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Korgun Koral

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Giannina L. Garcés-Ambrossi, Matthew J. McGirt, Vivek A. Mehta, Daniel M. Sciubba, Timothy F. Witham, Ali Bydon, Jean-Paul Wolinksy, George I. Jallo and Ziya L. Gokaslan

Object

With the introduction of electrophysiological spinal cord monitoring, surgeons have been able to perform radical resection of intramedullary spinal cord tumors (IMSCTs). However, factors associated with tumor resectability, tumor recurrence, and long-term neurological outcome are poorly understood.

Methods

The authors retrospectively reviewed 101 consecutive cases of IMSCT resection in adults and children at a single institution. Neurological function and MR images were evaluated preoperatively, at discharge, 1 month after surgery, and every 6 months thereafter. Factors associated with gross-total resection (GTR), progression-free survival (PFS), and long-term neurological improvement were assessed using multivariate regression analysis.

Results

The mean age of the patients was 41 ± 18 years and 17 (17%) of the patients were pediatric. Pathological type included ependymoma in 51 cases, hemangioblastoma in 15, pilocytic astrocytoma in 16, WHO Grade II astrocytoma in 10, and malignant astrocytoma in 9. A GTR was achieved in 60 cases (59%). Independent of histological tumor type, an intraoperatively identifiable tumor plane (OR 25.3, p < 0.0001) and decreasing tumor size (OR 1.2, p = 0.05) were associated with GTR. Thirty-four patients (34%) experienced acute neurological decline after surgery (associated with increasing age [OR 1.04, p = 0.02] and with intraoperative change in motor evoked potentials [OR 7.4, p = 0.003]); in 14 (41%) of these patients the change returned to preoperative baseline within 1 month. In 31 patients (31%) tumor progression developed by last follow-up (mean 19 months). Tumor histology (p < 0.0001) and the presence of an intraoperatively identified tumor plane (hazard ratio [HR] 0.44, p = 0.027) correlated with improved PFS. A GTR resulted in improved PFS for hemangioblastoma (HR 0.004, p = 0.04) and ependymoma (HR 0.2, p = 0.02), but not astrocytoma. Fifty-five patients (55%) maintained overall neurological improvement by last follow-up. The presence of an identifiable tumor plane (HR 3.1, p = 0.0004) and improvement in neurological symptoms before discharge (HR 2.3, p = 0.004) were associated with overall neurological improvement by last follow-up (mean 19 months).

Conclusions

Gross-total resection can be safely achieved in the vast majority of IMSCTs when an intraoperative plane is identified, independent of pathological type. The incidence of acute perioperative neurological decline increases with patient age but will improve to baseline in nearly half of patients within 1 month. Long-term improvement in motor, sensory, and bladder dysfunction may be achieved in a slight majority of patients and occurs more frequently in patients in whom a surgical plane can be identified. A GTR should be attempted for ependymoma and hemangioblastoma, but it may not affect PFS for astrocytoma. For all tumors, the intraoperative finding of a clear tumor plane of resection carries positive prognostic significance across all pathological types.

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Giannina L. Garcés-Ambrossi, Matthew J. McGirt, Roger Samuels, Daniel M. Sciubba, Ali Bydon, Ziya L. Gokaslan and George I. Jallo

Object

Although postsurgical neurological outcomes in patients with tethered cord syndrome (TCS) are well known, the rate and development of neurological improvement after first-time tethered cord release is incompletely understood. The authors reviewed their institutional experience with the surgical management of adult TCS to assess the time course of symptomatic improvement, and to identify the patient subgroups most likely to experience improvement of motor symptoms.

Methods

The authors retrospectively reviewed 29 consecutive cases of first-time adult tethered cord release. Clinical symptoms of pain and motor and urinary dysfunction were evaluated at 1 and 3 months after surgery, and then every 6 months thereafter. Rates of improvement in pain and motor or urinary dysfunction over time were identified, and presenting factors associated with improvement of motor symptoms were assessed using a multivariate survival analysis (Cox model).

Results

The mean patient age was 38 ± 13 years. The causes of TCS included lipomyelomeningocele in 3 patients (10%), tight filum in 3 (10%), lumbosacral lipoma in 4 (14%), intradural tumor in 3 (10%), previous lumbosacral surgery in 2 (7%), and previous repair of myelomeningocele in 14 (48%). The mean ± SD duration of symptoms before presentation was 5 ± 7 months. Clinical presentation included diffuse pain/parasthesias in both lower extremities in 13 patients (45%), or perineal distribution in 18 (62%), lower extremity weakness in 17 (59%), gait difficulties in 17 (59%), and bladder dysfunction in 14 (48%). Laminectomy was performed in a mean of 2.5 ± 0.7 levels per patient, and 9 patients (30%) received duraplasty. At 18 months postoperatively, 47% of patients had improved urinary symptoms, 69% had improved lower extremity weakness and gait, and 79% had decreased painful dysesthesias. Median time to symptomatic improvement was least for pain (1 month), then motor (2.3 months), and then urinary symptoms (4.3 months; p = 0.04). In patients demonstrating improvement, 96% improved within 6 months of surgery. Only 4% improved beyond 1-year postoperatively. In a multivariate analysis, the authors found that patients who presented with asymmetrical lower extremity weakness (p = 0.0021, hazard ratio 5.7) or lower extremity hyperreflexia (p = 0.037, hazard ratio = 4.1) were most likely to experience improvement in motor symptoms.

Conclusions

In the authors' experience, pain and motor and urinary dysfunction improve postoperatively in the majority of patients. The rate of symptomatic improvement was greatest for pain resolution, followed by motor, and then urinary improvement. Patients who experienced improvement in any symptom had done so by 6 months after tethered cord release. Patients with asymmetrical motor symptoms or lower extremity hyperreflexia at presentation were most likely to experience improvements in motor symptoms. These findings may help guide patient education and surgical decision-making.

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Matthew J. McGirt, Vivek Mehta, Giannina Garces-Ambrossi, Oren Gottfried, Can Solakoglu, Ziya L. Gokaslan, Amer Samdani and George I. Jallo

Object

Tethered cord syndrome (TCS) is frequently associated with scoliosis in the pediatric population. Following spinal cord untethering, many patients continue to experience progression of spinal deformity. However, the incidence rate, time course, and risk factors for scoliosis progression following tethered cord release remain unclear. The aim of this study was to determine factors associated with scoliosis progression and whether tethered cord release alone would halt curve progression in pediatric TCS.

Methods

The authors retrospectively reviewed 27 consecutive pediatric cases of spinal cord untethering associated with scoliosis. The incidence rate and factors associated with scoliosis progression (> 10° increased Cobb angle) after untethering were evaluated using the Kaplan-Meier method.

Results

The mean age of the patients was 8.9 years. All patients underwent cord untethering for lower-extremity weakness, back and leg pain, or bowel and bladder changes. Mean ± SD of the Cobb angle at presentation was 41 ± 16°. The cause of the spinal cord tethering included previous myelomeningocele repair in 14 patients (52%), fatty filum in 5 (18.5%), lipomeningocele in 3 (11%), diastematomyelia in 2 (7.4%), arthrogryposis in 1 (3.7%), imperforate anus with an S-2 hemivertebra in 1 (3.7%), and lipomyelomeningocele with occult dysraphism in 1 (3.7%). Mean follow-up was 6 ± 2 years. Twelve patients (44%) experienced scoliosis progression occurring a median of 2.4 years postoperatively and 8 (30%) required subsequent fusion for progression. At the time of untethering, scoliosis < 40° was associated with a 32% incidence of progression, whereas scoliosis > 40° was associated with a 75% incidence of progression (p < 0.01). Patients with Risser Grades 0–2 were also more likely to experience scoliosis progression compared with Risser Grades 3–5 (p < 0.05). Whereas nearly all patients with Risser Grades 0–2 with curves > 40° showed scoliosis progression (83%), 54% of patients with Risser Grades 0–2 with curves < 40° progressed, and no patients with Risser Grades 3–5 with curves < 40° progressed following spinal cord untethering.

Conclusions

In this experience with pediatric TCS-associated scoliosis, patients with Risser Grades 3–5 and Cobb angles < 40° did not experience curve progression after tethered cord release. Patients with Risser Grades 0–2 and Cobb angles > 40° were at greatest risk of curve progression after cord untethering. Pediatric patients with TCSassociated scoliosis should be monitored closely for curve progression using standing radiographs after spinal cord untethering, particularly those with curves > 40° or who have Risser Grades 0–2.

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Sophia F. Shakur, Matthew J. McGirt, Michael W. Johnson, Peter C. Burger, Edward Ahn, Benjamin S. Carson and George I. Jallo

Object

Angiocentric glioma was recently recognized as a distinct clinicopathological entity in the 2007 World Health Organization Classification of Tumours of the Central Nervous System. The authors present the first 3 pediatric cases of angiocentric glioma encountered at their institution and review the literature of reported cases to elucidate the characteristics and outcomes of pediatric patients with this novel tumor.

Methods

The children in the 3 cases of angiocentric glioma were 10, 10, and 13 years old. Two presented with intractable seizures and 1 with worsening headache and several months of decreasing visual acuity. Twenty-five cases, including the 3 first described in the present paper, were culled from the literature.

Results

In all 3 cases, MR imaging demonstrated a superficial, nonenhancing, T2-hyperintense lesion in the left temporal lobe. Histologically, the tumors were composed of monomorphous cells with a strikingly perivascular orientation that were variably reactive for glial fibrillary acidic protein and epithelial membrane antigen. Surgical treatment resulted in gross-total resection in all 3 cases. By 24, 9, and 6 months after surgery, all 3 patients remained seizure free without focal neurological deficits.

Conclusions

Among 25 cases of angiocentric glioma, seizure was the most common symptom at presentation. Magnetic resonance imaging demonstrated supratentorial, nonenhancing, T1-hypointense, T2-hyperintense lesions. Gross-total resection of this lesion yields excellent results.

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Matthew J. McGirt, Shlomi Constantini and George I. Jallo

Object

Postoperative progressive spinal deformity often complicates functional outcome after resection of pediatric intramedullary spinal cord tumors (IMSCTs). The authors propose a preoperative grading scale that correlates with the postoperative development of progressive spinal deformity requiring subsequent fusion.

Methods

The data obtained in 164 patients who underwent resection of an IMSCT at a single institution were retrospectively collected and analyzed to determine the development of progressive spinal deformity requiring fusion. A grading scale (range of scores I–V) was created based on the presence or absence of 4 preoperative variables: preoperative scoliosis, involvement of the thorocolumbar junction, age < 13 years, and number of surgeries for an IMSCT. The grading scale was then retrospectively applied to this series of 164 children to assess the correlation of variables with subsequent spinal deformity.

Results

Nine patients presented with Grade I status, 41 patients with Grade II, 58 patients with Grade III, 44 patients with Grade IV, and 12 patients with Grade V. Overall, 44 patients (27%) developed progressive spinal deformity requiring fusion at a mean follow-up of 5 years after surgery. A higher preoperative grade was associated with an increasing need for subsequent fusion for progressive spinal deformity (Grade I [0%], Grade II [5%], Grade III [26%], Grade IV [40%], and Grade V status [75%]).

Conclusions

Application of this grading scheme to a series of resected pediatric IMSCTs has demonstrated its correlation with the incidence of postoperative progressive spinal deformity requiring fusion. The application of a standardized grading scheme will assist in the process of surgical decision making and postoperative evaluation.

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Matthew J. McGirt, Frank J. Attenello, Ghazala Datoo, Muraya Gathinji, April Atiba, Jon D. Weingart, Benjamin Carson and George I. Jallo

Object

Indications for duraplasty in treatment of Chiari malformation Type I (CM-I) remain unclear. In the present study, the authors evaluate their surgical experience to determine whether intraoperative ultrasonography is effective in the selection of patients with CM-I who can be adequately treated with craniectomy alone without duraplasty.

Methods

The authors reviewed the records of 256 children who underwent first-time hindbrain decompression for CM-I. Craniectomy alone (without duraplasty) was performed when intraoperative ultrasonography suggested adequate decompression of the subarachnoid spaces ventral and dorsal to the tonsils after suboccipital craniectomy alone. Duraplasty was performed if intraoperative ultrasonography demonstrated persistent dural compression of the tonsils following craniectomy. Symptom recurrence as a function of time was compared between cases of duraplasty versus suboccipital decompression alone stratified by extent of tonsillar herniation.

Results

Duraplasty was performed in 140 patients (55%), and suboccipital decompression alone was performed in 116 patients (45%). Patients underwent follow-up for 29 ± 15 months. Symptoms included headache in 192 patients (75%) and lower cranial nerve and brainstem dysfunction in 68 (27%). In 38 patients (15%) there was tonsillar herniation rostral to the C-1 lamina, in 195 (76%) it extended between the C-1 and C-2 lamina, and in 23 patients (9%) there was herniation caudal to the lower border of the C-2 lamina. In children with tonsillar herniation caudal to C-1, ultrasonography-guided suboccipital decompression alone was associated with a 2-fold increase in the risk of symptom recurrence compared with those who also underwent duraplasty (p = 0.01). In children with tonsillar herniation rostral to C-1, outcome was equivalent between suboccipital decompression alone and duraplasty (p = 0.41).

Conclusions

In the setting of moderate-to-severe tonsillar CM-I, intraoperative ultrasonography demonstrating decompression of the subarachnoid spaces ventral and dorsal to the tonsils may not effectively select patients in whom bone decompression alone is sufficient. Duraplasty may be warranted in cases of tonsillar herniation that extends below the C-1 lamina regardless of intraoperative ultrasonography findings. More objective cerebrospinal fluid flow or volumetric measures may be needed intraoperatively to guide duraplasty in patients with more pronounced tonsillar herniation.

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Frank J. Attenello, Matthew J. McGirt, April Atiba, Muraya Gathinji, Ghazala Datoo, Jon Weingart, Benjamin Carson and George I. Jallo

Object

Chiari malformation Type I (CM-I) is often associated with scoliosis. It remains unclear which subgroups of patients are most likely to experience progression of spinal deformity after cervicomedullary decompression. The authors' goal was to determine the time frame of curvature progression and assess which patient subgroups are at greatest risk for progression of spinal deformity after surgery.

Methods

The authors retrospectively reviewed the records of all pediatric patients with significant scoliosis in whom suboccipital decompression was performed to treat for CM-I during a 10-year period at a single academic institution. Clinical, radiological, and operative variables were assessed as independent factors for failure (worsening of scoliosis) by using a univariate regression analysis.

Results

Twenty-one children (mean age 9 ± 3 years; 4 male) underwent hindbrain decompression for CM-I–associated scoliosis and were followed for a mean of 39 months. All patients harbored a syrinx. Eight patients (38%) experienced improvement in scoliosis curvature, whereas 10 (48%) suffered a progression. Thoracolumbar junction scoliosis (p = 0.04) and failure of the syrinx to improve (p = 0.05) were associated with 5- and 4-fold respective increases in the likelihood of deformity progression. Each increasing degree of preoperative Cobb angle was associated with an 11% increase in the likelihood of scoliotic curve progression (p < 0.05).

Conclusions

Over one third of patients with CM-I–associated scoliosis will improve after cervicomedullary decompression alone. Cervicomedullary decompression is a good first-line option, particularly in children with concordant posterior fossa symptoms. Patients presenting with more severe scoliosis (increasing Cobb angle) or scoliosis that crosses the thoracolumbar junction may benefit from earlier orthopedic involvement and should be monitored regularly for curvature progression after cervicomedullary decompression. In cases in which there is a failure of the syrinx to show improvement after suboccipital decompression, the patients are also more likely to develop curvature progression.

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Daniel M. Sciubba, Kaisorn L. Chaichana, Graeme F. Woodworth, Matthew J. McGirt, Ziya L. Gokaslan and George I. Jallo

Object

The indications remain unclear for fusion at the time of cervical laminectomy for intradural tumor resection. To identify patients who may benefit from initial fusion, the authors assessed clinical, radiological/imaging, and operative factors associated with subsequent symptomatic cervical instability requiring fusion after cervical laminectomy for intradural tumor resection.

Methods

The authors reviewed 10 years of data obtained in patients who underwent cervical laminectomy without fusion for intradural tumor resection and who had normal spinal stability and alignment preoperatively. The association of pre- and intraoperative variables with the subsequent need for fusion for progressive symptomatic cervical instability was assessed using logistic regression analysis, and percentages were compared using Fisher exact tests when appropriate.

Results

Thirty-two patients (mean age 41 ± 17 years) underwent cervical laminectomy without fusion for resection of an intradural tumor (18 intramedullary and 14 extramedullary). Each increasing number of laminectomies performed was associated with a 3.1-fold increase in the likelihood of subsequent vertebral instability (odds ratio 3.114, 95% confidence interval 1.207–8.034, p = 0.02). At a mean follow-up interval of 25.2 months, 33% (4 of 12) of the patients who had undergone a ≥ 3-level laminectomy required subsequent fusion compared with 5% (1 of 20) who had undergone a ≤ 2-level laminectomy (p = 0.03). Four (36%) of 11 patients initially presenting with myelopathic motor disturbance required subsequent fusion compared with 1 (5%) of 21 presenting initially with myelopathic sensory or radicular symptoms (p = 0.02). Age, the presence of a syrinx, intramedullary tumor, C-2 laminectomy, C-7 laminectomy, and laminoplasty were not associated with subsequent symptomatic instability requiring fusion.

Conclusions

In the authors' experience with intradural cervical tumor resection, patients presenting with myelopathic motor symptoms or those undergoing a ≥ 3-level cervical laminectomy had an increased likelihood of developing subsequent symptomatic instability requiring fusion. A ≥ 3-level laminectomy with myelopathic motor symptoms may herald patients most likely to benefit from cervical fusion at the time of tumor resection.