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Intracranial pial arteriovenous fistulas with single-vein drainage

Report of three cases and review of the literature

Yi-Chou Wang, Ho-Fai Wong and Yi-Shian Yeh

✓ Intracranial pial arteriovenous fistulas (AVFs) are rare vascular lesions, of which fewer than 90 cases have been reported in the literature. They are composed of one or more arterial feeding vessels and a single draining vein that usually result in a huge varix. Because of the high-flow shunting, a poor prognosis is associated with conservative treatment. Surgical or endovascular treatment also poses various challenges. The authors present the cases of three patients in whom n-butyl-2-cyanoacrylate-assisted embolization was performed. Outcome in all three cases was good. The necessity of staged procedures to obliterate the AVF is also discussed.

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Yi Ma, Yan-feng Li, Quan-cai Wang, Bin Wang and Hai-tao Huang

OBJECT

The object of this study was to investigate the immediate and long-term follow-up results of glossopharyngeal nerve rhizotomy (GPNR) with or without partial vagus nerve rhizotomy (VNR) for treating glossopharyngeal neuralgia (GPN).

METHODS

A retrospective review of the case notes of patients who had undergone surgery for GPN in the authors’ department between 2008 and 2013 was performed to investigate baseline characteristics and immediate outcomes during the hospitalization. For the long-term results, a telephone survey was performed, and information on pain recurrence and permanent complications was collected. Pain relief meant no pain or medication, any pain persisting after surgery was considered to be treatment failure, and any pain returning during the follow-up period was considered to be pain recurrence. For comparative study, the patients were divided into 2 cohorts, that is, patients treated with GPNR alone and those treated with GPNR+VNR.

RESULTS

One hundred three procedures, consisting of GPNR alone in 38 cases and GPNR+VNR in 65 cases, were performed in 103 consecutive patients with GPN. Seventy-nine of the 103 patients could be contacted for the follow-up study, with a mean follow-up duration of 2.73 years (range 1 month–5.75 years). While there were similar results (GPNR vs GPNR+VNR) in immediate pain relief rates (94.7% vs 93.8%), immediate complication rates (7.9% vs 4.6%), and long-term pain relief rates (92.3% vs 94.3%) between the 2 cohorts, a great difference was seen in long-term complications (3.8% vs 35.8%). The long-term complication rate for the combined GPNR+VNR cohort was 9.4 times higher than that in the GPNR cohort.

There was no operative or perioperative mortality. Immediate complications occurred in 6 cases, consisting of poor wound healing in 3 cases, and CSF leakage, hoarseness, and dystaxia in 1 case each. Permanent complications occurred in 20 patients (25.3%) and included cough while drinking in 10 patients, pharyngeal discomfort in 8 patients, and hoarseness and dysphagia in 1 case each.

CONCLUSIONS

In general, this study indicates that GPNR alone or in combination with VNR is a safe, simple, and effective treatment option for GPN. It may be especially valuable for patients who are not suitable for the microvascular decompression (MVD) procedure and for surgeons who have little experience with MVD. Of note, this study renews the significance of GPNR alone, which, the authors believe, is at least valuable for a subgroup of GPN patients, with significantly fewer long-term complications than those for rhizotomy for both glossopharyngeal nerve and rootlets of the vagus nerve.

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Yi Wang, Yong Wang, Yida Wang, Nobuyuki Taniguchi and Xian-Cheng Chen

Object

The goal of this study was to combine the use of ultrasound contrast agents with intraoperative ultrasound techniques to identify intraoperatively a patient's vascular anatomy, including feeding arteries and draining veins of an intracranial arteriovenous malformation (AVM).

Methods

The authors examined 12 consecutive patients with AVMs that had been diagnosed on the basis of preoperative findings on magnetic resonance images and digital subtraction angiograms obtained between September 2003 and December 2005. After each patient had undergone a routine craniotomy, a bolus of contrast agent was injected intravenously, and a real-time microbubble perfusion process was observed to identify the feeding arteries and draining veins of the AVM in a single cross-section. The so-called burst–refill technique was used to sweep the lesion in multiple sections and orientations to obtain information on the surrounding vascular anatomy, after which the findings were compared with those obtained during preoperative imaging.

Results

Intraoperative ultrasonography provided high-quality images in every case. Although plain imaging failed to show an identifiable AVM boundary, color Doppler flow imaging clearly delineated the shape and margin of the AVM. Nevertheless, neither mode of imaging enabled the surgeons to categorically distinguish between feeding and draining vessels.

The real-time perfusion process of microbubbles was first visualized 20 to 30 seconds after the SonoVue bolus injection, and the burst–refill technique made possible identification of the vascular anatomy of malformation lesions in multiple planes.

Conclusions

Using both an ultrasound contrast agent and the burst–refill technique provided a rapid, convenient, and precise way of locating AVM feeding arteries intraoperatively. The combined technique seems warranted in the intraoperative treatment of AVMs.

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Alexey V. Dimov, Ajay Gupta, Brian H. Kopell and Yi Wang

OBJECTIVE

Faithful depiction of the subthalamic nucleus (STN) is critical for planning deep brain stimulation (DBS) surgery in patients with Parkinson’s disease (PD). Quantitative susceptibility mapping (QSM) has been shown to be superior to traditional T2-weighted spin echo imaging (T2w). The aim of the study was to describe submillimeter QSM for preoperative imaging of the STN in planning of DBS.

METHODS

Seven healthy volunteers were included in this study. T2w and QSM were obtained for all healthy volunteers, and images of different resolutions were reconstructed. Image quality and visibility of STN anatomical features were analyzed by a radiologist using a 5-point scale, and contrast properties of the STN and surrounding tissue were calculated. Additionally, data from 10 retrospectively and randomly selected PD patients who underwent 3-T MRI for DBS were analyzed for STN size and susceptibility gradient measurements.

RESULTS

Higher contrast-to-noise ratio (CNR) values were observed in both high-resolution and low-resolution QSM images. Inter-resolution comparison demonstrated improvement in CNR for QSM, but not for T2w images. QSM provided higher inter-quadrant contrast ratios (CR) within the STN, and depicted a gradient in the distribution of susceptibility sources not visible in T2w images.

CONCLUSIONS

For 3-T MRI, submillimeter QSM provides accurate delineation of the functional and anatomical STN features for DBS targeting.

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Dunyue Lu, Yi Li, Asim Mahmood, Lei Wang, Tahir Rafiq and Michael Chopp

Object. This study was designed to investigate the effect of treatment with a novel composite material consisting of embryonic neurospheres and bone marrow—derived stromal cell spheres (NMSCSs) in a rat model of traumatic brain injury (TBI).

Methods. The NMSCS composite was injected into the TBI contusion site 24 hours after injury, and all rats were killed on Day 14 after the transplantation. The Rotarod test and the neurological severity score were used to evaluate neurological function. The transplanted NMSCS was analyzed in recipient rat brains by using histological staining and laser scanning confocal microscopy. The lesion volumes in the brains were also calculated using computer image analysis.

Conclusions. Rats that received NMSCS transplants had reduced lesion volume and showed improved motor and neurological function when compared with control groups 14 days after the treatment. These results suggest that transplantation of this novel biological material (NMSCS) may be useful in the treatment of TBI.

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Xiaochun Jiang, Yukui Yan, Minghua Hu, Xiande Chen, Yaxian Wang, Yi Dai, Degang Wu, Yongsheng Wang, Zhixiang Zhuang and Hongping Xia

OBJECT

Increased levels of H19 long noncoding RNA (lncRNA) have been observed in many cancers, suggesting that overexpression of H19 may be important in the development of carcinogenesis. However, the role of H19 in human glioblastoma is still unclear. The object of this study was to examine the level of H19 in glioblastoma samples and investigate the role of H19 in glioblastoma carcinogenesis.

METHODS

Glioblastoma and nontumor brain tissue specimens were obtained from tissue obtained during tumor resection in 30 patients with glioblastoma. The level of H19 lncRNA was detected by real-time quantitative reverse transcription polymerase chain reaction. The role of H19 in invasion, angiogenesis, and stemness of glioblastoma cells was then investigated using commercially produced cell lines (U87 and U373). The effects of H19 overexpression on glioblastoma cell invasion and angiogenesis were detected by in vitro Matrigel invasion and endothelial tube formation assay. The effects of H19 on glioblastoma cell stemness and tumorigenicity were investigated by neurosphere formation and an in vivo murine xenograft model.

RESULTS

The authors found that H19 is significantly overexpressed in glioblastoma tissues, and the level of expression was associated with patient survival. In the subsequent investigations, the authors found that overexpression of H19 promotes glioblastoma cell invasion and angiogenesis in vitro. Interestingly, H19 was also significantly overexpressed in CD133+ glioblastoma cells, and overexpression of H19 was associated with increased neurosphere formation of glioblastoma cells. Finally, stable overexpression of H19 was associated with increased tumor growth in the murine xenograft model.

CONCLUSIONS

The results of this study suggest that increased expression of H19 lncRNA promotes invasion, angiogenesis, stemness, and tumorigenicity of glioblastoma cells. Taken together, these findings indicate that H19 plays an important role in tumorigenicity and stemness of glioblastoma and thus could be a therapeutic target for treatment of glioblastoma in the future.

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Hock-Kean Liew, Chih-Wei Hsu, Mei-Jen Wang, Jon-Son Kuo, Ting-Yi Li, Hsiao-Fen Peng, Jia-Yi Wang and Cheng-Yoong Pang

Object

Intracerebral hemorrhage (ICH) accounts for about 15% of all deaths due to stroke. It frequently causes brain edema, leading to an expansion of brain volume that exerts a negative impact on ICH outcomes. The ICH-induced brain edema involves inflammatory mechanisms. The authors' in vitro study shows that urocortin (UCN) exhibits antiinflammatory and neuroprotective effects. Therefore, the neuroprotective effect of UCN on ICH in rats was investigated.

Methods

Intracerebral hemorrhage was induced by an infusion of bacteria collagenase type VII-S or autologous blood into the unilateral striatum of anesthetized rats. At 1 hour after the induction of ICH, UCN (0.05, 0.5, and 5 μg) was infused into the lateral ventricle on the ipsilateral side. The authors examined the injury area, brain water content, blood-brain barrier permeability, and neurological function.

Results

The UCN, administered in the ipsilateral lateral ventricle, was able to penetrate into the injured striatum. Posttreatment with UCN reduced the injury area, brain edema, and blood-brain barrier permeability and improved neurological deficits of rats with ICH.

Conclusions

Posttreatment with UCN through improving neurological deficits of rats with ICH dose dependently provided a potential therapeutic agent for patients with ICH or other brain injuries.

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Wei Shi, Shan Wang, Huifang Zhang, Guoqin Wang, Yi Guo, Zhenxing Sun, Youtu Wu, Peihai Zhang, Linkai Jing, Benqi Zhao, MM, Jian Xing, James Wang and Guihuai Wang

OBJECTIVE

Laminoplasty has been used in recent years as an alternative approach to laminectomy for preventing spinal deformity after resection of intramedullary spinal cord tumors (IMSCTs). However, controversies exist with regard to its real role in maintaining postoperative spinal alignment. The purpose of this study was to examine the incidence of progressive spinal deformity in patients who underwent laminoplasty for resection of IMSCT and identify risk factors for progressive spinal deformity.

METHODS

Data from IMSCT patients who had undergone laminoplasty at Beijing Tsinghua Changgung Hospital between January 2014 and December 2016 were retrospectively reviewed. Univariate tests and multivariate logistic regression analysis were used to assess the statistical relationship between postoperative spinal deformity and radiographic, clinical, and surgical variables.

RESULTS

One hundred five patients (mean age 37.0 ± 14.5 years) met the criteria for inclusion in the study. Gross-total resection (> 95%) was obtained in 79 cases (75.2%). Twenty-seven (25.7%) of the 105 patients were found to have spinal deformity preoperatively, and 10 (9.5%) new cases of postoperative progressive deformity were detected. The mean duration of follow-up was 27.6 months (SD 14.5 months, median 26.3 months, range 6.2–40.7 months). At last follow-up, the median functional scores of the patients who did develop progressive spinal deformity were worse than those of the patients who did not (modified McCormick Scale: 3 vs 2, and p = 0.04). In the univariate analysis, age (p = 0.01), preoperative spinal deformity (p < 0.01), extent of tumor involvement (p < 0.01), extent of abnormal tumor signal (p = 0.02), and extent of laminoplasty (p < 0.01) were identified as factors associated with postoperative progressive spinal deformity. However, in subsequent multivariate logistic regression analysis, only age ≤ 25 years and preoperative spinal deformity emerged as independent risk factors (p < 0.05), increasing the odds of postoperative progressive deformity by 4.1- and 12.4-fold, respectively (p < 0.05).

CONCLUSIONS

Progressive spinal deformity was identified in 25.7% patients who had undergone laminoplasty for IMSCT resection and was related to decreased functional status. Younger age (≤ 25 years) and preoperative spinal deformity increased the risk of postoperative progressive spinal deformity. The risk of postoperative deformity warrants serious reconsideration of providing concurrent fusion during IMSCT resection or close follow-up after laminoplasty.

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Po-Chuan Hsieh, Yi-Ming Wu, Alvin Yi-Chou Wang, Ching-Chang Chen, Chien-Hung Chang, Shy-Chyi Chin, Tai-Wei Erich Wu, Chieh-Tsai Wu and Shih-Tseng Lee

OBJECTIVE

Diverse treatment results are observed in patients with poor-grade aneurysmal subarachnoid hemorrhage (aSAH). Significant initial perfusion compromise is thought to predict a worse treatment outcome, but this has scant support in the literature. In this cohort study, the authors correlate the treatment outcomes with a novel poor-outcome imaging predictor representing impaired cerebral perfusion on initial CT angiography (CTA).

METHODS

The authors reviewed the treatment results of 148 patients with poor-grade aSAH treated at a single tertiary referral center between 2007 and 2016. Patients with the “venous delay” phenomenon on initial CTA were identified. The outcome assessments used the modified Rankin Scale (mRS) at the 3rd month after aSAH. Factors that may have had an impact on outcome were retrospectively analyzed.

RESULTS

Compared with previously identified outcome predictors, the venous delay phenomenon on initial CTA was found to have the strongest correlation with posttreatment outcomes on both univariable (p < 0.0001) and multivariable analysis (OR 4.480, 95% CI 1.565–12.826; p = 0.0052). Older age and a higher Hunt and Hess grade at presentation were other factors that were associated with poor outcome, defined as an mRS score of 3 to 6.

CONCLUSIONS

The venous delay phenomenon on initial CTA can serve as an imaging predictor for worse functional outcome and may aid in decision making when treating patients with poor-grade aSAH.

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Fon-Yih Tsuang, Abel Po-Hao Huang, Yi-Hsin Tsai, Jo-Yu Chen, Jing-Er Lee, Yong-Kwang Tu and Kuo-Chuan Wang

Object

Traumatic subdural effusion (TSE) is a common sequela of traumatic brain injury. Surgical intervention is suggested only when TSE exerts mass effect. The authors have found that many patients with TSE exerting mass effect have concomitant hydrocephalus. Patient experiencing this occurrence were studied, and the pathogenesis of this phenomenon was discussed in the context of recent advances in the understanding of CSF circulation.

Methods

During a 2-year period, the authors' institution treated 14 patients with TSE who developed hydrocephalus, after 1 of the patients suffered subdural drainage and other 13 received subdural peritoneal shunt (SPSs). Thirteen of those who had SPSs received programmable ventriculoperitoneal shunts (VPSs) for the hydrocephalus. The clinical characteristics as well as the imaging and operative findings of these patients were reviewed.

Results

All patients with symptomatic TSE exerting mass effect received SPSs. All of these patients had a modified Frontal Horn Index of more than 0.33 at presentation, and high opening pressure on durotomy. Following a brief period (4–7 days) of clinical improvement, the condition of all patients deteriorated due to hydrocephalus. Programmable VPSs were inserted with the initial pressure set at approximately 8–10 cm H2O according to opening pressure at ventriculostomy. Shunt valve pressure was gradually decreased to 5–7 cm H2O, according to clinical and radiological follow-up.

Conclusions

Elevated modified Frontal Horn Index in patients with TSE is suggestive of concomitant hydrocephalus. The authors propose that tearing of the dura-arachnoid plane following trauma contributes to TSE and may also impede CSF circulation, causing hydrocephalus. Shunt pressure was adjusted to relative low pressure, indicating the old age of the patients and poor reexpansion of brain parenchyma after the mass effect. Subdural peritoneal shunts and VPSs are indicated in those patients with TSE exerting mass effect with concomitant hydrocephalus.