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Takeshi Funaki, Jun C. Takahashi, Yasushi Takagi, Takayuki Kikuchi, Kazumichi Yoshida, Takafumi Mitsuhara, Hiroharu Kataoka, Tomohisa Okada, Yasutaka Fushimi, and Susumu Miyamoto

OBJECT

Unstable moyamoya disease, reasonably defined as cases exhibiting either rapid disease progression or repeated ischemic stroke, represents a challenge in the treatment of moyamoya disease. Despite its overall efficacy, direct bypass for such unstable disease remains controversial in terms of safety. This study aims to reveal factors associated with unstable disease and to assess its impact on postoperative silent or symptomatic ischemic lesions.

METHODS

This retrospective cohort study included both pediatric and adult patients with moyamoya disease who had undergone 140 consecutive direct bypass procedures at Kyoto University Hospital. “Unstable moyamoya disease” was defined as either the rapid progression of a steno-occlusive lesion or repeat ischemic stroke, either occurring within 6 months of surgery. The extent of progression was determined through a comparison of the findings between 2 different MR angiography sessions performed before surgery. The clinical variables of the stable and unstable disease groups were compared, and the association between unstable disease and postoperative diffusion-weighted imaging (DWI)–detected lesion was assessed through univariate and multivariate analyses with generalized estimating equations.

RESULTS

Of 134 direct bypass procedures performed after patients had undergone at least 2 sessions of MR angiography, 24 (17.9%) were classified as cases of unstable disease. Age younger than 3 years (p = 0.029), underlying disease causing moyamoya syndrome (p = 0.049), and radiographic evidence of infarction (p = 0.030) were identified as factors associated with unstable disease. Postoperative DWI-defined lesions were detected after 13 of 140 procedures (9.3%), although only 4 lesions (2.9%) could be classified as a permanent complication. The incidence of postoperative DWI-detected lesions in the unstable group was notable at 33.3% (8 of 24). Univariate analysis revealed that unstable disease (p < 0.001), underlying disease (p = 0.028), and recent stroke (p = 0.012) were factors associated with DWI-detected lesions. Unstable disease remained statistically significant after adjustment for covariates in both the primary and sensitivity analyses (primary analysis: OR 6.62 [95% CI 1.79–24.5]; sensitivity analysis: OR 5.36 [95% CI 1.47–19.6]).

CONCLUSIONS

Unstable moyamoya disease, more prevalent in younger patients and those with underlying disease, is a possible risk factor for perioperative ischemic complications. Recognition of unstable moyamoya disease may contribute to an improved surgical result through focused perioperative management based on appropriate surgical risk stratification.

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Takeshi Funaki, Jun C. Takahashi, Kazumichi Yoshida, Yasushi Takagi, Yasutaka Fushimi, Takayuki Kikuchi, Yohei Mineharu, Tomohisa Okada, Takaaki Morimoto, and Susumu Miyamoto

OBJECT

The authors’ aim in this paper was to determine whether periventricular anastomosis, a novel term for the abnormal collateral vessels typical of moyamoya disease, is reliably measured with MR angiography and is associated with intracranial hemorrhage.

METHODS

This cross-sectional study sampled consecutive patients with moyamoya disease or moyamoya syndrome at a single institution. Periventricular anastomoses were detected using MR angiography images reformatted as sliding-thin-slab maximum-intensity-projection coronal images and were scored according to 3 subtypes: lenticulostriate, thalamic, and choroidal types. The association between periventricular anastomosis and hemorrhagic presentation at onset was evaluated using multivariate analyses.

RESULTS

Of 136 eligible patients, 122 were analyzed. Eighteen (14.8%) patients presented with intracranial hemorrhage with neurological symptoms at onset. Intra- and interrater agreement for rating of the periventricular anastomosis score was good (κw = 0.65 and 0.70, respectively). The prevalence of hemorrhagic presentation increased with the periventricular anastomosis score: 2.8% for Score 0, 8.8% for Score 1, 18.9% for Score 2, and 46.7% for Score 3 (p < 0.01 for trend). Univariate analysis revealed that age (p = 0.02) and periventricular anastomosis score (p < 0.01) were factors tentatively associated with hemorrhagic presentation. The score remained statistically significant after adjustment for age (OR 3.38 [95% CI 1.84–7.00]).

CONCLUSIONS

The results suggest that periventricular anastomosis detected with MR angiography can be scored with good intra- and interrater reliability and is associated with hemorrhagic presentation at onset in moyamoya disease. The clinical utility of periventricular anastomosis as a predictor for hemorrhage should be validated in further prospective studies.

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Tao Yang, Kazumichi Yoshida, Takakuni Maki, Yasutaka Fushimi, Kiyofumi Yamada, Masakazu Okawa, Yu Yamamoto, Naoki Takayama, Keita Suzuki, and Susumu Miyamoto

OBJECTIVE

Carotid webs (CWs) have increasingly been recognized as a cause of recurrent ischemic stroke. However, the natural history and clinical course of CWs remain unclear. The authors aimed to clarify the prevalence, imaging features, and optimal treatment of CWs in a Japanese cohort study.

METHODS

A series of 444 consecutive Japanese patients who had undergone CTA of the head and neck between April 2011 and October 2016 was retrospectively reviewed. CW was diagnosed on CT angiograms as a membrane-like intraluminal filling defect along the posterior wall of the carotid bulb or the origin of the internal carotid artery (ICA) on oblique sagittal images and a corresponding thin septum on axial images.

RESULTS

Two patients with CWs were identified among 132 patients with suspected stroke. The prevalence of CWs among symptomatic patients with suspected stroke was 1.5%. The prevalence of asymptomatic CWs was 2.2% (7 of 312 cases). The CWs were located in the posterior wall of the carotid bulb in 7 patients and just distal to the ICA origin in 2 patients. There were no apparent differences in the location or lesion length between symptomatic and asymptomatic CWs. Four of the 7 asymptomatic CWs remained asymptomatic for at least 2 years of follow-up. Two patients with symptomatic CWs developed recurrent cerebral infarction and transient ischemic attack despite being on a regimen of oral antiplatelet agents, and carotid endarterectomy was performed as radical treatment. Patients with CWs were younger than controls (median age 55 vs 69 years, p = 0.003) and were less frequently male than controls (33% vs 72%, p = 0.025). CW cases showed significantly fewer common atherosclerosis risk factors than the control group (p < 0.05).

CONCLUSIONS

Although limited to CTA patients, this study reported on the prevalence and common site of CWs, focusing on symptomatic and asymptomatic Japanese patients. Extensive cross-sectional and prospective observational studies are warranted to elucidate the overall prevalence and natural history of CWs.

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Ken-Ichiro Kikuta, Yasushi Takagi, Kazuhiko Nozaki, Takashi Hanakawa, Tsutomu Okada, Nobuhro Mikuni, Yukio Miki, Yasutaka Fushimi, Akira Yamamoto, Keisuke Yamada, Hidenao Fukuyama, and Nobuo Hashimoto

Object. The aim of this study was to investigate the incidence of asymptomatic microbleeds (MBs) in patients with moyamoya disease (MMD) by using a 3-tesla magnetic resonance (MR) imaging unit.

Methods. Data on 63 patients hospitalized with MMD between 1999 and 2004 were retrospectively examined to determine the incidence of asymptomatic MBs. Gradient-echo T2*-weighted MR imaging studies obtained using 3- and 1.5-tesla units were available in 25 patients. These patients consisted of five men and 20 women, ranging in age from 17 to 66 years (mean age 41 ± 14 years). Ischemic MMD was diagnosed in 18 patients, and hemorrhagic MMD in seven. The incidence of MBs was also evaluated using the same 3-tesla MR imaging unit in 34 healthy volunteers including seven men and 27 women, ranging in age from 18 to 71 years (mean age 33 ± 12 years). Using the 3-tesla MR unit, asymptomatic MBs were demonstrated in 11 patients (44%); they were detected in seven patients (28%) by using the 1.5-tesla unit. In the 3-tesla MR studies in healthy individuals, MBs were found in two patients (5.8%). Based on 3-tesla MR studies, the incidence of MBs was significantly higher in patients with MMD compared with that in healthy individuals. Asymptomatic MBs were demonstrated in eight (44%) of 18 patients with ischemic MMD and three (43%) of seven patients with hemorrhagic MMD.

Conclusions. Microbleeds are significantly more common in patients with MMD than in healthy individuals regardless of the disease type. The evaluation of MBs with T2*-weighted 3-tesla MR imaging might contribute to the treatment of MMD.