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Wen-Yuh Chung, David Hung-Chi Pan, Cheng-Ying Shiau, Wan-Yuo Guo, and Ling-Wei Wang

Object. The goal of this study was to elucidate the role of gamma knife radiosurgery (GKS) and adjuvant stereotactic procedures by assessing the outcome of 31 consecutive patients harboring craniopharyngiomas treated between March 1993 and December 1999.

Methods. There were 31 consecutive patients with craniopharyngiomas: 18 were men and 13 were women. The mean age was 32 years (range 3–69 years). The mean tumor volume was 9 cm3 (range 0.3–28 cm3). The prescription dose to the tumor margin varied from 9.5 to 16 Gy. The visual pathways received 8 Gy or less. Three patients underwent stereotactic aspiration to decompress the cystic component before GKS. The tumor response was classified by percentage reduction of tumor volume as calculated based on magnetic resonance imaging studies. Clinical outcome was evaluated according to improvement and dependence on replacement therapy.

An initial postoperative volume increase with enlargement of a cystic component was found in three patients. They were treated by adjuvant stereotactic aspiration and/or Ommaya reservoir implantation. Tumor control was achieved in 87% of patients and 84% had fair to excellent clinical outcome in an average follow-up period of 36 months. Treatment failure due to uncontrolled tumor progression was seen in four patients at 26, 33, 49, and 55 months, respectively, after GKS. Only one patient was found to have a mildly restricted visual field; no additional endocrinological impairment or neurological deterioration could be attributed to the treatment. There was no treatment-related mortality.

Conclusions. Multimodality management of patients with craniopharyngiomas seemed to provide a better quality of patient survival and greater long-term tumor control. It is suggested that GKS accompanied by adjuvant stereotactic procedures should be used as an alternative in treating recurrent or residual craniopharyngiomas if further microsurgical excision cannot promise a cure.

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Lin-Feng Wang, Ying-Ze Zhang, Yong Shen, Yan-Ling Su, Jia-Xin Xu, Wen-Yuan Ding, and Ying-Hua Zhang

Object

The aim of this study was to investigate the clinical significance of both the signal intensity ratio obtained from MR imaging and clinical manifestations on the prognosis of patients with cervical ossification of the posterior longitudinal ligament.

Methods

The authors retrospectively reviewed the records of 58 patients with cervical ossification of the posterior longitudinal ligament who underwent cervical laminoplasty from February 1999 to July 2007. Magnetic resonance imaging (1.5-T) was performed in all patients before surgery. Sagittal T2-weighted images of the cervical spinal cord compressed by the ossified posterior longitudinal ligament showed increased intramedullary signal intensity, whereas the sagittal images obtained at the C7–T1 disc levels were of normal intensity. The signal intensity ratio between regions of intramedullary increased signal intensity and the normal C7–T1 disc level was calculated based on the signal intensity values generated from the MR imaging workstation. Patients were divided into 3 groups according to their signal intensity ratio (high, intermediate, and low signal intensity groups).

Results

There were significant differences between the 3 groups regarding recovery rate (p < 0.001), age (p = 0.022), duration of disease (p = 0.001), Babinski sign (p < 0.001), ankle clonus (p < 0.001), and both pre- and postoperative Japanese Orthopaedic Association score (p < 0.001). There was no significant difference in sex among the 3 groups (p = 0.391).

Conclusions

Patients with low signal intensity ratios that changed on T2-weighted imaging experienced a good surgical outcome. Low increased signal intensity might reflect mild neuropathological alteration in the spinal cord and greater recuperative potential. An increased signal intensity ratio with positive pyramidal signs indicates less recuperative potential of the spinal cord and a poor surgical outcome.

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Jian-tao Liang, Yu-hai Bao, Hong-qi Zhang, Li-rong Huo, Zhen-yu Wang, and Feng Ling

Object

The authors conducted a study to assess the clinical pattern, radiological features, therapeutic strategies, and long-term outcomes in patients with intramedullary spinal cord cavernomas (ISCCs) based on a large case series.

Methods

This retrospective study identified 96 patients (60 males, 36 females) surgically (81 cases) or conservatively (15 cases) treated for ISCCs between May 1993 and November 2007. Each diagnosis was based on MR imaging and spinal angiography evidence. For all surgically treated patients, the diagnosis was verified pathologically. The neurological outcomes pre- and postoperatively, as well as long-term follow-up, were assessed using the Aminoff-Logue Disability Scale.

Results

The mean age at the onset of symptoms was 34.5 years (range 9–80 years). Of the lesions, 68 (71%) were located in the thoracic spine, 25 (26%) in the cervical spine, and only 3 (3%) in the lumbar spine. The median symptom duration was 19.7 months. The clinical behavior of the lesion was a slow progression in 73 cases and an acute decline in 23 cases. Long-term follow-up data (mean 45.8 months, range 10–183 months) were available for 75 patients (64 surgical cases and 11 conservative cases). In the surgical group, a complete resection was achieved in 60 patients, and incomplete resection was detected in 4 patients after operation. At the end of the follow-up period in the operative group, 23 patients (36%) improved, 35 (55%) remained unchanged, and 6 (9%) worsened. In the nonoperative group, 5 patients improved, 6 patients remained unchanged, and none worsened.

Conclusions

For differential diagnosis, spinal angiography was necessary in some cases. For most symptomatic lesions, complete microsurgical resection of the symptomatic ISCC is safe and prevents rebleeding and further neurological deterioration. However, in patients whose lesions were small and located ventrally in the spinal cord, one can also opt for a rigorous follow-up, considering the high surgical risk.

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Alice M. K. Wong, Yu-Cheng Pei, Tai-Ngar Lui, Chia-Ling Chen, Chin-Man Wang, and Chia-Ying Chung

Object. Both botulinum toxin type A (BTA) injection and selective posterior rhizotomy (SPR) are well-recognized treatments for children with spastic cerebral palsy (CP); however, there has been no study in which the longterm effectiveness of these two approaches has been compared.

Methods. The study population comprised 62 ambulatory children with spastic diplegic CP who were participating in the same rehabilitation program and 19 healthy volunteers. The children with CP were divided into the following three groups: BTA (22 cases), SPR (20 cases), and no treatment (20 cases); the healthy volunteers served as the control group. A computer-assisted gait analysis system was used to assess gait performance. Gait was assessed in the three groups of children at 1 week before treatment, and 3, 6, 12, and 20 months after treatment.

Based on the analysis of walking velocity, cadence, and step length, the BTA group demonstrated rapid improvement posttreatment but the improvement became insignificant after 12 months even with repeated BTA injections at 4-month intervals. In contrast, the SPR group displayed initial deterioration of gait parameters during the first 3 months posttreatment and then improved continuously from 6 to 20 months. The control group did not display a significant change in gait.

Conclusions. The findings suggest that the effectiveness of BTA injection is more short-lived and SPR initially decreases gait performance but is expected to improve gait performance at between 6 and 20 months after the procedure.

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D. Hung-Chi Pan, Wan-Yuo Guo, Wen-Yuh Chung, Cheng-Ying Shiau, Yue-Cune Chang, and Ling-Wei Wang

Object. A consecutive series of 240 patients with arteriovenous malformations (AVMs) treated by gamma knife radiosurgery (GKS) between March 1993 and March 1999 was evaluated to assess the efficacy and safety of radiosurgery for cerebral AVMs larger than 10 cm3 in volume.

Methods. Seventy-six patients (32%) had AVM nidus volumes of more than 10 cm3. During radiosurgery, targeting and delineation of AVM nidi were based on integrated stereotactic magnetic resonance (MR) imaging and x-ray angiography. The radiation treatment was performed using multiple small isocenters to improve conformity of the treatment volume. The mean dose inside the nidus was kept between 20 Gy and 24 Gy. The margin dose ranged between 15 to 18 Gy placed at the 55 to 60% isodose centers. Follow up ranged from 12 to 73 months.

There was complete obliteration in 24 patients with an AVM volume of more than 10 cm3 and in 91 patients with an AVM volume of less than 10 cm3. The latency for complete obliteration in larger-volume AVMs was significantly longer. In Kaplan—Meier analysis, the complete obliteration rate in 40 months was 77% in AVMs with volumes between 10 to 15 cm3, as compared with 25% for AVMs with a volume of more than 15 cm3. In the latter, the obliteration rate had increased to 58% at 50 months. The follow-up MR images revealed that large-volume AVMs had higher incidences of postradiosurgical edema, petechiae, and hemorrhage. The bleeding rate before cure was 9.2% (seven of 76) for AVMs with a volume exceeding 10 cm3, and 1.8% (three of 164) for AVMs with a volume less than 10 cm3. Although focal edema was more frequently found in large AVMs, most of the cases were reversible. Permanent neurological complications were found in 3.9% (three of 76) of the patients with an AVM volume of more than 10 cm3, 3.8% (three of 80) of those with AVM volume of 3 to 10 cm3, and 2.4% (two of 84) of those with an AVM volume less than 3 cm3. These differences in complications rate were not significant.

Conclusions. Recent improvement of radiosurgery in conjunction with stereotactic MR targeting and multiplanar dose planning has permitted the treatment of larger AVMs. It is suggested that gamma knife radiosurgery is effective for treating AVMs as large as 30 cm3 in volume with an acceptable risk.

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Kang-Du Liu, Wen-Yuh Chung, Hsiu-Mei Wu, Cheng-Ying Shiau, Ling-Wei Wang, Wan-You Guo, and David Hung-Chi Pan

Object. The authors sought to determine the value of gamma knife surgery (GKS) in the treatment of cavernous hemangiomas (CHs).

Methods. Between 1993 and 2002, a total of 125 patients with symptomatic CHs were treated with GKS. Ninety-seven patients presented with bleeding and 45 of these had at least two bleeding episodes. Thirteen patients presented with seizures combined with hemorrhage, and 15 patients presented with seizures alone. The mean margin dose of radiation was 12.1 Gy and the mean follow-up time was 5.4 years.

In the 112 patients who had bled the number of rebleeds after GKS was 32. These rebleeds were defined both clinically and based on magnetic resonance imaging for an annual rebleeding rate of 32 episodes/492 patient-years or 6.5%. Twenty-three of the 32 rebleeding episodes occurred within 2 years after GKS. Nine episodes occurred after 2 years; thus, the annual rebleeding rate after GKS was 10.3% for the first 2 years and 3.3% thereafter (p = 0.0038). In the 45 patients with at least two bleeding episodes before GKS, the rebleeding rate dropped from 29.2% (55 episodes/188 patient-years) before treatment to 5% (10 episodes/197 patient-years) after treatment (p < 0.0001). Among the 28 patients who presented with seizures, 15 (53%) had good outcomes (Engel Grades I and II). In this study of 125 patients, symptomatic radiation-induced complications developed in only three patients.

Conclusions. Gamma knife surgery can effectively reduce the rebleeding rate after the first symptomatic hemorrhage in patients with CH. In addition, GKS may be useful in reducing the severity of seizures in patients with CH.

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David Hung-chi Pan, Wen-yuh Chung, Wan-yuo Guo, Hsiu-mei Wu, Kang-du Liu, Cheng-ying Shiau, and Ling-wei Wang

Object. The aim of this study was to assess the efficacy and safety of radiosurgery for the treatment of dural arteriovenous fistulas (DAVFs) located in the region of the transverse—sigmoid sinus.

Methods. A series of 20 patients with DAVFs located in the transverse—sigmoid sinus, who were treated with gamma knife surgery between June 1995 and June 2000, was evaluated. According to the Cognard classification, the DAVF was Type I in four patients, Type IIa in seven, Type IIb in two, and combined Type IIa+b in seven. Nine patients had previously been treated with surgery and/or embolization, whereas 11 patients underwent radiosurgery alone. Radiosurgery was performed using multiple-isocenter irradiation of the delineated DAVF nidus. The target volume ranged from 1.7 to 40.7 cm3. The margin dose delivered to the nidus ranged from 16.5 to 19 Gy at a 50 to 70% isodose level.

Nineteen patients were available for follow-up review, the duration of which ranged from 6 to 58 months (median 19 months). Of the 19 patients, 14 (74%) were cured of their symptoms. At follow up, magnetic resonance imaging and/or angiography demonstrated complete obliteration of the DAVF in 11 patients (58%), subtotal obliteration (95% reduction of the nidus) in three (16%), and partial obliteration in another five (26%). There was no neurological complication related to the treatment. One patient experienced a recurrence of the DAVF 18 months after angiographic confirmation of total obliteration, and underwent a second course of radiosurgery.

Conclusions. Stereotactic radiosurgery provides a safe and effective option for the treatment of DAVFs involving the transverse and sigmoid sinuses. For some aggressive DAVFs with extensive retrograde cortical venous drainage, however, a combination of endovascular embolization and surgery may be necessary.

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Xin Wang, Zhiqi Mao, Zhiqiang Cui, Xin Xu, Longsheng Pan, Shuli Liang, Zhipei Ling, and Xinguang Yu

OBJECTIVE

Primary Meige syndrome is characterized by blepharospasm and orofacial–cervical dystonia. Deep brain stimulation (DBS) is recognized as an effective therapy for patients with this condition, but previous studies have focused on clinical effects. This study explored the predictors of clinical outcome in patients with Meige syndrome who underwent DBS.

METHODS

Twenty patients who underwent DBS targeting the bilateral subthalamic nucleus (STN) or globus pallidus internus (GPi) at the Chinese People’s Liberation Army General Hospital from August 2013 to February 2018 were enrolled in the study. Their clinical outcomes were evaluated using the Burke–Fahn–Marsden Dystonia Rating Scale at baseline and at the follow-up visits; patients were accordingly divided into a good-outcome group and a poor-outcome group. Putative influential factors, such as age and course of disease, were examined separately, and the factors that reached statistical significance were subjected to logistic regression analysis to identify predictors of clinical outcomes.

RESULTS

Four factors showed significant differences between the good- and poor-outcome groups: 1) the DBS target (STN vs GPi); 2) whether symptoms first appeared at multiple sites or at a single site; 3) the sub-item scores of the mouth at baseline; and 4) the follow-up period (p < 0.05). Binary logistic regression analysis revealed that initial involvement of multiple sites and the mouth score were the only significant predictors of clinical outcome.

CONCLUSIONS

The severity of the disease in the initial stage and presurgical period was the only independent predictive factor of the clinical outcomes of DBS for the treatment of patients with Meige syndrome.

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Wen-Yuh Chung, Cheng-Ying Shiau, Hsiu-Mei Wu, Kang-Du Liu, Wan-Yuo Guo, Ling-Wei Wang, and David Hung-Chi Pan

Object

The effectiveness and safety of radiosurgery for small- to medium-sized cerebral arteriovenous malformations (AVMs) have been well established. However, the management for large cerebral AVMs remains a great challenge to neurosurgeons. In the past 5 years the authors performed preplanned staged radiosurgery to treat extra-large cerebral AVMs.

Methods

An extra-large cerebral AVM is defined as one with nidus volume > 40 ml. The nidus volume of cerebral AVM is measured from the dose plan—that is, as being the volume contained within the best-fit prescription isodose. From January 2003 to December 2007, the authors treated 6 patients with extra-large AVMs by preplanned staged GKS. Staged radiosurgery is implemented by rigid transformation with translation and rotation of coordinates between 2 stages. The average radiation-targeted volume was 60 ml (range 47–72 ml). The presenting symptoms were seizure in 4 patients and a bleeding episode in 2. One patient had undergone a previous craniotomy and evacuation of hematoma. The mean interval between the 2 radiosurgical sessions was 6.9 months (range 4.5–9.1 months). The prescribed marginal dose given to the nidus volume in each stage ranged from 16 to 18.6 Gy. The expected marginal dose of total nidus was 17–19 Gy. Regular follow-up MR imaging was performed every 6 months. The mean follow-up period was 28 months (range 12–54 months).

Results

Most of the patients exhibited clinical improvement: relief of headache and reduced frequency of seizure attack. All patients had significant regression of nidus observed on MR imaging follow-up. Two patients had angiogram-confirmed complete obliteration of the nidus 45 and 60 months after the second-stage radiosurgical session. One patient experienced minor bleeding 8 months after the second-stage radiosurgery with mild headache. She had satisfactory recovery without clinical neurological deficit after conservative treatment.

Conclusions

These preliminary results indicate that staged radiosurgery is a practical strategy to treat patients with extra-large cerebral AVMs. It takes longer to obliterate the AVMs. The observed high signal T2 changes after the radiosurgery appeared clinically insignificant in 6 patients followed up for an average of 28 months. Longer follow-up is necessary to confirm its long-term safety.

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Weijun Wang, Nian-Ling Zhu, Jason Chua, Steve Swenson, Fritz K. Costa, Stephanie Schmitmeier, Barbara A. Sosnowski, Toshiaki Shichinohe, Noriyuki Kasahara, and Thomas C. Chen

Object. Adenovirus vector (AdV)—mediated gene delivery has been recently demonstrated in clinical trials as a novel potential treatment for malignant gliomas. Combined coxsackievirus B and adenovirus receptor (CAR) has been shown to function as an attachment receptor for multiple adenovirus serotypes, whereas the vitronectin integrins (αvβ3 and αvβ5) are involved in AdV internalization. In resected glioma specimens, the authors demonstrated that malignant gliomas have varying levels of CAR, αvβ3, and αvβ5 expression.

Methods. A correlation between CAR expression and the transduction efficiency of AdV carrying the green fluorescent protein in various human glioblastoma multiforme (GBM) cell lines and GBM primary cell lines was observed. To increase transgene activity in in vitro glioma cells with low or deficient levels of CAR, the authors used basic fibroblast growth factor (FGF2) as a targeting ligand to redirect adenoviral infection through its cognate receptor, FGF receptor 1 (FGFR1), which was expressed at high levels by all glioma cells. These findings were confirmed by in vivo study data demonstrating enhanced transduction efficiency of FGF2-retargeted AdV in CAR-negative intracranial gliomas compared with AdV alone, without evidence of increased angiogenesis.

Conclusions. Altogether, the results demonstrated that AdV-mediated gene transfer using the FGF2/FGFR system is effective in gliomas with low or deficient levels of CAR and suggested that FGF2-retargeting of AdV may be a promising approach in glioma gene therapy.