Full text of DOI:10.1017/S0317167100001426

CASE REPORT
Endovascular Treatment of a
Lenticulostriate Artery Aneurysm with
N-butyl Cyanoacrylate
Ramiro Larrazabal, David Pelz, J. Max Findlay
ABSTRACT: Background: Aneurysms arising from lenticulostriate artery branches in moyamoya-type
disease are challenging lesions to treat, due to their fragility and deep location. Surgery is difficult and
endovascular options may be limited. Methods: A 57-year-old woman presented with a right ganglionic
parenchymal hemorrhage due to a ruptured lenticulostriate artery aneurysm associated with ipsilateral
middle cerebral artery occlusion. The aneurysm and parent feeding artery were occluded using
endovascular injection of N-butyl cyanoacrylate. Results: The aneurysm was successfully obliterated
and although some glue did enter the more distal middle cerebral artery, there was no change in the
patient’s neurologic status. Conclusions: In highly selected cases where lenticulostriate aneurysms
cannot be directly accessed for surgery or endovascular coiling, obliteration with liquid acrylic glue may
be considered as a therapeutic option.
RÉSUMÉ: Traitement endovasculaire par le N-butyl cyanoacrylate d’un anévrisme artériel lenticulostrié.
Introduction: Les anévrismes qui prennent naissance sur des branches de l’artère lenticulostriée dans la maladie de
type moyamoya sont des lésions difficiles à traiter à cause de leur fragilité et de leur site profond. La chirurgie est
difficile et les options endovasculaires limitées. Méthodes: Une femme âgée de 57 ans a consulté pour une
hémorragie parenchymateuse ganglionaire droite due à la rupture d’un anévrisme de l’artère lenticulostriée associé
à une occlusion de l’artère cérébrale moyenne ipsilatérale. L’anévrisme et l’artère nourricière ont été occlus au
moyen d’une injection endovasculaire de N-butyl cyanoacrylate. Résultats: L’anévrisme a été oblitéré avec succès
et, bien qu’un peu de colle ait pénétré dans l’artère cérébrale moyenne distale, il n’y a pas eu de changement du statut
neurologique de la patiente. Conclusions: Dans des cas bien choisis où les anévrismes lenticulostriés sont hors
d’atteinte de la chirurgie ou de la mise en place d’un serpentin par voie endovasculaire, l’oblitération par une colle
acrylique liquide peut être une option thérapeutique valable.
Can. J. Neurol. Sci. 2001; 28: 256-259
which consisted of right MCA M1 occlusion and ipsilateral moyamoya-
type vessels (Figure 1B). The remainder of the middle cerebral territory
on the right was collateralized from leptomeningeal vessels from the
right anterior (ACA) and posterior cerebral (PCA) arteries. The rest of
the vessels were normal. Over the following days, the patient gradually
regained consciousness and began to purposefully move the right side,
but remained paralysed on the left.
A follow-up angiogram was done 17 days after admission. This
clearly demonstrated a 4 mm aneurysm arising from one prominent
lenticulostriate branch of the right MCA (Figure 2A,B). This was
Direct surgery for aneurysms located in the basal ganglia
region, arising from moyamoya-type vessels, is difficult because
of the danger of damaging fragile collateral vessels. Surgery is
not recommended for small aneurysms located within the basal
ganglia.1 Platinum coils and particles have been used to treat
peripheral cerebral aneurysms associated with moyamoya
disease.2,3 We performed embolization of a lenticulostriate artery
aneurysm associated with ipsilateral middle cerebral artery
(MCA) occlusion using N-butyl cyanoacrylate (NBCA). We
believe this is the first report of this treatment for aneurysms
arising from moyamoya-type vessels.
CASE REPORT
From the Department of Diagnostic Radiology and Clinical Neurological Sciences,
University of Western Ontario, London, Ontario, (RL, DP); Division of Neurosurgery,
University of Alberta, MacKenzie Health Sciences Centre, Edmonton, Alberta (JMF)
Canada.
RECEIVED DECEMBER 18, 2000. ACCEPTED IN FINAL FORM APRIL 9, 2001.
Reprint requests to: David M. Pelz, Department of Diagnostic Radiology, London
Health Sciences Centre, University Campus, 339 Windermere Road, PO Box 5339,
London, Ontario, Canada N6A 5A5
A 57-year-old female presented with witnessed loss of consciousness
before a fall. On admission, she had decerebrate posturing and her
Glasgow Coma Scale (GCS) was 4. A computed tomography (CT)
scan revealed extensive intraparenchymal right ganglionic and
intraventricular hemorrhage (Figure 1A). The patient underwent an
urgent cerebral angiogram. This demonstrated a moyamoya-type pattern,
T25H6E CANADIAN JOURNAL OF NEUROLOGICAL SCIENCES
256

LE JOURNAL CANADIEN DES SCIENCES NEUROLOGIQUES
Figure 1A: Noncontrast computed
tomography (CT) scan on the day of
admission demonstrates intraparenchymal
right ganglionic and intraventricular
hemorrhage.
Figure 1B: Right common carotid digital subtraction
angiogram (DSA), anteroposterior (AP) projection,
on the same day reveals complete occlusion of the
middle cerebral artery (MCA) at the M1 segment
(small arrow) and ipsilateral moyamoya-type vessels
(large arrow).
considered to be the source of bleeding. Due to the difficulties associated
with surgery in this area, endovascular treatment was performed on day
21. At this time, the small aneurysm originating from the largest
lenticulostriate vessel was 1-2 mm larger than on the previous cerebral
angiogram (Figure 3).
A Tracker-10*microcatheter (*Boston Scientific/Target – Fremont,
CA) was advanced through a 6 French introducing catheter, into the
distal right internal carotid artery. Probing with a Transcend 10* soft tip
guide wire, resulted in supraselective catheterization of the large
lenticulostriate feeder to the aneurysm.
A supraselective contrast injection showed good filling of this
tortuous lenticulostriate branch which filled not only the aneurysm but
also a leash of small end arteries supplying the right lentiform nucleus
(Figure 4).
There was continuity of this vessel with the more distal right MCA.
An injection of approximately 0.3 cc of NBCA diluted 2:1 with lipiodol
was then made. Most of the glue deposited within the lenticulostriate
branch and the aneurysm. A small amount of glue did pass into the more
distal horizontal segment of the right MCA but this was non-occlusive.
A subsequent right internal carotid arteriogram showed complete
occlusion of the lenticulostriate branch and the aneurysm (Figure 5A,B).
There was still visualization of an extensive collateral network filling the
distal M1 segment of the right MCA, although the flow was less in the
sylvian segment as compared to the pre-embolization injection.
Extensive leptomeningeal supply from the right ACA and PCA was
unchanged. Following this, there was no worsening of the patient’s
neurological status as she remained completely hemiplegic on the left
side.
There was no significant change in the CT performed after the
embolization. This showed evolution of the intracerebral hemorrhage
with low density in the right basal ganglia region. Casts of embolic
material were present in the right M1 segment. The ventricular
hemorrhage had resolved and ventricular size was normal.
Before being discharged, after 24 days of hospitalization, the patient
was bright and alert. She followed commands briskly on the right side.
She remains spastic and hemiparetic on the left side. She has been in
rehabilitation since discharge from the hospital.
DISCUSSION
Aneurysms of lenticulostriate arteries are relatively rare.
They have been demonstrated in hypertensive patients, patients
with moyamoya disease and also in a patient with systemic lupus
erythematosus.4 These aneurysms may also be seen on feeding
arteries in patients with deep ganglionic-thalamic AVMs. In the
presence of a major intracranial vessel occlusion, aneurysms of
the lenticulostriate arteries are likely associated with moyamoya
disease. In classical moyamoya disease, prominent ganglionic
collateral vessels are usually associated with bilateral terminal
internal carotid artery occlusion. Our case most likely
represented a spontaneous MCA occlusion with development of
moyamoya-like ganglionic collateral vessels. The lenticulostriate
aneurysms in both conditions appear to behave similarly.
The incidence of aneurysms associated with moyamoya
disease is 1.5 to 12.9%.5 Kawaguchi et al1 demonstrated that
18% of 131 aneurysms associated with moyamoya disease were
located in the basal ganglia. There have been 24 reported cases
of spontaneous MCA occlusion. Nine of these cases presented
with cerebral aneurysms located in abnormal collateral vascular
networks and all the reported cases presented with cerebral
hemorrhage at their onset.
Takahashi et al6 reported a case of rapidly growing cerebral
aneurysm in the basal collateral vascular network associated
with spontaneous MCA occlusion in a 54-year-old female
patient. The aneurysm disappeared spontaneously shortly after
performing superficial temporal artery-middle cerebral artery
(STA-MCA) anastomosis. It has been suggested that STA-MCA
anastomosis in moyamoya disease reduces the incidence of
transient ischemic attacks, and also decreases the hemodynamic
stress on the abnormal basal vascular network and results in
reduction in size or thrombosis of the aneurysms.6 However,
Ikezaki et al7 have found that nearly 18% of the patients with
Volume 28, No. 3 – August 2001
257

Figure 2A: Right common carotid DSA, AP projection
on the 17th day post-admission demonstrates the MCA
M1 occlusion, ipsilateral moyamoya-type vessels and
a 4 mm aneurysm (large arrow) arising from the most
prominent lenticulostriate branch (small arrow).
Figure 2B: Magnified oblique view of the aneurysm
(arrow).
Figure 4: Supraselective contrast injection of the
large lenticulostriate artery, AP view shows filling of
this tortuous vessel (large arrow) and also filling of
multiple small end arteries (small arrows) which
supply the right lentiform nucleus. The aneurysm is
well seen (curved arrow), as is the distal M1 segment
of the MCA (arrowhead).
Figure 3: Right common carotid DSA, AP projection
on the 21st day of admission shows slight increase in
size of the aneurysm (arrow).
Figure 5A: Right common carotid DSA, AP view after
the embolization indicates complete obliteration of the
lenticulostriate branch and the aneurysm. An
extensive collateral network is seen filling the distal
horizontal segment of the right MCA (arrow).
Figure 5B: Extensive leptomeningeal supply from
the right ACA and PCA is appreciated later on the
same angiogram, with further reconstitution of distal
sylvian MCA branches.
258

LE JOURNAL CANADIEN DES SCIENCES NEUROLOGIQUES
hemorrhagic moyamoya disease experienced rebleeding
regardless of the treatment modalities. The prognosis of
rebleeding in patients with hemorrhagic moyamoya disease is
extremely poor, and several authors have come to the conclusion
that there is still no clear evidence that revascularization surgery
significantly prevents rebleeding in these patients.7,8,9 Factors
related to rebleeding and poorer outcome are sex (with women
being more susceptible), massive intracranial hemorrhage and
early recurrence. We believe that the risk of rebleeding in our
patient was high, given that she was a female, with a massive
intracranial hemorrhage and with a rapidly growing cerebral
aneurysm associated with spontaneous MCA occlusion. A
revascularization procedure was not a promising option for
treatment and it was not considered.
A direct surgical approach has been thought to be necessary
in treating aneurysms in patients with moyamoya disease and
associated subarachnoid hemorrhage.10 However, it has been
shown that compression of the perforating vessels around
aneurysms of the parent artery during surgical clipping can
induce circulatory compromise leading to postoperative
hemiplegia or severe brain edema.10 Furthermore, excessive
hypotension and decreased PCO2 in the blood during surgery
must be avoided, because patients with moyamoya disease are
particularly susceptible to cerebral ischemia. In a review of 111
cases of aneurysms associated with moyamoya disease,
Kawaguchi et al1 have suggested that direct surgery is not
recommended for aneurysms found in the basal ganglia or in the
collateral vessels. In our case, the fragility of adjacent vessels
and the danger of damaging important collateral vessels made us
consider an endovascular approach.
Guglielmi electrically detachable coils (GDC) have been used
in the treatment of major artery aneurysms associated with
moyamoya disease.11 There has been a case report of
endovascular treatment using platinum coils of a peripheral
artery aneurysm associated with moyamoya disease.2
Endovascular treatment of a small aneurysm of the right anterior
choroidal artery was performed with preservation of the parent
artery. We considered this a good treatment option, but we were
unable to place our microcatheter far enough into the feeding
artery to safely deploy a coil.
Endovascular embolization of rapidly growing aneurysms
arising from moyamoya-type vessels using the mixture of NBCA
and lipiodol has, to our knowledge, not been reported. We used
this approach because the microcatheter could only be placed in
the proximal segment of the parent vessel and not into the
aneurysm. We recognize the risk of NBCA either refluxing into
the proximal MCA or occluding collateral channels but, given
the fact that the patient was already hemiplegic on the left side,
we believed that little or no further clinical deterioration could be
caused with a slow and careful injection of a small volume of
liquid embolizing agent. We believed that urgent treatment of the
unstable aneurysm was necessary to prevent further bleeding.
We do not recommend this type of treatment as a routine
practice in aneurysms arising from moyamoya-type vessels. This
option can be considered when the aneurysm can not be directly
accessed for coil treatment and the likelihood of causing further
neurologic injury is low.
REFERENCES
1. Kawaguchi S, Sakaki T, Morimoto T, Kakizaki T, Kamada K.
Characteristics of intracranial aneurysms associated with
moyamoya disease. A review of 111 cases. Acta Neurochir
(Wien) 1996;138(11):1287-1294.
2. Sugiura Y, Matsuzawa Y. Endovascular treatment using platinum
coil of a peripheral artery aneurysm associated with moyamoya
disease: case report. No Shinkei Geka 1998;26(1):73-77.
3. Maekawa M, Nemoto S, Awaya S, Teramoto A. Moyamoya disease
with intraventricular hemorrhage due to rupture of lateral
posterior choroidal artery aneurysm: case report. No Shinkei
Geka 1999;27(11):1047-1051.
4. Kidoguchi J, Chiba M, Murakami T, et al. A case of systemic lupus
erythematosus associated with an aneurysm of the lenticulostriate
artery. No Shinkei Geka 1987;15(11):1221-1225.
5. Shibuya P, Hayashi N. A case of posterior cerebral artery aneurysm
associated with idiopathic bilateral internal carotid artery
occlusion: case report. Surg Neurol 1999;52:617-622.
6. Takahashi M, Fujimoto T, Suzuki R, et al. A case of spontaneous
middle cerebral artery occlusion associated with a cerebral
aneurysm angiographically disappearing after STA-MCA
anastomosis. No Shinkei Geka 1997;25(8):727-732.
7. Ikezaki K, Fukui M, Inamura T, et al. The current status of the
treatment for hemorrhagic type moyamoya disease based on a
1995 nationwide survey in Japan. Clin Neurol Neurosurg
1997;99(S2):183-186.
8. Choi JU, Kim DS, Kim EY, Lee KC. Natural history of moyamoya
disease: comparison of activity of daily living in surgery and
nonsurgery groups. Clin Neurol Neurosurg 1997;99(S2):11-18.
9. Yoshida Y, Yoshimoto T, Shirane R, Sakurai Y. Clinical course,
surgical management, and long-term outcome of moyamoya
patients with rebleeding after an episode of intracerebral
hemorrhage. An extensive follow-up study. Stroke 1999;30:2272-
2276.
10. Kodama N, Sato M, Sasaki T. Treatment of ruptured cerebral
aneurysm in moyamoya disease. Surg Neurol 1996;46:62-66.
11. Massoud TF, Guglielmi G, Viñuela F, Duckwiler GR. Saccular
aneurysms in Moyamoya disease: endovascular treatment using
electrically detachable coils. Surg Neurol 1994;41:462-467.
Volume 28, No. 3 – August 2001
259