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Isaac Josh Abecassis, John D. Nerva, Jason Barber, Jason Rockhill, Richard G. Ellenbogen, Louis J. Kim and Laligam N. Sekhar

OBJECTIVE

Brain arteriovenous malformations (bAVMs) are rare in pediatric patients but represent the most common cause of hemorrhagic stroke in this population. Pediatric patients demonstrate superior outcomes in comparison with adult patients with similar lesions and presentations. Most studies of clinical outcomes of pediatric bAVMs use the modified Rankin Scale (mRS), despite a lack of validation in pediatric patients.

METHODS

The authors interviewed the parents of 26 pediatric patients who underwent multimodality bAVM treatment and administered the Pediatric Quality of Life Inventory (PedsQL)—a well-validated tool for pediatric outcomes that quantifies performance in a physical, emotional, social, and school domains. They also reviewed clinical information from the patients' medical charts. Statistical analysis was performed using a log-transformed t-test, the Mann-Whitney exact test, the Kruskal-Wallis test, and Spearman correlation. In addition, the literature was reviewed for prior reports of clinical outcome of pediatric cases of bAVM.

RESULTS

The average PedsQL health-related quality of life score was 71 ± 24, with an average age at diagnosis of 12.5 years and an average follow-up period of 6.8 years. Seventeen patients (65%) presented with hemorrhage and 4 (15%) with seizures. PedsQL scores correlated strongly and at a statistically significant level (p < 0.001) with mRS, Pediatric Overall Performance Category (POPC), Pediatric Cerebral Performance Category (PCPC), and Glasgow Outcome Scale scores. Multivariate modeling validated special education, corrective devices, and cure status as significant predictors of PedsQL scores. Statistically significant risk factors for undergoing placement of a ventriculoperitoneal shunt included lower Glasgow Coma Scale motor scores on admission (p = 0.042), cerebellar location (p = 0.046), and nidus volume (p = 0.017). Neither treatment modality nor location statistically affected clinical outcomes at follow-up.

CONCLUSIONS

There have been few studies of long-term clinical outcomes of bAVM in pediatric patients, and previously published studies have used conventional metrics that have been validated in the adult population, such as the mRS. Although these metrics can serve as reasonable surrogates, an accurate understanding of overall health-related quality of life is contingent on utilizing validated toolsets, such as the PedsQL.

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Kim J. Burchiel

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Max C. Lee, Michael Y. Wang, Richard G. Fessler, Jason Liauw and Daniel H. Kim

Object

Placement of instrumentation in the setting of a spinal infection has always been controversial. Although the use of allograft and autograft bone has been accepted as safe, demonstrations of the effectiveness of titanium have been speculative, based on several retrospective reviews. The authors' goal in this study was to demonstrate the effectiveness of instrumentation in the setting of a spinal infection by retrospectively reviewing their cases over the last 4 years and searching the literature regarding instrumentation in patients with pyogenic spinal infections.

Methods

The authors conducted a retrospective review of their cumulative data on spinal infections. Diagnosis was based on subjective and objective clinical findings, along with radiographic and laboratory evaluation of infection and mechanical stability. Patients with medically managed disease and those who did not receive instrumentation were eliminated from this review.

Of 105 patients with spinal infections who were admitted to the neurosurgical service between January 2000 and June 2004, 30 underwent surgical debridement necessitating spinal instrumentation. There were 17 women and 13 men in this group ranging from 28 to 86 years of age. Follow-up duration ranged from 3 to 54 months. There was one death, which occurred 3 months postsurgery. In three patients a deep wound infection developed, necessitating intervention, and two patients experienced a graft expulsion. Twenty-nine patients went on to demonstrate adequate fusion based on follow-up neuroimaging studies.

Conclusions

The goal of neurosurgical intervention in the setting of spinal infection is to obtain an organism culture and the debridement of infection while maintaining neurological and mechanical stability. The authors demonstrate the effectiveness of radical debridement of infected bone and placement of instrumentation in patients with spinal infections.

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Jeffrey P. Blount, Jason Cormier, Hyunmi Kim, Pongkiat Kankirawatana, Kristen O. Riley and Robert C. Knowlton

Intracranial monitoring using electroencephalography (IC-EEG) continues to play a critical role in the assessment of patients with medically intractable localization-related epilepsy. There has been minimal change in grid or electrode design in the last 15–20 years, and the surgical approaches for implantation are unchanged. Intracranial monitoring using EEG allows detailed definition of the region of ictal onset and defines the epileptogenic zone, particularly with regard to adjacent potentially eloquent tissue. Recent developments of IC-EEG include the coregistration of functional imaging data such as magnetoencephalography to the frameless navigation systems. Despite significant inherent limitations that are often overlooked, IC-EEG remains the gold standard for localization of the epileptogenic cortex. Intracranial electrodes take a variety of different forms and may be placed either in the subdural (subdural strips and grids, depth electrodes) or extradural spaces (sphenoidal, peg, and epidural electrodes). Each form has its own advantages and shortcomings but extensive subdural implantation of electrodes is most common and is most comprehensively discussed. The indications for intracranial electrodes are reviewed.

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Yiping Li, Jason Kim, Dustin Simpson, Beverly Aagaard-Kienitz, David Niemann, Ignatius N. Esene and Azam Ahmed

OBJECTIVE

The literature suggests that blood-brain barrier disruption (BBBD) plays a significant role in the development of neurological events in patients with diffusion-weighted imaging (DWI) that is negative for lesions. In this prospective, single-center cohort study, the authors compared the imaging characteristics of patients suffering transient neurological events (TNEs) with those in patients suffering permanent neurological events (PNEs) after having undergone elective embolization of unruptured intracranial aneurysms.

METHODS

This prospective cohort study was conducted between July 2016 and June 2019. Inclusion criteria were adults undergoing elective neuroendovascular procedures and the absence of contraindications to MRI. All subjects underwent brain MRI including postcontrast FLAIR (pcFLAIR) sequences for evaluation of BBBD within 24 hours postprocedure.

RESULTS

In total, 128 patients harboring 133 unruptured aneurysms were enrolled, 109 of whom (85.2%) showed some degree of BBBD on pcFLAIR MRI and 50 of whom (39.1%) suffered an ischemic insult per DWI. In total, 23 patients (18%) suffered neurological complications, 16 of which (12.5%) were TNEs and 7 of which (5.5%) were PNEs. The median extent of BBBD was focal in asymptomatic patients as compared to hemispheric and lobar in the TNE and PNE groups, respectively (p < 0.001). The American Society of Anesthesiologists physical status classification predicted the extent of BBBD (p = 0.046).

Lesions on DWI were noted in 34 asymptomatic patients (32.4%) compared to 9 patients (56.3%) with TNEs and all 7 patients (100%) with PNEs (p < 0.001). The median number of DWI lesions was 0 (range 0–18 lesions) in the asymptomatic group compared to 1.5 (range 0–8 lesions) and 8 (range 1–13 lesions) in the TNE and PNE groups, respectively (p < 0.001). Smoking (p = 0.008), older age (p = 0.002), and longer surgery (p = 0.006) were positively associated with the number of lesions on DWI.

On multivariate analysis, intraarterial verapamil (p = 0.02, OR 8.01, 95% CI 1.35–47.43) and extent of BBBD (p < 0.001, OR 58.58, 95% CI 9.48–361.84) were positively associated with the development of TNEs, while intravenous infusion of midazolam during surgery (p = 0.02, OR 6.03, 95% CI 1.29–28.20) was negatively associated. An increased number of lesions on DWI was the only significant predictor for the development of PNEs (p < 0.001, OR 49.85, 95% CI 5.56–447.10).

CONCLUSIONS

An increasing extent of BBBD was associated with the development of TNEs, whereas an increasing number of lesions on DWI was significantly associated with the development of PNEs. BBBD imaging using pcFLAIR may serve as a valuable biomarker for detecting subtle cerebral ischemia and stratifying the risk for ischemic events.

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Justin M. Caplan, Mari Groves, Ignacio Jusue-Torres, Jennifer E. Kim, Jason Liauw, Ali Bydon and Rafael J. Tamargo

Spinal vascular lesions are rare and may be classified as a) dural arteriovenous fistulas (AVFs), b) arteriovenous malformations, or c) perimedullary AVFs. In this narrated video illustration, we present the case of a 71-year-old woman who presented with progressive bilateral lower extremity weakness and urinary retention who was diagnosed with a thoracic spinal perimedullary arteriovenous fistula. The diagnostic studies included a thoracic MRI and spinal angiogram. A multilevel thoracic laminoplasty was performed for microsurgical obliteration of the AVF. The techniques of intraoperative angiography, thoracic laminoplasty and microsurgical obliteration and resection of the AVF are reviewed.

The video can be found here: http://youtu.be/5vVp3oq5sLg.

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Pongkiat Kankirawatana, Ismail S. Mohamed, Jason Lauer, Inmaculada Aban, Hyunmi Kim, Rong Li, Allan Harrison, AS, Monisha Goyal, Curtis J. Rozzelle, Robert Knowlton and Jeffrey P. Blount

OBJECTIVE

The goal of this study was to evaluate the predictive value and relative contribution of noninvasive presurgical functional imaging modalities based on the authors’ institutional experience in pursuing seizure-free surgical outcomes in children with medically refractory epilepsy.

METHODS

This was a retrospective, single-institution, observational cohort study of pediatric patients who underwent evaluation and surgical treatment for medically refractory partial epilepsy between December 2003 and June 2016. During this interval, 108 children with medically refractory partial epilepsy underwent evaluation for localization and resective epilepsy surgery. Different noninvasive functional imaging modalities, including ictal SPECT, FDG-PET, and magnetoencephalography–magnetic source imaging, were utilized to augment a standardized paradigm (electroencephalography/semiology, MRI, and neuropsychology findings) for localization. Outcomes were evaluated at a minimum of 2 years (mean 7.5 years) utilizing area under the receiver operating characteristic curve analysis. Localizing modalities and other clinical covariates were examined in relation to long-term surgical outcomes.

RESULTS

There was variation in the contribution of each test, and no single presurgical workup modality could singularly and reliably predict a seizure-free outcome. However, concordance of presurgical modalities yielded a high predictive value. No difference in long-term outcomes between inconclusive (normal or diffusely abnormal) and abnormal focal MRI results were found. Long-term survival analyses revealed a statistically significant association between seizure freedom and patients with focal ictal EEG, early surgical intervention, and no history of generalized convulsions.

CONCLUSIONS

Comprehensive preoperative evaluation utilizing multiple noninvasive functional imaging modalities is not redundant and can improve pediatric epilepsy surgical outcomes.

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Justin M. Caplan, Ignacio Jusue-Torres, Jennifer E. Kim, Andrew Luksik, Jason Liauw, Allan Gottschalk and Rafael J. Tamargo

Aneurysms of the posterior circulation remain challenging lesions given their proximity to the brainstem and cranial nerves. Many of these aneurysms may best be approached through a retrosigmoid-suboccipital craniectomy with a far-lateral transcondylar extension. In this narrated video illustration, we present the case of a 37-year-old man with an incidentally discovered right-sided anterior inferior cerebellar artery (AICA) aneurysm. Diagnostic studies included CT angiography and cerebral angiography. A suboccipital craniectomy and far-lateral transcondylar extension were performed for microsurgical trapping and excision of the AICA aneurysm. The techniques of the retrosigmoid craniectomy, C-1 laminectomy, condylectomy and microsurgical trapping of the aneurysm are reviewed.

The video can be found here: http://youtu.be/JiM3CXVwXnk.

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Jason S. Weinstein, Kenneth C. Liu, Johnny B. Delashaw Jr., Kim J. Burchiel, Harry R. van Loveren, Fernando L. Vale, Siviero Agazzi, Mark S. Greenberg, Donald A. Smith and John Tew Jr.

Object

The DuraSeal dural sealant system, a polyethylene glycol hydrogel, has been shown to be safe and effective when used with commercial and autologous duraplasty materials. The authors report on the safety and effectiveness of this sealant when used in conjunction with nonautologous duraplasty materials.

Methods

In this retrospective, nonrandomized, multicenter study, the safety and efficacy of a dural sealant system was assessed in conjunction with primarily collagen-based nonautologous duraplasty materials in a sample of 66 patients undergoing elective cranial procedures at 3 institutions. This cohort was compared with 50 well-matched patients from the DuraSeal Pivotal Trial who were treated with this sealant system and autologous duraplasty material.

Results

The key end points of the study were the incidences of CSF leaks, surgical site infections, and meningitis 90 days after surgery. The incidence of postoperative CSF leakage was 7.6% in the study group (retrospective population) and 6.0% in the Pivotal Trial population. The incidence of meningitis was 0% and 4.0% in the retrospective and Pivotal Trial groups, respectively. There were no serious device-related adverse events or unanticipated adverse device effects noted for either population.

Conclusions

This study demonstrates that the DuraSeal sealant system is safe and effective when used for watertight dural closure in conjunction with nonautologous duraplasty materials.

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John Y. K. Lee, Steve S. Cho, Ryan Zeh, John T. Pierce, Maria Martinez-Lage, Nithin D. Adappa, James N. Palmer, Jason G. Newman, Kim O. Learned, Caitlin White, Julia Kharlip, Peter Snyder, Philip S. Low, Sunil Singhal and M. Sean Grady

OBJECTIVE

Pituitary adenomas account for approximately 10% of intracranial tumors and have an estimated prevalence of 15%–20% in the general US population. Resection is the primary treatment for pituitary adenomas, and the transsphenoidal approach remains the most common. The greatest challenge with pituitary adenomas is that 20% of patients develop tumor recurrence. Current approaches to reduce recurrence, such as intraoperative MRI, are costly, associated with high false-positive rates, and not recommended. Pituitary adenomas are known to overexpress folate receptor alpha (FRα), and it was hypothesized that OTL38, a folate analog conjugated to a near-infrared (NIR) fluorescent dye, could provide real-time intraoperative visual contrast of the tumor versus the surrounding nonneoplastic tissues. The preliminary results of this novel clinical trial are presented.

METHODS

Nineteen adult patients who presented with pituitary adenoma were enrolled. Patients were infused with OTL38 2–4 hours prior to surgery. A 4-mm endoscope with both visible and NIR light capabilities was used to visualize the pituitary adenoma and its margins in real time during surgery. The signal-to-background ratio (SBR) was recorded for each tumor and surrounding tissues at various endoscope-to-sella distances. Immunohistochemical analysis was performed to assess the FRα expression levels in all specimens and classify patients as having either high or low FRα expression.

RESULTS

Data from 15 patients (4 with null cell adenomas, 1 clinically silent gonadotroph, 1 totally silent somatotroph, 5 with a corticotroph, 3 with somatotrophs, and 1 somatocorticotroph) were analyzed in this preliminary analysis. Four patients were excluded for technical considerations. Intraoperative NIR imaging delineated the main tumors in all 15 patients with an average SBR of 1.9 ± 0.70. The FRα expression level of the adenomas and endoscope-to-sella distance had statistically significant impacts on the fluorescent SBRs. Additional considerations included adenoma functional status and time from OTL38 injection. SBRs were 3.0 ± 0.29 for tumors with high FRα expression (n = 3) and 1.6 ± 0.43 for tumors with low FRα expression (n = 12; p < 0.05). In 3 patients with immunohistochemistry-confirmed FRα overexpression (2 patients with null cell adenoma and 1 patient with clinically silent gonadotroph), intraoperative NIR imaging demonstrated perfect classification of the tumor margins with 100% sensitivity and 100% specificity. In addition, for these 3 patients, intraoperative residual fluorescence predicted postoperative MRI results with perfect concordance.

CONCLUSIONS

Pituitary adenomas and their margins can be intraoperatively visualized with the preoperative injection of OTL38, a folate analog conjugated to NIR dye. Tumor-to-background contrast is most pronounced in adenomas that overexpress FRα. Intraoperative SBR at the appropriate endoscope-to-sella distance can predict adenoma FRα expression status in real time. This work suggests that for adenomas with high FRα expression, it may be possible to identify margins and to predict postoperative MRI findings.