Novel Insights from Clinical Practice Pediatr Neurosurg 2014–15;50:168–172 DOI: 10.1159/000381861

Received: September 26, 2014 Accepted after revision: March 19, 2015 Published online: May 20, 2015

Endovascular Occlusion of Cervical Internal Carotid Artery Pseudoaneurysm in a Child Treated by N-Butyl Cyanoacrylate: A Rare Case Report Anita Jagetia Divyajyoti Sharma Daljit Singh Sanjiv Sinha Pragati Ganjoo Poonam Narang Veeresh Mathod GB Pant Hospital and Maulana Azad Medical College, New Delhi, India

Established Facts • Pseudoaneurysms of the internal carotid artery, although uncommon, are being treated more commonly in the present era by endovascular procedures as well as by open surgery.

Novel Insights • Infective neck swellings are commoner than those of vascular origin; however, infection is not always the cause. • The cheapest modality for diagnosis is a Doppler ultrasound of the neck swelling. • In a child, radiation exposure should be kept at as small as possible while performing an endovascular procedure. • N-butyl cyanoacrylate is an effective and cheaper alternative than costlier agents to treat such lesions.

Abstract We report a rare case of spontaneous extracranial cervical internal carotid artery (ICA) pseudoaneurysm in a female child aged 3 years who presented with a swelling in the neck which had bled following an attempted incision as it had been thought to be an abscess. A CT angiogram and an MR angiogram were not very conclusive to diagnose the exact site of origin and the morphology of the aneurysm. Digital

© 2015 S. Karger AG, Basel 1016–2291/15/0503–0168$39.50/0 E-Mail [email protected] www.karger.com/pne

subtraction angiography revealed a dissecting pseudoaneurysm of the right extracranial cervical ICA. The right ICA was ending as a pseudosac, and the right cerebral circulation was filling up through the right posterior cerebral artery. To minimize the radiation exposure, a microcatheter was placed inside the diagnostic catheter. The aneurysm sac was occluded using N-butyl cyanoacrylate since there was no distal flow to the brain from the artery beyond the aneurysm. It was a safe, effective and cheaper alternative to open surgery or to other endovascular management options available. Not all neck swellings are abscesses, and they should be examined and evaluated to exclude a vascular cause. © 2015 S. Karger AG, Basel

Dr. Anita Jagetia J-71, Vijaylakshmi Apartment IP Extension, Patparganj, Delhi 110092 (India) E-Mail ID_ anitajagetia @ gmail.com

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Key Words Pseudoaneurysm · Pediatric age · Endovascular treatment · N-butyl cyanoacrylate

Introduction

Case Report A female child aged 3 years was referred for the treatment of a swelling on the right side of its neck, which was bleeding in bouts as she had been wrongly diagnosed as a case of cold abscess, and the swelling had been incised. This child had no history suggestive

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A pseudoaneurysm of the cervical internal carotid artery (ICA) may manifest as a swelling outside the cervical region or inside the pharynx. Though rare, a swelling appearing outside the cervical region can mimic a cold abscess there and, if not examined thoroughly, may be disastrous if incised. We are reporting a case of pseudoaneurysm in a child who was referred for the management of a swelling in the cervical region. This child was misdiagnosed as a case of abscess, and an incision was made in the swelling in its neck which led to profuse bleeding. The bleeding site was packed and dressed to stop bleeding.

This child was managed by an endovascular occlusion of the aneurysm. Endovascular management of this entity is based on the angiographic morphology of the aneurysm and the cerebral angiographic anatomy. The management of such an entity is discussed in detail, especially in a young child.

Fig. 1. a Bleeding site dressed with a compressive bandage. b An incision scar presa

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ent behind the right ear lobule after removing the dressing.

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Endovascular Occlusion of Cervical ICA Pseudoaneurysm in a Child

Pediatr Neurosurg 2014–15;50:168–172 DOI: 10.1159/000381861

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Fig. 2. Volume-rendering technique CT angiographic image. Lateral (a) and anteroposterior (b) views showing a saccular pseudoaneurysm arising from the right ICA at the level of the mandibular condyle. The axial (c) and sagittal image (d) of an MRI of the neck was suggestive of a mass with different stages of blood on the right side of the skull base.

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Fig. 3. Digital subtraction angiography images. a Dissecting the pseudoaneurysm of the right extracranial cervical ICA after the bifurcation of the common carotid artery with no distal cerebral blood flow from the parent artery. Collateral blood flow to the right cerebrum from the contralateral anterior cerebral artery (b) and the right vertebral artery (c). d Complete occlusion of the pseudoaneurysm and parent artery with NBCA.

of an upper respiratory tract infection. The bleeding site was dressed with a compressive bandage (fig. 1a), and the wound was not explored at the time of admission since it had bled 7–8 times in bouts and her hemoglobin had decreased to 6 g%. Hemodynamically, the child was stable and had no neurological deficit. However, blood transfusion was done to build up the hemoglobin level. The CT angiogram (fig. 2a, b) was suggestive of a saccular pseudoaneurysm arising from the right ICA at the level of the mandibular condyle, though it could not give much information about the right ICA beyond a common carotid bifurcation. An MRI of the neck (fig. 2c, d) was suggestive of a mass with different stages of blood coagulation on the right side of the skull base. Digital subtraction angiography of the neck vessels and brain was done using a 5-Fr diagnostic H1 catheter, which showed a dissecting pseudoaneurysm of the right extracranial cervical ICA after the bifurcation of the common carotid artery with no distal cerebral blood flow from the parent artery (fig. 3a). There were no dural-pial collaterals or dangerous anastomoses between the external carotid artery and the ICA. The right cerebrum was being supplied by the contralateral anterior cerebral artery and the right vertebral artery (fig. 3b, c). Instead of an exchange with a guiding catheter, a flow-guided microcatheter (ultraflow Ev3) was taken in the same diagnostic catheter in view of decreasing the radiation exposure to the child. The pseudoaneurysm was selectively cannulated. Since the artery was terminating in the aneurysm, the occlusion of the parent artery was the only treatment option pos-

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Pediatr Neurosurg 2014–15;50:168–172 DOI: 10.1159/000381861

sible in this case. N-butyl cyanoacrylate (NBCA) glue (25% NBCA and 75% lipidol) was injected into it in order to occlude the aneurysm of the parent artery, which was successfully achieved (fig. 3d). An incision scar was present behind the right ear lobule when the dressing was removed after the embolization (fig. 1b), and there was no active bleeding. The child was discharged asymptomatically.

Discussion

A pseudoaneurysm of cervical ICA is a rare entity, and the most common cause is trauma [1]. Other causes are upper respiratory tract infections [2–4], radiation exposure [5], iatrogenic [6], postoperative [7] or oropharyngeal tumors [8], and sometimes they occur spontaneously [9]. Deep neck space infection, retropharyngeal abscesses, peritonsillar abscesses, invasive fungal sinusitis and chronic otitis media have all been reported to cause vessel weakness and the formation of aneurysms and may manifest as external or internal swellings. However, in the present case, the patient had no predisposing factors, and the swelling was spontaneous in origin. Jagetia/Sharma/Singh/Sinha/Ganjoo/ Narang/Mathod

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Pseudoaneurysm may present as a neck swelling (pulsatile or nonpulsatile) and may mimic a cold abscess. It may manifest as a rapidly increasing mass, bruit, compression of adjacent structures such as of the respiratory air pathway, causing stridor, dyspnea, hoarse voice and dysphagia, and may cause brain ischemia, oral or nasal hemorrhage (epistaxis), embolic episodes such as transient ischemic attacks, amaurosis fugax and neck pain due to an enlargement of the pseudoaneurysm or a rupture of the pseudoaneurysm into the carotid sheath or intramural dissection [10]. When its presentation resembles a cold abscess, an incision or a fine needle aspiration cytology of the swelling may cause a life-threatening hemorrhage as in this case. Besides that, there can be dysphagia and/or respiratory distress. When the swelling is not visible externally, it may manifest as epistaxis and should alert the clinician for the possibility of a pseudoaneurysm of the cervical carotid artery [11]. ICA pseudoaneurysms can be diagnosed by a color Doppler ultrasound of the neck vessels [12]; however, this is difficult to perform in a patient who has bled externally as in the present case. A compression bandage had stopped the bleeding, and it was not advisable to remove the dressing to perform a Doppler study. However, that can be a cheap and very informative diagnostic modality to perform in doubtful cases of neck swelling. A computerized scan with carotid angiography, MRI of the neck and MR angiography should be performed to understand the morphology of the aneurysm; however, conventional digital subtraction angiography is the gold standard to confirm a pseudoaneurysm, its site of origin and the brain collateral circulation to plan its management. In present case, the child was 3 years old, and in order to reduce the dose of radiation during the procedure, a microcatheter was put inside the 5-Fr diagnostic catheter in place of an exchange with a guiding catheter. The goal of the treatment was to correct the lesion with preservation of the cerebral blood flow. The treatment options described in the literature are open ligation of the ICA, superficial temporal artery-middle cerebral artery bypass with ICA occlusion, stent graft placement (covered), direct coil embolization, stent-assisted coil embolization, ICA occlusion with detachable balloons, Onyx embolization of the ICA and histoacryl or NBCA embolization of the ICA [7, 13–15]. Open ligation of the carotid artery, when chosen in a patient with good collaterals from other vessels, becomes more invasive and traumatic. Coiling or balloon occlusion would have been costlier, would

have required a bigger catheter in this small child with small-caliber vessels and would have caused more radiation exposure. Stenting was not a suitable option for this particular case as there was no lumen to put it in. Since there was no abnormal communication between the external carotid artery and the ICA, occlusion of the terminal ICA with the pseudoaneurysm was considered safe as well. The bypass procedure was not considered a treatment option as this child had bled repeatedly and had undergone blood transfusion for the same; the main goal of the treatment was to save the child’s life through the preservation of the cerebral blood flow which was best achieved in the described way. Oynx or NBCA were the better options available to occlude the aneurysm sac and parent artery. Oynx is costlier, so we opted for NBCA, which is a cheaper, safe and effective option available for the occlusion of a pseudoaneurysm of the extracranial ICA as compared to Onyx. To the best of our knowledge, this is the second case report, after the report by Raffin et al. [7], in a child where an extracranial ICA pseudoaneurysm was treated using NBCA.

Endovascular Occlusion of Cervical ICA Pseudoaneurysm in a Child

Pediatr Neurosurg 2014–15;50:168–172 DOI: 10.1159/000381861

Conclusion

A neck swelling should be examined thoroughly and should be differentiated from vascular anomalies. It can be diagnosed by a cheaper modality, such as a Doppler ultrasound of the neck, when in doubt. All possible measures should be taken to reduce the dose of radiation, especially in a child. A complete assessment of the aneurysm, cerebral circulation and collaterals should be done to plan the management. Endovascular treatment is safe, least invasive, effective and becomes cost effective too when cheaper and safe embolizing agents are used.

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Endovascular Occlusion of Cervical Internal Carotid Artery Pseudoaneurysm in a Child Treated by N-Butyl Cyanoacrylate: A Rare Case Report.

We report a rare case of spontaneous extracranial cervical internal carotid artery (ICA) pseudoaneurysm in a female child aged 3 years who presented w...
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