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Hyun-Seung Kang, Dong Gyu Kim and Dae Hee Han

✓ The authors report on a patient who presented with a large symptomatic glial cyst of the pineal gland communicating with the third ventricle. The hole between the ventricle and the cyst, suspected on magnetic resonance images, was found at surgery. The to-and-fro flow mechanism is considered to be involved in the pathogenesis of growth in this unusual large glial cyst of the pineal gland, although this mechanism cannot be applied universally.

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Hyun-Seung Kang, Moon Hee Han, Bae Ju Kwon, Byung-Woo Yoon and Kee-Hyun Chang

✓ The concept of a cerebrofacial arteriovenous metameric syndrome (CAMS) recently has been posited. A 22-year-old woman presented with gait ataxia. She had unilateral angiomatous lesions involving the lower face, the auricle, and the retromastoid area, as well as the tongue on the right side. Angiography revealed the presence of extensive arteriovenous malformations (AVMs) in the ipsilateral cerebellum and pons, in addition to those located in the facial region. The authors report a case of multiple AVMs of the face, cerebellum, and pons, corresponding to CAMS Type 3, and include a review of the literature.

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Chang-Hyun Lee, Jaebong Lee, James D. Kang, Seung-Jae Hyun, Ki-Jeong Kim, Tae-Ahn Jahng and Hyun-Jib Kim

OBJECT

Posterior cervical surgery, expansive laminoplasty (EL) or laminectomy followed by fusion (LF), is usually performed in patients with multilevel (≥ 3) cervical spondylotic myelopathy (CSM). However, the superiority of either of these techniques is still open to debate. The aim of this study was to compare clinical outcomes and postoperative kyphosis in patients undergoing EL versus LF by performing a meta-analysis.

METHODS

Included in the meta-analysis were all studies of EL versus LF in adults with multilevel CSM in MEDLINE (PubMed), EMBASE, and the Cochrane library. A random-effects model was applied to pool data using the mean difference (MD) for continuous outcomes, such as the Japanese Orthopaedic Association (JOA) grade, the cervical curvature index (CCI), and the visual analog scale (VAS) score for neck pain.

RESULTS

Seven studies comprising 302 and 290 patients treated with EL and LF, respectively, were included in the final analyses. Both treatment groups showed slight cervical lordosis and moderate neck pain in the baseline state. Both groups were similarly improved in JOA grade (MD 0.09, 95% CI −0.37 to 0.54, p = 0.07) and neck pain VAS score (MD −0.33, 95% CI −1.50 to 0.84, p = 0.58). Both groups evenly lost cervical lordosis. In the LF group lordosis seemed to be preserved in long-term follow-up studies, although the difference between the 2 treatment groups was not statistically significant.

CONCLUSIONS

Both EL and LF lead to clinical improvement and loss of lordosis evenly. There is no evidence to support EL over LF in the treatment of multilevel CSM. Any superiority between EL and LF remains in question, although the LF group shows favorable long-term results.

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Suk-Hyung Kang, Seung-Chul Rhim, Sung-Woo Roh, Sang-Ryong Jeon and Hyun-Chul Baek

Object

The authors studied cervical range of motion (ROM) before and after cervical laminoplasty to determine factors associated with cervical ROM in patients with cervical myelopathy.

Methods

Between July 2003 and August 2005, 20 patients underwent a modified Hirabayashi-type unilateral open-door laminoplasty to treat multilevel cervical spondylosis or ossification of the posterior longitudinal ligament (OPLL). Clinically, the authors assessed Japanese Orthopaedic Association (JOA) score, duration of symptoms, disease entity, and the age and sex of patients to ascertain the relation of these factors to ROM before and after cervical laminoplasty. Intraoperative findings such as ligament detachment from the C-2 spinous process and cervicothoracic junction involvement were noted. Radiological and imaging findings such as the length of the lesion, cervical axial canal area, antero-posterior (AP) diameter of the cervical canal, angle of the opened lamina after surgery, cervical sagittal angles, cervical curvature index (CCI), and signal change of the cord on magnetic resonance imaging were evaluated.

The mean follow-up period was 19.45 months (range 13–38 months). The preoperative average ROM in 18 patients (after excluding two patients with trauma) was 36.73 ± 15.73°; postoperatively it was 25.24 ± 16.06°. Thus, ROM decreased by 9.64 ± 10.09° (31.80%) after surgery (p = 0.002), reflecting the mean in the same 18 patients. Preoperative ROM was related to the age of patients, CCI, preoperative JOA score, and AP diameter of the cervical canal. In cases of OPLL the ROM was lower than that in cases of spondylosis. Postoperative cervical ROM was related to preoperative ROM, postoperative AP diameter of the cervical canal, laminar angle, patient age, and follow-up duration. None of the studied parameters, however, correlated with a decreased cervical ROM.

Conclusions

Cervical ROM was reduced after cervical laminoplasty. Postlaminoplasty cervical ROM had a positive correlation with extended motion; however, gradually it became reduced. In this study, no correlative factor was associated with a reduction in cervical ROM. Further study is also needed.

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Jin Pyeong Jeon, Jeong Eun Kim, Jun Hyong Ahn, Won-Sang Cho, Young Dae Cho, Young-Je Son, Jae Seung Bang, Hyun-Seung Kang, Chul-Ho Sohn, Hyun-Tai Chung, Chang Wan Oh and Dong Gyu Kim

OBJECT

Treatment strategies for venous-predominant arteriovenous malformation (vp-AVM) remain unclear due to the limited number of cases and a lack of long-term outcomes. The purpose of this study was to report the authors’ experience with treatment outcomes with a review of the pertinent literature in patients with vp-AVM.

METHODS

Medical and radiological data from 1998 to 2011 were retrospectively evaluated. The degree of the arteriovenous (AV) shunt was categorized into 2 groups, a high- and low-flow AV shunt based on the angiographic findings.

RESULTS

Sixteen patients with a mean age of 45.3 years (range 16–78 years) and a mean follow-up of 79.9 months (range 25–264 months) were examined. Symptomatic lesions were noted in 13 patients: intracranial hemorrhage (ICH) in 9, seizure in 1, and headache in 3. A high-flow shunt was observed on angiography in 13 patients. Among these 13 patients, 12 patients were symptomatic. Nine patients presenting with ICH underwent hematoma removal with additional Gamma Knife surgery (GKS; n = 4), GKS only (n = 2), or conservative treatment (n = 3). The 3 asymptomatic patients received conservative treatment, and 1 rebleeding episode was observed. Seven of 8 patients who underwent GKS as an initial or secondary treatment modality experienced a marked reduction in the AV shunt on follow-up angiography, but complete obliteration was not observed.

CONCLUSIONS

Poor lesion localization makes a vp-AVM challenging to treat. Symptomatic patients with a high-flow shunt are supposedly best treated with GKS, despite the fact that only 87.5% of the vp-AVMs treated this way showed a reduction in the malformation volume, and none were cured.

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Eung Koo Yeon, Young Dae Cho, Dong Hyun Yoo, Su Hwan Lee, Hyun-Seung Kang, Jeong Eun Kim, Won-Sang Cho, Hyun Ho Choi and Moon Hee Han

OBJECTIVE

The authors conducted a study to ascertain the long-term durability of coiled aneurysms completely occluded at 36 months’ follow-up given the potential for delayed recanalization.

METHODS

In this retrospective review, the authors examined 299 patients with 339 aneurysms, all shown to be completely occluded at 36 months on follow-up images obtained between 2011 and 2013. Medical records and radiological data acquired during the extended monitoring period (mean 74.3 ± 22.5 months) were retrieved, and the authors analyzed the incidence of (including mean annual risk) and risk factors for delayed recanalization.

RESULTS

A total of 5 coiled aneurysms (1.5%) occluded completely at 36 months showed recanalization (0.46% per aneurysm-year) during the long-term surveillance period (1081.9 aneurysm-years), 2 surfacing within 60 months and 3 developing thereafter. Four showed minor recanalization, with only one instance of major recanalization. The latter involved the posterior communicating artery as an apparent de novo lesion, arising at the neck of a firmly coiled sac, and was unrelated to coil compaction or growth. Additional embolization was undertaken. In a multivariate analysis, a second embolization for a recurrent aneurysm (HR = 22.088, p = 0.003) independently correlated with delayed recanalization.

CONCLUSIONS

Almost all coiled aneurysms (98.5%) showing complete occlusion at 36 months postembolization proved to be stable during extended observation. However, recurrent aneurysms were predisposed to delayed recanalization. Given the low probability yet seriousness of delayed recanalization and the possibility of de novo aneurysm formation, careful monitoring may be still considered in this setting but at less frequent intervals beyond 36 months.

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Hyun Ho Choi, Young Dae Cho, Dong Hyun Yoo, Su Hwan Lee, Eung Koo Yeon, Hyun-Seung Kang, Won-Sang Cho, Jeong Eun Kim and Moon Hee Han

OBJECTIVE

In the presence of symmetric A1 flow, the safety and efficacy of compromising the anterior communicating artery (ACoA) during coil embolization of ACoA aneurysms has yet to be evaluated. Herein, the authors describe their experience, focusing on procedural safety.

METHODS

Between October 2012 and July 2017, 285 ACoA aneurysms with symmetric A1 flows were treated at the authors’ institution by endovascular coil embolization. Clinical and angiographic outcome data were subjected to binary logistic regression analysis.

RESULTS

ACoA compromise was chosen in the treatment of 71 aneurysms (24.9%), which were completely (n = 15) or incompletely (n = 56) compromised. In the remaining 214 lesions, the ACoA was preserved. Although 9 patients (3.2%) experienced procedure-related thromboembolisms (compromised, 4; preserved, 5), all but 1 patient (with ACoA compromise) were asymptomatic. In multivariate analysis, subarachnoid hemorrhage at presentation was the sole independent risk factor for thromboembolism (OR 15.98, p < 0.01), with ACoA compromise being statistically unrelated. In 276 aneurysms (96.8%) with follow-up of > 6 months (mean 20.9 ± 13.1 months, range 6–54 months), recanalization was confirmed in 21 (minor, 15; major, 6). A narrow (≤ 4 mm) saccular neck (p < 0.01) and ACoA compromise (p = 0.04) were independently linked to prevention of recanalization.

CONCLUSIONS

During coil embolization of ACoA aneurysms, the ACoA may be compromised without serious complications if A1 flows are symmetric. This approach may also confer some long-term protection from recanalization, serving as a valid treatment option for such lesions.

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Hyun-Seung Kang, Youn-Joo Moon, Young-Yim Kim, Woong-Yang Park, Ae Kyung Park, Kyu-Chang Wang, Jeong Eun Kim, Ji Hoon Phi, Ji Yeoun Lee and Seung-Ki Kim

Object

Moyamoya disease (MMD) is a cerebrovascular occlusive disease affecting bilateral internal carotid termini. Smooth-muscle cells are one of the major cell types involved in this disease process. The characteristics of circulating smooth-muscle progenitor cells (SPCs) in MMD are poorly understood. The authors purified SPCs from the peripheral blood of patients with MMD and sought to identify differentially expressed genes (DEGs) in SPCs from these patients.

Methods

The authors cultured and isolated SPCs from the peripheral blood of patients with MMD (n = 25) and healthy control volunteers (n = 22). After confirmation of the cellular phenotype, RNA was extracted from the cells and DEGs were identified using a commercially available gene chip. Real-time quantitative reverse transcription polymerase chain reaction was performed to confirm the putative pathogenetic DEGs.

Results

The SPC-type outgrowth cells in patients with MMD invariably showed a hill-and-valley appearance under microscopic examination, and demonstrated high α–smooth muscle actin, myosin heavy chain, and calponin expression (96.5% ± 2.1%, 42.8% ± 18.6%, and 87.1% ± 8.2%, respectively), and minimal CD31 expression (less than 1%) on fluorescence-activated cell sorter analysis. The SPCs in the MMD group tended to make more irregularly arranged and thickened tubules on the tube formation assay. In the SPCs from patients with MMD, 286 genes (124 upregulated and 162 downregulated) were differentially expressed; they were related to cell adhesion, cell migration, immune response, and vascular development.

Conclusions

With adequate culture conditions, SPCs could be established from the peripheral blood of patients with MMD. These cells showed specific DEGs compared with healthy control volunteers. This study provides a novel experimental cell model for further research of MMD.

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Tackeun Kim, O-Ki Kwon, Jae Seung Bang, Heeyoung Lee, Jeong Eun Kim, Hyun-Seung Kang, Won-Sang Cho and Chang Wan Oh

OBJECTIVE

Brain arteriovenous malformation (BAVM) is a rare cerebrovascular disease that causes intracranial hemorrhage. Although several reports have demonstrated the epidemiological features of BAVM in Western countries, no epidemiological investigations regarding BAVM have been reported in Korea. The authors aimed to investigate the national epidemiology of ruptured BAVM in a Korean population.

METHODS

The authors used data from the National Health Insurance Service–National Sample Cohort (NHIS-NSC). The original cohort included approximately 1 million individuals (2% of the Korean population) with 12-year claim data (2002–2013). To construct an initial cohort for investigation, the authors selected 1,016,820 registered individuals in 2005. Subjects with a history of cerebrovascular disease (code I6xx) and BAVM (Q282) between 2002 and 2004 were washed-out to identify incident cases. During the 9-year follow-up (2005–2013), the incidence of BAVM was calculated using the earliest date of diagnosis of ruptured or unruptured BAVM. Direct standardization was applied to the crude incidence. Mortality and disability were evaluated using registration data. Related diagnostic procedures were also analyzed.

RESULTS

A total of 8,802,696 person-years of observation were noted. During observation, 308 patients were diagnosed with a ruptured BAVM. The crude incidence of ruptured BAVM was 3.5 per 100,000 person-years. There was no sex difference in incidence. The mortality rate for patients with a ruptured BAVM 1 month after diagnosis was 12.7%. At 1-year and 5-year follow-up examinations, mortality rates were 17.2% and 22.1%, respectively. Severe disability–free survival rates of patients with ruptured AVMs were 75.3% and 69.8% at 1-year and 5-year follow-up, respectively.

CONCLUSIONS

The standardized incidence of ruptured BAVMs was 3.6 per 100,000 person-years in Korea. Ruptured BAVMs resulted in high mortality and disability rates.

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Tackeun Kim, Chang Wan Oh, O-Ki Kwon, Gyojun Hwang, Jeong Eun Kim, Hyun-Seung Kang, Won-Sang Cho and Jae Seung Bang

OBJECT

Moyamoya disease (MMD) is a progressive disease that can cause recurrent stroke. The authors undertook this retrospective case-control study with a large sample size in an attempt to assess the efficacy of direct or combined revascularization surgery for ischemia in adults with MMD.

METHODS

The authors investigated cases involving patients with moyamoya disease presenting with ischemia who visited Seoul National University Bundang Hospital and Seoul National University Hospital between 2000 and 2014. Among 441 eligible patients, 301 underwent revascularization surgery and 140 were treated conservatively. Variables evaluated included age at diagnosis, sex, surgical record, Suzuki stage, and occurrence of stroke. Patients were stratified into 2 groups based on whether or not they had undergone revascularization surgery. Actuarial 1-, 5-, and 10-year stroke rates were calculated using the life table method. Risk factor analysis for 5-year stroke occurrence was conducted with multivariate regression.

RESULTS

Of the 441 patients, 301 had been surgically treated (revascularization group) and 140 had not (control group). The mean follow-up durations were 45 and 77 months, respectively. The actuarial 10-year cumulative incidence rate for any kind of stroke was significantly lower in the revascularization group (9.4%) than in the control group (19.6%) (p = 0.041); the relative risk reduction (RRR) was also superior (52.0%) in the revascularization group, and the number needed to treat was 10. The 10-year rate of ischemic stroke was greater (13.3%) in the control group than in the revascularization group (3.9%) (p = 0.019). The RRR for ischemic stroke in the revascularization group was 70.7%, and the number needed to treat was 11. However, the actuarial 1- and 5-year rates of ischemic stroke did not significantly differently between the groups. Overall, revascularization surgery was shown to be an independent protective factor, as revealed by multivariate analysis.

CONCLUSIONS

Direct or combined revascularization for patients with adult-onset moyamoya disease presenting with ischemia can prevent further stroke.