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Sebastian Siller, Rami Kasem, Thomas-Nikolaus Witt, Joerg-Christian Tonn, and Stefan Zausinger

OBJECTIVE

Various neurological diseases are known to cause progressive painless paresis of the upper limbs. In this study the authors describe the previously unspecified syndrome of compression-induced painless cervical radiculopathy with predominant motor deficit and muscular atrophy, and highlight the clinical and radiological characteristics and outcomes after surgery for this rare syndrome, along with its neurological differential diagnoses.

METHODS

Medical records of 788 patients undergoing surgical decompression due to degenerative cervical spine diseases between 2005 and 2014 were assessed. Among those patients, 31 (3.9%, male to female ratio 4.8 to 1, mean age 60 years) presented with painless compressive cervical motor radiculopathy due to neuroforaminal stenosis without signs of myelopathy; long-term evaluation was available in 23 patients with 49 symptomatic foraminal stenoses. Clinical, imaging, and operative findings as well as the long-term course of paresis and quality of life were analyzed.

RESULTS

Presenting symptoms (mean duration 13.3 months) included a defining progressive flaccid radicular paresis (median grade 3/5) without any history of radiating pain (100%) and a concomitant muscular atrophy (78%); 83% of the patients were smokers and 17% patients had diabetes. Imaging revealed a predominantly anterior nerve root compression at the neuroforaminal entrance in 98% of stenoses. Thirty stenoses (11 patients) were initially decompressed via an anterior surgical approach and 19 stenoses (12 patients) via a posterior surgical approach. Overall reoperation rate due to new or recurrent stenoses was 22%, with time to reoperation shorter in smokers (p = 0.033). Independently of the surgical procedure chosen, long-term follow-up (mean 3.9 years) revealed a stable or improved paresis in 87% of the patients (median grade 4/5) and an excellent general performance and quality of life.

CONCLUSIONS

Painless cervical motor radiculopathy predominantly occurs due to focal compression of the anterior nerve root at the neuroforaminal entrance. Surgical decompression is effective in stabilizing or improving motor function with a resulting favorable long-term outcome.

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Thomas C. Vukovich, Adelheid Gabriel, Bruno Schaefer, Mario Veitl, Christian Matula, and Christian K. Spiss

✓ Patients undergoing brain tumor surgery are at high risk for the occurrence of a thromboembolic event. To identify a laboratory marker suitable for risk estimation the authors studied the perioperative time pattern of routine coagulation parameters and the specific hemostasis activation marker D-dimer in 28 consecutive patients at high risk (11 patients with glioma and eight patients with meningioma) and low risk (nine patients with metastases) for thromboembolism, as previously reported. As is typical during major surgery, most of the routine parameters declined, probably because of hemodilution, and recovered postoperatively to values higher than baseline, probably because of an acute-phase reaction. On Days 2 and 7 after surgery no difference in the routine parameters was recorded between patients at high (meningioma and glioma) and low risk (metastasis). The level of D-dimer was elevated at baseline in patients with metastasis, indicating a hemostatic hyperactivity that is usual in cancer patients. During surgery a marked increase in D-dimer levels occurred in patients with meningioma and glioma (pre- and postoperative median 90/2000 and 100/1020 ng/ml, respectively), but the increase was less pronounced in patients with metastasis (320/660 ng/ml). Postoperatively, D-dimer declined in patients with metastases to lower than preoperative levels (Day 7, 270 ng/ml); in patients with meningioma or glioma, however, D-dimer levels remained elevated until Day 7 (450 and 200 ng/ml, respectively). These results indicate that levels of Ddimer correlate with the reported high risk for thromboembolism in patients with meningioma and glioma, and D-dimer should be evaluated for its use in estimating individual risk and the efficiency of its use in the control of prophylactic treatment.

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Thomas Chr. Vukovich, Adelheid Gabriel, Bruno Schaefer, Mario Veitl, Christian Matula, and Christian K. Spiss

Patients undergoing brain tumor surgery are at high risk for the occurrence of a thromboembolic event. To identify a laboratory marker suitable for risk estimation the authors studied the perioperative time pattern of routine coagulation parameters and the specific hemostasis activation marker D-dimer in 28 consecutive patients at high risk (11 patients with glioma and eight patients with menigioma) and low risk (9 patients with metastases) for thromboembolism, as previously reported. As is typical during major surgery, most of the routine parameters declined, probably because of hemodilution, and recovered postoperatively to values higher than baseline, probably because of an acute-phase reaction. On Days 2 and 7 after surgery no difference in the routine parameters was recorded between patients at high (meningioma and glioma) and low risk (metastases). The level of D-dimer was elevated at baseline in patients with metastases, indicating a hemostatic hyperactivity that is usual in cancer patients. During surgery a marked increase in D-dimer levels occurred in patients with meningioma and glioma (pre- and postoperative median 90/2000 and 100/1020 ng/ml, respectively), but the increase was less pronounced in patients with metastases (320/660 ng/ml). Postoperatively, D-dimer declined in patients with metastases to lower levels than preoperatively (Day 7, 270 ng/ml); in patients with meningioma or glioma, however, D-dimer levels remained elevated until Day 7 (450 and 200 ng/ml). These results indicate that levels of D-dimer correlate with the reported high risk for thromboembolism in patients with meningioma and glioma, and D-dimer should be evaluated for its use in estimating individual risk and the efficiency of its use in the control of prophylactic treatment.

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Christian N. Ramsey III, Ben L. Proctor, Robert S. Baker, and Thomas Pittman

✓Ophthalmological problems such as papilledema, loss of visual acuity, and oculomotor palsies are common in patients with shunt malfunctions. The authors report on three patients with shunts who presented with visual loss. In each case, the diagnosis of shunt malfunction was delayed because other classic signs and symptoms of intracranial hypertension were absent. All three patients underwent optic nerve sheath fenestration (ONSF) with resultant stabilization or improvement of visual symptoms before the shunt malfunction was recognized and treated. Consideration of these cases suggests a potential protective role of ONSF for patients with shunt malfunction.

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Christian Dorfer, Gregor Kasprian, Angelika Mühlebner, and Thomas Czech

Hypothalamic hamartomas are rare lesions for which different classification schemes have been proposed. The authors report on an exceptionally large solid-cystic hamartoma that led to hydrocephalus, precocious puberty, and intractable gelastic seizures. They discuss potential mechanisms of the development of hypothalamic hamartomas.

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Christian Schichor, Walter Rachinger, Dominik Morhard, Stefan Zausinger, Thomas J. Heigl, Maximilian Reiser, and Jörg-Christian Tonn

Object

In vascular neurosurgery, there is a demand for intraoperative imaging of blood vessels as well as for rapid information about critical impairment of brain perfusion. This study was conducted to analyze the feasibility of intraoperative CT angiography and brain perfusion mapping using an up-to-date multislice CT scanner in a prospective pilot series.

Methods

Ten patients with unruptured aneurysms underwent intraoperative scanning with a 40-slice sliding-gantry CT scanner. Multimodal CT acquisition was obtained in 8 patients consisting of dynamic perfusion CT (PCT) scanning followed by intracranial CT angiography. Two of these patients underwent CT angiography and PCT 2 times in 1 session as a control after repositioning cerebral aneurysm clips. In another 2 patients, CT angiography was performed alone. The quality of all imaging obtained was assessed in a blinded consensus reading performed by an experienced neurosurgeon and an experienced neuroradiologist. A 6-point scoring system ranging from excellent to insufficient was used for quality evaluation of PCT and CT angiography.

Results

In 9 of 10 PCT data sets, the quality was rated excellent or good. In the remaining case, the quality was rated insufficient for diagnostic evaluation due to major streak artifacts induced by the titanium pins of the head clamp. In this particular case, the quality of the related CT angiography was rated good and sufficient for intraoperative decision making. The quality of all 12 CT angiography data sets was rated excellent or good. In 1 patient with an anterior communicating artery aneurysm, PCT scanning led to a repositioning of the clip because of an ischemic pattern of the perfusion parameter maps due to clip stenosis of an artery. The subsequent PCT scan obtained in this patient revealed an improved perfusion of the related vascular territory, and follow-up MR imaging showed only minor ischemia of the anterior cerebral artery territory.

Conclusions

Intraoperative CT angiography and PCT scanning were shown to be feasible with short acquisition time, little interference with the surgical workflow, and very good diagnostic imaging quality. Thus, these modalities might be very helpful in vascular neurosurgery. Having demonstrated their feasibility, the impact of these methods on patients' outcomes has now to be analyzed prospectively in a larger series.

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Ricardo Santos de Oliveira, Giuseppe Cinalli, Thomas Roujeau, Christian Sainte-Rose, Alain Pierre-Kahn, and Michel Zerah

Object

The authors of this retrospective review and analysis of the literature cover an institutional series of neurenteric cysts of the central nervous system in children treated in the magnetic resonance imaging era during a 14-year period.

Methods

Sixteen patients 20 days to 14 years of age are described. The most frequent signs and symptoms at presentation were acute spinal cord compression (11 patients), paresis of a cranial nerve (two patients), meningitis or infection (two patients), and intracranial hypertension (one patient). The locations of the cysts were as follows: in the spinal canal in 12 patients (75%), the clivus in two (12.5%), the cavernous sinus in one (6%), and the craniocervical junction in one (6%). The most common location was the ventral aspect of the spinal canal (seven patients). Associated spinal deformities were found in five patients. All patients underwent surgery, with a posterior approach used in all of the spinal cases. Total resection was achieved in 12 of the 16 cases and partial resection in four. Of the four patients who underwent subtotal resection, the cyst recurred in three, requiring further surgery.

Conclusions

Neurenteric cysts are uncommon congenital anomalies that can present acutely in the pediatric population. Total removal is usually possible and is associated with a good prognosis.

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Vinay Gupta, Christian G. Samuleson, Susan Su, and Thomas C. Chen

✓ Although most meningiomas are treated surgically, it may not be possible to completely remove atypical, malignant, and surgically inaccessible meningiomas; in the majority of these cases there is tumor recurrence. The authors have already reported initial preclinical results on the efficacy of imatinib in the treatment of meningiomas; however, a recent Phase II trial of imatinib in patients with recurrent meningiomas did not demonstrate significant antitumor activity. To enhance the activity of imatinib, the authors investigated the use of a combination therapy with nelfinavir on primary meningioma cells and meningioma cell lines IOMM-Lee and CH157.

Cytotoxicity was measured using methylthiotetrazole and colony formation assays. In low-dose combination therapy with imatinib, nelfinavir potentiated the antiproliferative and anti–colony formation effects of imatinib. Primary meningioma cells responded better to combination therapy than to imatinib alone. Treatment induced a dose-dependent antiproliferative effect, decreased cell survival, and inhibited colony formation. Western blotting demonstrated decreased levels of survivin protein on combination therapy. Because meningiomas have very high levels of survivin protein, survivin inhibition by nelfinavir may represent a potential mechanism for the additive effect observed with imatinib. Moreover, an increase in the proapoptotic Bax/Bcl-2 protein ratio was demonstrated with the combination of imatinib and nelfinavir.

The authors propose that nelfinavir not only potentiates imatinib efficacy, it also abrogates resistance to imatinib by decreasing survivin protein levels in meningiomas. In an in vivo assay, this combination therapy was found to be more effective than imatinib alone. More preclinical work with in vivo models is needed to determine if this new combination therapy will translate into a viable future therapy for meningiomas.

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Christian Schneider, Eveline Teresa Hidalgo, Thomas Schmitt-Mechelke, and Karl F. Kothbauer

Object

Presently, the best available treatment for intramedullary spinal cord tumors (IMSCTs) in children is microsurgery with the objective of maximal tumor removal and minimal neurological morbidity. The latter has become manageable with the development and standard use of intraoperative neurophysiological monitoring. Traditionally, the perioperative neurological evaluation is based on surgical or spinal cord injury scores focusing on sensorimotor function. Little is known about the quality of life after such operations; therefore, this study was designed to investigate the impact of surgery for IMSCTs on the quality of life in children.

Methods

Twelve consecutive pediatric patients treated for IMSCT were included in this retrospective fixed cohort study. A multidimensional questionnaire-based quality of life instrument, the Pediatric Quality of Life Questionnaire version 4 (PedsQL 4.0), was chosen to analyze follow-up data. This validated instrument particularly allows for a comparison between a patient cohort and a healthy pediatric sample population.

Results

Of 11 mailed questionnaires (1 patient had died of progressive disease), 10 were returned, resulting in a response rate of 91%. There were 8 low-grade lesions (5 pilocytic astrocytomas, 1 ganglioglioma, 1 hemangioblastoma, and 1 cavernoma) and 4 high-grade lesions (2 anaplastic gangliogliomas, 1 glioblastoma, and 1 glioneuronal tumor). The mean age at diagnosis was 7.5 years, the mean follow-up was 4.2 years, and 83% of the patients were male. Total resection was achieved in 5 patients and subtotal resection in 7. Four patients had undergone 2 or more resections. The 4 patients with high-grade tumors and 2 with incompletely resected low-grade tumors underwent adjuvant treatment (2 chemotherapy and 4 both radiotherapy and chemotherapy). The mean modified McCormick Scale score at the time of diagnosis was 1.7; at the time of follow-up, 1.5. The mean PedsQL 4.0 total score in the low-grade group was 78.5; in the high-grade group, 82.6. There was no significant difference in PedsQL 4.0 scores between the patient cohort and the normal population.

Conclusions

In a small cohort of children who had undergone surgery for IMSCTs with a mean follow-up of 4.2 years, quality of life scores according to the PedsQL 4.0 instrument were not different from those in a normal sample population.