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Isolated Posterior Spinal Artery Aneurysm Presenting With Spontaneous Thrombosis

After Subarachnoid Hemorrhage: A Case Report

Kento Takebayashi, MD, Tomomi Ishikawa, MD, Masato Murakami, MD, Takayuki
Funatsu, MD, Tasuya Ishikawa, MD, Takaomi Taira, MD, Takakazu Kawamata, MD

PII: S1878-8750(19)32958-4
DOI: https://doi.org/10.1016/j.wneu.2019.11.118
Reference: WNEU 13787

To appear in: World Neurosurgery

Received Date: 31 October 2019

Revised Date: 19 November 2019
Accepted Date: 20 November 2019

Please cite this article as: Takebayashi K, Ishikawa T, Murakami M, Funatsu T, Ishikawa T, Taira T,
Kawamata T, Isolated Posterior Spinal Artery Aneurysm Presenting With Spontaneous Thrombosis After
Subarachnoid Hemorrhage: A Case Report, World Neurosurgery (2019), doi: https://doi.org/10.1016/
j.wneu.2019.11.118.

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Isolated Posterior Spinal Artery Aneurysm Presenting With Spontaneous Thrombosis After

Subarachnoid Hemorrhage: A Case Report

Running title: Isolated posterior spinal artery aneurysm

Kento Takebayashi, MD; Tomomi Ishikawa, MD; Masato Murakami, MD; Takayuki Funatsu, MD;

Tasuya Ishikawa, MD; Takaomi Taira, MD; Takakazu Kawamata, MD

Department of Neurosurgery, Tokyo Women’s Medical University, Tokyo, Japan

Correspondence: Kento Takebayashi, MD

Department of Neurosurgery, Tokyo Women’s Medical University

8-1 Kawada-cho, Shinjuku-ku, Tokyo 162-8666, Japan

Tel: 03-3353-8111

Fax: 03-5269-7387

E-mail: tikurin0221@yahoo.co.jp

Type of manuscript: Case report

Classifications: Spine, Vascular

Email addresses for coauthors:

Tomomi Ishikawa: tomo.nov12@gmail.com

Masato Murakami: murakami.masato@twmu.ac.jp

Takayuki Funatsu: cabmetallions@hotmail.com

Tasuya Ishikawa: ishitatsu1001@gmail.com

Takaomi Taira: ttaira@twmu.ac.jp

Takakazu Kawamata: tkawamata@twmu.ac.jp

Keywords: Posterior spinal artery aneurysm, Subarachnoid hemorrhage, Radiculopial artery

Declarations of Interest

None
 Takebayashi 1

Abstract

Background: The cause of subarachnoid hemorrhage is more likely to be intracranial than spinal. Bleeding,

although common with spinal arteriovenous malformations and spinal cord tumors, rarely occurs with

ruptured isolated spinal artery aneurysms. Here, we report a case of isolated thoracic posterior spinal artery

aneurysm presenting with thrombosis after subarachnoid hemorrhage.

Case description: A 67-year-old woman presented with sudden-onset nausea and low back and right thigh

pain that worsened with movement. Computed tomography (CT) and magnetic resonance imaging (MRI)

scans of the head suggested a small subarachnoid hemorrhage in the high-convexity sulcus, and lumbar

puncture showed bloody cerebrospinal fluid. There was no apparent intracranial aneurysm on CT

angiography; however, spinal MRI showed a lesion on the right side of the spinal cord at Th10.

Contrast-enhanced CT showed an enhancing lesion at this site on day 7 that was not present on day 15.

Selective right Th10 intercostal artery angiography on day 22 showed no evidence of aneurysm. The lesion

was suspected to be a thrombotic spinal artery aneurysm. Given the unclear natural history of this entity,

surgery was performed on day 36. After right Th10 hemilaminectomy and opening the dura, the arachnoid and

adhesions were found to be thickened. A fusiform-shaped thrombosed aneurysm continuous with the

radiculopial artery was removed. The patient was discharged without neurological deterioration.

Conclusions: Isolated spinal artery aneurysm is a rare cause of subarachnoid hemorrhage. It is expected that

additional cases will clarify the natural history and indications for treatment.

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 Takebayashi 2

Introduction

Subarachnoid hemorrhage most often occurs as a result of spinal disease rather than an intracranial disorder.

Although frequently reported in association with spinal arteriovenous malformations and spinal cord tumors,

bleeding due to an isolated spinal artery aneurysm has been rarely reported. The frequency of spinal artery

aneurysm is reported to be less than one in 3000 spinal angiograms.1–3 Given the rarity of subarachnoid

hemorrhage due to spinal artery aneurysm, the natural history and indications for treatment of this entity are

still poorly understood. Here, we report a surgical case of thoracic isolated posterior spinal artery aneurysm

presenting with thrombosis after subarachnoid hemorrhage.

Case description

A 67-year-old previously healthy woman was brought to our hospital with sudden nausea and low back pain

after a ballet class. Her symptoms improved and she was discharged home. However, her symptoms worsened

on day 5 and she was admitted to our hospital. She had back and right thigh pain that worsened with

movement but was without headache or motor weakness. Computed tomography (CT) and magnetic

resonance imaging (MRI) of the head suggested a small subarachnoid hemorrhage in the high-convexity

sulcus, and lumbar puncture showed bloody cerebrospinal fluid. There was no apparent intracranial aneurysm

on a CT angiogram, but spinal MRI showed a lesion on the right side of the spinal cord at Th10 (Fig. 1).

Contrast-enhanced CT showed an enhancing lesion at the same site on day 7 (Fig. 2a) that was no longer

evident on day 15 (Fig. 2b). Selective angiography of the right Th10 intercostal artery on day 22 showed no

evidence of aneurysm (Fig. 3). The Adamkiewicz artery was confirmed by selective injection of the left Th10

intercostal artery.

Based on her medical history, MRI findings, and the time course of the findings from contrast-enhanced CT, it

was thought that the patient had a ruptured posterior spinal artery aneurysm that had become thrombotic.

Surgery was performed on day 36 to prevent rebleeding at the request of the patient and family after

counselling that a conservative wait-and-see approach without surgery was an option. Thickening of the

arachnoid and adhesions was found after right Th10 hemilaminectomy and opening the dura using a

microsurgical technique. Careful dissection of the arachnoid revealed a fusiform-shaped mass continuous with

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the radiculopial artery and dorsal nerve roots (Fig. 4a). Although indocyanine green videoangiography showed

no obvious blood flow inside the aneurysm, the parent vessel was blocked with a temporary clip. After

confirming that there was no change in electrophysiological monitoring for motor- and somatosensory-evoked

potentials, the aneurysm was removed (Fig. 4b). Histopathological examination revealed dilated vascular

tissue with irregular elastic plates showing thrombus formation (Fig. 5). The patient was discharged home

without any neurological symptoms after surgery.

Discussion

Blood is supplied to the ventral spinal cord by the anterior spinal artery which continues from the

radiculomedullary artery, and to the dorsal spinal cord by the posterior spinal artery which continues from the

radiculopial artery.

In this patient, the aneurysm occurred in the posterior spinal artery. Compared with the anterior spinal artery,

aneurysms in the posterior spinal artery are uncommon with only 19 cases reported in the literature.4

Aneurysms involving the cervical spine tend to have a poorer prognosis than those involving the

thoracolumbar spine5 because the hematoma often spreads into the cranium.

Although a spinal artery aneurysm is difficult to diagnose because of their rarity, it may be included in the

differential diagnosis of intracranial subarachnoid hemorrhage of unknown origin. In our case, this pathology

was rightly suspected because the main symptom was lower back pain without headache. Although MRI,

contrast-enhanced CT, and spinal angiography were useful, it should be noted that, as was the case for our

patient, the contrast effect may change over time because the aneurysm fills with arterial blood immediately

after rupture but gradually becomes obstructed, resulting in thrombosis. Isolated spinal artery aneurysm are

thought to occur by dissection, and these aneurysms often become thrombosed and occlusive because the

diameter of the vessel involved is smaller than that of the intracranial artery.

Therapeutic approaches for ruptured posterior spinal artery aneurysms include endovascular treatment, direct

surgery, and conservative management. Endovascular treatment is rare because the diameter of the

radiculopial artery is small and technically difficult to access by microcatheter. Rather, direct surgery has been

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used in most cases because a posterior surgical approach is relatively easy given that the posterior spinal

artery is located on the dorsolateral side of the spinal cord. It is difficult to preserve the parent artery during

surgery; therefore, surgical resection is typically performed.6–10 Given that the posterior spinal arteries have

abundant anastomoses with the pial network, single vessel occlusion is unlikely to disturb the circulation in

the spinal cord. However, intraoperative electrophysiological monitoring is considered desirable if possible.2

The natural history of ruptured posterior spinal artery aneurysm remains unclear; however, there have been a

few reports of spontaneous resolution of these aneurysms in patients who have been followed up without

surgical intervention11–13 and one report of death as a result of re-rupture.14 The aneurysm in the present case

was treated surgically at the request of the patient and family, although it was thought to be thrombosed based

on the findings preoperatively. Recent reports suggest that dissecting spinal artery aneurysms often occlude

spontaneously, as in this case, and propose conservative management as an option.11–13

In the future, the accumulation of more cases is expected to clarify the natural history and indications for

treatment of isolated posterior spinal artery aneurysm presenting with spontaneous thrombosis after

subarachnoid hemorrhage.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or

not-for-profit sectors.

Acknowledgments

We would like to thank Editage [http://www.editage.com] for editing and reviewing this manuscript for

English language.

Data Availability

The data analyzed in this report are available from the corresponding author upon reasonable request.

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Figure Legends

Fig. 1. Right parasagittal (a) and axial (b) T2-weighted magnetic resonance images obtained on day 9 show an

intradural low-intensity mass at the Th10 level.

Fig. 2. Contrast-enhanced computed tomography scan acquired on day 7 showing enhancement on the right

dorsal aspect of the spinal cord (a). The enhancing lesion was no longer present on day 15 (b).

Fig. 3. Selective spinal digital subtraction angiography on day 22 showing no evidence of aneurysm or fistula

in the right Th10 intercostal artery.

Fig. 4. Intraoperative view (orientation: left, caudal; right, rostral) showing a fusiform aneurysm on the

dorsolateral aspect of the spinal cord continuing from the radiculopial artery (a). The thrombosed aneurysm

was resected with the parent artery (b).

Fig. 5. Histopathological examination with (a) hematoxylin and eosin and (b) Elastica van Gieson staining

shows granulation tissue with fibrosis and dilated vascular tissue with an irregular elastic plate with thrombus

formation. Original magnification ×4.

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Fig. 1 (a, b)
Fig. 2 (a, b)
Fig. 3
Fig. 4 (a, b)
Fig. 5 (a,b)
Abbreviations:

CT, computed tomography

MRI, magnetic resonance iimaging