRI revealed a Fig. 3 MRI T2* image shows a high-intensity mass at the low-signal-intensity lesion, suggestive of hemosiderin right maxillary sinus with a low-intensity ring indicating deposition, in the right maxillary sinus. MRA did not show hemosiderin deposition. any abnormalities in the internal carotid artery, suggesting a lesion of a branch of the external carotid artery. However, it artery aneurysms are related to fracture of the cervical ver- was difficult to identify it. Angiography is a gold standard tebrae. Some internal carotid artery aneurysms are associ- for traumatic aneurysm diagnosis.7) In the present case, ated with iatrogenic factors, such as transsphenoidal angiography also led to a diagnosis of a lesion of the exter- surgery and irradiation.1,2) nal carotid artery system. Selective angiography through Pseudoaneurysms of the external carotid artery system microcatheter insertion showed a pseudoaneurysm origi- frequently cause intermittent massive epistaxis as the initial nating from the inferior orbital fissure-passing area of the symptom.3,4) As responsible blood vessels, branches at the infraorbital artery. maxillary artery region, such as the sphenopalatine, infraor- To treat extracranial traumatic aneurysms, endovascular bital, and alveolar arteries, have been reported.4) In the treatment is primarily performed. For pseudoaneurysms present case, massive epistaxis intermittently occurred of the external carotid artery system, responsible blood 3 months after blow-out fracture, and marked anemia was vessels or pseudoaneurysms are embolized using particle noted. In many cases, epistaxis is observed a few weeks to embolization substances or coils.4) However, occlusion at

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Fig. 4 Carotid angiography and endovascular treatment. (A) and (B) Selective-angiogram through the microcatheter shows a pseudoaneurysm originating from the infraorbital artery at the inferior orbital fissure (arrow). (A) A-P view, (B) lateral view. (C) and (D) Images during N-butyl 2-cyanoacrylate (NBCA) injection to the infraorbital artery. NBCA flowed out to the orbital portion of the infraorbital artery (arrow) (C) A-P view, (D) lateral view. (E) and (F) Post-treatment control angiography shows the occlusion of the pseudoaneurysm. (E) A-P view, (F) lateral view.

the proximal region of a responsible blood vessel may cause material may be important for preventing recurrence. recurrent pseudoaneurysms through retrograde blood flow However, when selecting a liquid embolic material for the from the periphery. Furthermore, embolization of pseudo- external carotid artery system, especially periorbital blood aneurysms alone may induce recurrent pseudoaneurysms vessels, the material may aberrate to the site of anastomosis for the following reasons: coil flasking/aberration in the with internal carotid or ocular arteries, that is, dangerous tissue around a thrombus or elimination of foreign bodies anastomosis. The infraorbital artery branches from the third such as coils.4) Complete embolization of the responsible segment of the maxillary artery, showing an anterior course blood vessel to pseudoaneurysm with a liquid embolic at an area adjacent to the posterior wall of the maxillary

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Conclusion

If epistaxis or anemia is observed after head injury, the possibility of a traumatic aneurysm must be considered. It is important to embolize external carotid artery pseudoaneurysms involving the distal to proximal areas. Transar- terial embolization was effective. Furthermore, a collateral pathway from other sites may appear after embolization; postoperative follow-up is important.

Disclosure Statement

No conflict of interest.

References

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