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Tomohito Hishikawa, Koji Tokunaga, Kenji Sugiu, and Isao Date

Object

There is no description of the change in the posterior cerebral artery (PCA) in the diagnostic criteria of moyamoya disease (MMD). However, PCAs are often involved in the clinical setting, and an understanding of the significance of PCA lesions is therefore of great importance when evaluating the disease progression and predicting prognosis. The aim of this study was to assess the difference in posterior circulation involvement in pediatric and adult patients with MMD.

Methods

The records of 120 consecutive patients with MMD were reviewed. The clinical manifestations at diagnosis were evaluated on the basis of symptoms and CT and MRI findings. The degree of steno-occlusive internal carotid artery (ICA) lesions and the existence of steno-occlusive PCA lesions were evaluated by observing a total of 240 ICAs and PCAs on angiography. Angiographic correlation between anterior and posterior circulation was assessed in pediatric and adult patients with MMD.

Results

Seventeen (26%) of 66 pediatric patients and 18 (33%) of 54 adult patients exhibited steno-occlusive PCA lesions. There was no significant difference in the prevalence of PCA lesions between pediatric and adult patients with MMD (p = 0.36). The prevalence of infarction in pediatric and adult patients with PCA involvement was significantly higher than that in pediatric and adult patients without PCA involvement (p = 0.0003 and p = 0.003, respectively). There was no significant difference in the distribution of infarction areas between pediatric and adult patients with PCA involvement (p = 0.62). On the basis of the staging system used, steno-occlusive lesions in ICAs ipsilateral to PCAs with lesions were in significantly advanced stages compared with lesions in ICAs ipsilateral to PCAs without lesions in both pediatric and adult cases (p < 0.0001 and p = 0.0008, respectively). Pediatric patients had less advanced steno-occlusive lesions in ICAs ipsilateral to PCAs with lesions compared with adults (p < 0.05).

Conclusions

The clinical significance of posterior circulation involvement in MMD was similar between pediatric and adult patients. The only significant difference was that less advanced ICA lesions could complicate posterior circulation involvement in pediatric patients.

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Takao Yasuhara, Takashi Tamiya, Kenji Sugiu, Satoshi Inoue, and Takashi Ohmoto

✓ The authors describe a case of de novo formation and rupture of an aneurysm located at the junction of the left internal carotid artery and the superior hypophyseal artery in a middle-aged woman 2 months after another aneurysm, located on the anterior communicating artery, had been clipped. This case is rare because of the short interval between the last angiographic study performed at the first operation and the diagnosis of the de novo aneurysm; in this case the interval was only 47 days, compared with other cases in the literature in which the intervals were 3 to 34 years. Aneurysms can enlarge considerably in 2 to 4 weeks and can rupture at or soon after their formation. This case provides insight into aneurysm formation and rupture.

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Kenji Sugiu, Jean-Baptiste Martin, Beatrix Jean, and Daniel A. Rüfenacht

✓ In this article the authors describe a rescue balloon procedure for coil implantation in three cases. In each patient, the coil seemed likely to unravel. The coils stretched when attempts were made to remove the partially implanted but trapped device. The inflation of a nondetachable microballoon in front of the aneurysm orifice allowed the surgeons to complete implantation of the coil and to avoid a more forceful and potentially harmful retrieval. This rescue balloon method may be useful for emergency situations, such as coil stretching with or without migration.

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Hernando Rafael

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Kenji Sugiu, Kazushi Kinugasa, Shinya Mandai, Koji Tokunaga, and Takashi Ohmoto

✓ Experimental aneurysms were created using a microsurgical technique to produce anastomosed venous pouches in the bilateral common carotid arteries of 12 dogs. The 24 aneurysms were then thrombosed via an endovascular approach with injection of a cellulose acetate polymer (CAP) solution that the authors have developed for use as a liquid thrombotic material. Angiography performed 1 to 4 weeks after CAP injection revealed complete thrombosis of the aneurysm with patency of the parent artery in 16 aneurysms. Histological analysis disclosed that the aneurysmal orifice in these cases was completely covered with newly formed endothelial cells 2 weeks after CAP thrombosis. Three other aneurysms exhibited parent artery occlusion caused by protrusion of the CAP mass through the aneurysmal orifice into the parent artery; this was thought to be caused by over-injection of the CAP solution. Histological analysis of the remaining five aneurysms, initially shown to have incomplete occlusion, revealed that they each possessed a residual neck that was partially covered with endothelial cells. No rupture of the aneurysms or migration of CAP into the distal arteries was observed.

These results suggest that using an endovascular approach, direct thrombosis of cerebral aneurysms with CAP is safe and effective. This technique may prove to be an alternative treatment for such aneurysms. However, there is a potential risk of regrowth or rupture of aneurysms that retain a residual neck and long-term follow-up studies will be required to evaluate this issue.

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Koji Tokunaga, Kenji Sugiu, Masahiro Kameda, Kyoji Sakai, Kaoru Terasaka, Toru Higashi, and Isao Date

✓ Intracerebral hemorrhage occurred in this 61-year-old woman with preexisting diplopia and proptosis. Results of angiography demonstrated a persistent primitive trigeminal artery (PPTA)—cavernous sinus fistula with cortical venous reflux. Two microcatheters were introduced transarterially through the PPTA into the two draining pathways in the cavernous sinus. Coils were delivered in both pathways simultaneously to prevent further venous overload on either path. The fistula was successfully occluded without complication while the PPTA was preserved. The authors describe this double-catheter technique for coil embolization of a fistula and review the literature concerning PPTA—cavernous sinus fistulas.

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Direct thrombosis of aneurysms with cellulose acetate polymer

Part II: Preliminary clinical experience

Kazushi Kinugasa, Shinya Mandai, Yoshinori Terai, Ichiro Kamata, Kenji Sugiu, Takashi Ohmoto, and Akira Nishimoto

✓ The authors report the treatment of seven intracranial aneurysms in six patients with direct infusion of cellulose acetate polymer solution, a new liquid thrombotic material. These aneurysms were considered inoperable because of their size or location, or because of the patient's neurological condition. This material avoids the difficulties associated with balloon occlusion, and completely fills even irregularly shaped aneurysms. Cellulose acetate polymer solution hardens in about 5 minutes and remains solid once inside the aneurysm. Because this technique is less invasive than surgery, it can be used for high-risk patients in the acute stage of subarachnoid hemorrhage. Transient motor aphasia occurred in one patient. A small residual neck, which caused rebleeding 3 months after the treatment, remained in another patient. This article describes the new material, the procedure for direct thrombosis, and preliminary clinical results.

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Keiichi Iseda, Shigeki Ono, Keisuke Onoda, Motoyoshi Satoh, Hiroaki Manabe, Mitsuhisa Nishiguchi, Kenji Takahashi, Koji Tokunaga, Kenji sugiu, and Isao Date

Object

Inflammation in the subarachnoid space and apoptosis of arterial endothelial cells have been implicated in the development of delayed cerebral vasospasm after subarachnoid hemorrhage (SAH). The authors investigated mechanisms of possible antivasospastic effects of N-benzyl-oxycarbonyl-Val-Ala-Asp-fluoromethylketone (Z-VAD-FMK), a caspase inhibitor that can inhibit both inflammatory and apoptotic systems, in animal models of SAH.

Methods

Rabbits were assigned to three groups of eight animals each and were subjected to SAH by injection of blood into the cisterna magna. The experiments were performed in the following groups: SAH only, SAH + vehicle, and SAH + Z-VAD-FMK. The Z-VAD-FMK (1 mg) or vehicle (5% dimethyl sulfoxide) was intrathecally administered before SAH induction. Diameters of the basilar artery (BA) were measured on angiograms obtained before and 2 days after SAH. The BA diameter on Day 2 was expressed as a percentage of that before SAH. Interleukin (IL)–1β in the cerebrospinal fluid (CSF) was examined using Western blotting, and brains were immunohistochemically examined for caspase-1 and IL-1β. In a separate experiment, 20 rats were subjected to SAH and their brains were immunohisto-chemically assessed for caspase-1, IL-1β, and macrophages.

Results

In rabbits, Z-VAD-FMK significantly attenuated cerebral vasospasm (the BA diameter on Day 2 in SAH-only, SAH + vehicle, and SAH + Z-VAD-FMK groups was 66.6 ± 3.2%, 66.3 ± 3.7%, and 82.6 ± 4.9% of baseline, respectively), and suppressed IL-1β release into the CSF and also suppressed immunoreactivities of caspase-1 and IL-1β in macrophages infiltrating into the subarachnoid space. Immunoreactivities for caspase-1 and IL-1β were observed in immunohistochemically proven infiltrating macrophages in rats.

Conclusions

These results indicate that caspase activation may be involved in the development of SAH-induced vasospasm through inflammatory reaction.

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Masafumi Hiramatsu, Kenji Sugiu, Tomoya Ishiguro, Hiro Kiyosue, Kenichi Sato, Keisuke Takai, Yasunari Niimi, and Yuji Matsumaru

OBJECTIVE

The aim of this retrospective multicenter cohort study was to assess the details of the angioarchitecture of arteriovenous fistulas (AVFs) at the craniocervical junction (CCJ) and to determine the associations between the angiographic characteristics and the clinical presentations and outcomes.

METHODS

The authors analyzed angiographic and clinical data for patients with CCJ AVFs from 20 participating centers that are members of the Japanese Society for Neuroendovascular Therapy (JSNET). Angiographic findings (feeding artery, location of AV shunt, draining vein) and patient data (age, sex, presentation, treatment modality, outcome) were tabulated and stratified based on the angiographic types of the lesions, as diagnosed by a member of the CCJ AVF study group, which consisted of a panel of 6 neurointerventionalists and 1 spine neurosurgeon.

RESULTS

The study included 54 patients (median age 65 years, interquartile range 61–75 years) with a total of 59 lesions. Five angiographic types were found among the 59 lesions: Type 1, dural AVF (22 [37%] of 59); Type 2, radicular AVF (17 [29%] of 59); Type 3, epidural AVF (EDAVF) with pial feeders (8 [14%] of 59); Type 4, EDAVF (6 [10%] of 59); and Type 5, perimedullary AVF (6 [10%] of 59). In almost all lesions (98%), AV shunts were fed by radiculomeningeal arteries from the vertebral artery that drained into intradural or epidural veins through AV shunts on the dura mater, on the spinal nerves, in the epidural space, or on the spinal cord. In more than half of the lesions (63%), the AV shunts were also fed by a spinal pial artery from the anterior spinal artery (ASA) and/or the lateral spinal artery. The data also showed that the angiographic characteristics associated with hemorrhagic presentations—the most common presentation of the lesions (73%)—were the inclusion of the ASA as a feeder, the presence of aneurysmal dilatation on the feeder, and CCJ AVF Type 2 (radicular AVF). Treatment outcomes differed among the angiographic types of the lesions.

CONCLUSIONS

Craniocervical junction AVFs commonly present with hemorrhage and are frequently fed by both radiculomeningeal and spinal pial arteries. The AV shunt develops along the C-1 or C-2 nerve roots and can be located on the spinal cord, on the spinal nerves, and/or on the inner or outer surface of the dura mater.

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Noboru Kusaka, Kenji Sugiu, Koji Tokunaga, Atsushi Katsumata, Ayumi Nishida, Katsunari Namba, Hirofumi Hamada, Hiroyuki Nakashima, and Isao Date

Object. Vascular endothelial growth factor (VEGF) is a secreted mitogen associated with angiogenesis. The conceptual basis for therapeutic angiogenesis after plasmid human VEGF gene (phVEGF) transfer has been established in patients presenting with limb ischemia and myocardial infarction. The authors hypothesized that overexpression of VEGF using a gene transfer method combined with indirect vasoreconstruction might induce effective brain angiogenesis in chronic cerebral hypoperfusion, leading to prevention of ischemic attacks.

Methods. A chronic cerebral hypoperfusion model induced by permanent ligation of both common carotid arteries in rats was used in this investigation. Seven days after induction of cerebral hypoperfusion, encephalomyosynangiosis (EMS) and phVEGF administration in the temporal muscle were performed. Fourteen days after treatment, the VEGF gene therapy group displayed numbers and areas of capillary vessels in temporal muscles that were 2.2 and 2.5 times greater, respectively, in comparison with the control group. In the brain, the number and area of capillary vessels in the group treated with the VEGF gene were 1.5 and 1.8 times greater, respectively, relative to the control group.

Conclusions. In rat models of chronic cerebral hypoperfusion, administration of phVEGF combined with indirect vasoreconstructive surgery significantly increased capillary density in the brain. The authors' results indicate that administration of phVEGF may be an effective therapy in patients with chronic cerebral hypoperfusion, such as those with moyamoya disease.