Filter

Refine by Publication

Refine by Date

—

Refine by Access

  • Print
  • Save

Browse

You are looking at 1 - 4 of 4 items for

  • Refine by Access: all x
  • By Author: Tanaka, Yoji x
  • By Author: Maehara, Taketoshi x
Clear All
Restricted access

Regression of periventricular anastomosis after indirect revascularization in pediatric patients with moyamoya disease

Elizabeth Yi Zheng, Shoko Hara, Motoki Inaji, Yoji Tanaka, Tadashi Nariai, and Taketoshi Maehara

OBJECTIVE

The aim of this study was to evaluate whether indirect revascularization in pediatric patients with moyamoya disease leads to periventricular anastomosis (PVA) regression, which is markedly developed in moyamoya vessels and is regarded as a risk factor for hemorrhage.

METHODS

Pediatric patients with moyamoya disease treated with indirect revascularization from 2011 to 2021 were included in this study. Magnetic resonance angiography and arterial spin labeling images acquired before and 1 year after surgery were assessed to obtain a visual scale of postoperative collateral artery formation, moyamoya vessels, PVA, and quantitative values of cerebral blood flow (CBF). The relationship between background information (age, sex, RNF213 p.R4810K variant status, and preoperative CBF) and postoperative collateral artery formation, as well as postoperative CBF improvement and regression of moyamoya vessels and PVA, was evaluated.

RESULTS

Of 89 hemispheres in 58 patients (34 females; mean [SD] patient age 8.0 [3.4] years), 74.2% showed good postoperative collateral artery formation and a significant increase in CBF (p < 0.001). Postoperative PVA showed significant regression (postoperative score 1.46 [1.06] vs 2.02 [1.69], p = 0.001), especially in those arising from choroidal arteries (postoperative score 0.28 [0.50] vs 0.72 [0.67], p < 0.001). Compared with hemispheres without good collateral artery formation, those with good collateral artery formation were more likely to show a higher increase in CBF (9.74 [12.44] ml/min/100 g vs −4.86 [9.68] ml/min/100 g, p < 0.001) and regression of PVA (54.5% [36/66] vs 30.4% [7/23], p = 0.015). Although not statistically significant, patients with postoperative PVA progression were younger than those with regression (6.75 [3.03] years vs 8.18 [3.17] years, p = 0.188), and patients with the RNF213 p.R4810K variant were more likely to show regression (28/57 [49.1%] hemispheres vs 5/13 [38.5%] hemispheres, p = 0.069).

CONCLUSIONS

Indirect revascularization in pediatric patients with moyamoya disease resulted in good collateral extracranial artery formation and an increase in CBF and PVA regression, especially of vessels arising from choroidal arteries. With good postoperative collateral artery development, patients were more likely to show improved CBF and regression of moyamoya vessels, including PVA. Whether postoperative PVA changes reduce future hemorrhage risk requires further investigation.

In Journal of Neurosurgery: Pediatrics Volume 32: Issue 6 (Dec 2023)

Free access

Absence of the RNF213 p.R4810K variant may indicate a severe form of pediatric moyamoya disease in Japanese patients

Shoko Hara, Maki Mukawa, Hiroyuki Akagawa, Thiparpa Thamamongood, Motoki Inaji, Yoji Tanaka, Taketoshi Maehara, Hidetoshi Kasuya, and Tadashi Nariai

OBJECTIVE

The authors’ objective was to investigate the influence of the RNF213 p.R4810K variant on the clinical presentation and outcomes of Japanese pediatric patients with moyamoya disease.

METHODS

A total of 129 Japanese patients with pediatric-onset moyamoya disease (onset age ≤ 15 years) who visited the authors’ department from 2012 to 2020 participated in this study. After RNF213 p.R4810K genotyping of each patient was performed, the relationship between genotype and clinical presentation or outcomes, including onset age, initial presentation, surgical outcomes, and subsequent cerebrovascular events, was evaluated. Patients without the p.R4810K variant were tested for RNF213 variants other than p.R4810K. The authors especially focused on the results of patients who presented with moyamoya disease at younger than 1 year of age (infantile onset).

RESULTS

Compared with the patients with heterozygous variants, patients without the p.R4810K variant were younger at onset (7.1 ± 3.7 vs 4.4 ± 0.9 years), and all 4 patients with infantile onset lacked the p.R4810K variant. A greater proportion of patients without the p.R4810K variant presented with infarction than patients with the heterozygous variant (24.0% vs 7.6%) and a decreased proportion presented with transient ischemic attack (36.0% vs 71.7%). No significant correlation was observed between p.R4810K genotype and clinical outcomes, including surgical outcomes and subsequent cerebrovascular events; however, a decreased proportion of patients without the p.R4810K variant had good surgical outcomes compared with that of patients with the heterozygous variant (76.5% vs 92.2%). Among the 25 patients without the p.R4810K variant, 8 rare variants other than p.R4810K were identified. Three of 4 patients with infantile onset had RNF213 variants other than p.R4810K, which had a more severe functional effect on this gene than p.R4810K.

CONCLUSIONS

Absence of the RNF213 p.R4810K variant may be a novel biomarker for identification of a severe form of pediatric moyamoya disease.

In Journal of Neurosurgery: Pediatrics Volume 29: Issue 1 (Jan 2022)

Restricted access

Long-term follow-up of surgically treated juvenile patients with moyamoya disease

Clinical article

Maki Mukawa, Tadashi Nariai, Yoshiharu Matsushima, Yoji Tanaka, Motoki Inaji, Taketoshi Maehara, Masaru Aoyagi, and Kikuo Ohno

Object

Surgical revascularization is considered an effective treatment for juvenile patients with moyamoya disease (MMD). Yet the long-term outcome in surgically treated patients still needs to be clarified. More than 30 years have passed since the authors' department started intensively treating pediatric patients with MMD using indirect anastomosis techniques. In this study the authors surveyed the current status of these patients.

Methods

Activities of daily living (ADLs) were surveyed and present clinical status was assessed based on the modified Rankin Scale (mRS). Cerebrovascular events subsequent to surgical treatment were also recorded.

Results

Since 1979, 208 patients younger than 19 years of age with MMD were surgically treated and followed up for > 3 years. Data were available on 172 patients (83%), who had been followed up for a mean of 14.3 years (range 3–32 years). Activity of daily living outcomes were as follows: 138 patients (80.2%) had mRS scores of 0–2, 29 (16.9%) a score of 3, 1 (0.6%) a score of 4, 1 (0.6%) a score of 5, and 3 (1.7%) a score of 6. Cerebrovascular events occurred 8 or more years after surgery in 6 patients (3.4%), that is, 6 hemorrhages and 3 infarctions. The cumulative risk of late-onset stroke at 10, 20, and 30 years after surgical intervention was 0.8%, 6.3%, and 10.0%, respectively.

Conclusions

This long-term survey demonstrated that most surgically treated pediatric patients with MMD maintain good ADL outcomes. However, a significant number of new cerebrovascular events occurred more than 10 years after the initial surgery. Additional follow-up will help to identify which events may occur during the adult years of patients treated as children.

In Journal of Neurosurgery: Pediatrics Volume 10 (2012): Issue 5 (Nov 2012)

Restricted access

Glioma surgery using a multimodal navigation system with integrated metabolic images

Clinical article

Yoji Tanaka, Tadashi Nariai, Toshiya Momose, Masaru Aoyagi, Taketoshi Maehara, Toshiki Tomori, Yoshikazu Yoshino, Tsukasa Nagaoka, Kiichi Ishiwata, Kenji Ishii, and Kikuo Ohno

Object

A multimodal neuronavigation system using metabolic images with PET and anatomical images from MR images is described here for glioma surgery. The efficacy of the multimodal neuronavigation system was evaluated by comparing the results with that of the conventional navigation system, which routinely uses anatomical images from MR and CT imaging as guides.

Methods

Thirty-three patients with cerebral glioma underwent 36 operations with the aid of either a multimodal or conventional navigation system. All of the patients were preliminarily examined using PET with l-methyl-[ C] methionine (MET) for surgical planning. Seventeen of the operations were performed with the multimodal navigation system by integrating the MET-PET images with anatomical MR images. The other 19 operations were performed using a conventional navigation system based solely on MR imaging.

Results

The multimodal navigation system proved to be more useful than the conventional navigation system in determining the area to be resected by providing a clearer tumor boundary, especially in cases of recurrent tumor that had lost a normal gyral pattern. The multimodal navigation system was therefore more effective than the conventional navigation system in decreasing the mass of the tumor remnant in the resectable portion. A multivariate regression analysis revealed that the multimodal navigation system–guided surgery benefited patient survival significantly more than the conventional navigation–guided surgery (p = 0.016, odds ratio 0.52 [95% confidence interval 0.29–0.88]).

Conclusions

The authors' preliminary intrainstitutional comparison between the 2 navigation systems suggested the possible premise of multimodal navigation. The multimodal navigation system using MET-PET fusion imaging is an interesting technique that may prove to be valuable in the future.

In Journal of Neurosurgery Volume 110: Issue 1 (Jan 2009)