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  • Author or Editor: Shinya Tokunaga x
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Kazushi Kinugasa, Ichiro Kamata, Nobuyuki Hirotsune, Koji Tokunaga, Kenji Sugiu, Akira Handa, Hiroyuki Nakashima, Takashi Ohmoto, Shinya Mandai and Yuzo Matsumoto

✓ Twelve patients with Hunt and Hess neurological Grades III to V underwent thrombosis of aneurysms using cellulose acetate polymer within 23 hours of aneurysm rupture. On computerized tomography (CT), nine of these patients had diffuse or localized thick subarachnoid blood clots, two had diffuse thin clots, and one had intraventricular clots. Immediately after thrombosis, all patients were administered tissue plasminogen activator (TPA) through spinal or ventricular catheters. The pressure of the lumbar cerebrospinal fluid was maintained at 100 to 150 mm H2O. The TPA was given as multiple injections of 2 mg on Day 0 and 1 to 2 mg on the following 1 to 2 days. In two patients the second injection of TPA was not given because of severe brain damage resulting from the initial subarachnoid hemorrhage. Ten patients showed complete clearance of the cisternal clot on CT within 72 hours after thrombosis. Seven partially thrombosed aneurysms and five multiple aneurysms were clipped during delayed surgery. Only one patient experienced mild vasospasm as shown on the follow-up angiogram. Eight patients improved clinically and had a good recovery, two had severe disability, and two died. Urgent thrombosis of a ruptured aneurysm followed by immediate postthrombotic administration of TPA may be a safe and reasonable means of preventing vasospasm and improving patient outcome.

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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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Kai Yamasaki, Chikako Kiyotani, Keita Terashima, Yuko Watanabe, Masayuki Kanamori, Yuhki Koga, Nobuhiro Hata, Fuminori Iwasaki, Hiroaki Goto, Katsuyoshi Koh, Jun Kurihara, Shinya Tokunaga, Yoshiki Arakawa, Daiichiro Hasegawa, Yoshiyuki Kosaka and Junichi Hara

OBJECTIVE

The prognosis of atypical teratoid/rhabdoid tumors (ATRTs) has improved in recent years with the use of multimodal therapy, mainly in cases not involving metastatic disease. The authors wanted to obtain historical control data and evaluate the suitable treatments in Japanese children with ATRTs that were proven negative for INI-1 immunostaining.

METHODS

The authors retrospectively collected clinical information on 38 pediatric patients with ATRTs treated from 2005 to 2016 and analyzed the data for this series.

RESULTS

The median age of the patient population was 1.3 years, and the male/female ratio was approximately 2:1. Twenty-three patients (60.5%) had metastases. The effects of treatment on prognosis were analyzed for 34 patients after exclusion of 4 patients who could not receive curative treatment. At a median follow-up of 40.9 months, the mean (± SD) progression-free survival (PFS) and overall survival (OS) were 66.6% ± 8.3% and 45.9% ± 8.7% at 2 years and 44.2% ± 9.9% and 34.2% ± 8.9% at 5 years, respectively. The metastasis stage at diagnosis (M0–1 vs M2–4) (HR 2.68, 95% CI 1.08–6.65; p = 0.0338) and gross tumor resection (yes vs no) (HR 3.49, 95% CI 1.01–12.1; p = 0.0481) were prognostic factors for PFS but not for OS. Postoperative chemotherapy was performed in all 34 cases. High-dose chemotherapy was performed in 19 (55.8%) of 34 patients and showed a positive impact on OS (HR 0.31, 95% CI 0.11–0.86; p = 0.0254); the most commonly used regimen was a double-conditioning regimen of thiotepa plus melphalan. Local radiotherapy had a positive impact on both PFS and OS; however, craniospinal irradiation (CSI) performed in 12 patients as the primary therapy was associated with a poor outcome. Disseminated recurrence within 12 months from diagnosis was the most common pattern of treatment failure regardless of CSI.

CONCLUSIONS

There has been an improvement in outcomes for pediatric ATRT patients since the introduction of multimodal therapy in Japan, mainly in patients without metastases. Even if selection bias is taken into consideration, CSI did not contribute to an improved prognosis. Novel treatment approaches are required for pediatric ATRT patients with metastases.