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Kai J. Miller, Taylor J. Abel, Adam O. Hebb, and Jeffrey G. Ojemann

Object

Emerging research in evoked broadband electrocorticographic (ECoG) measurement from the cortical surface suggests that it might cleanly delineate the functional organization of cortex. The authors sought to demonstrate whether this could be done in a same-session, online manner to identify receptive and expressive language areas.

Methods

The authors assessed the efficacy of simple integration of “χ-band” (76–200 Hz) change in the ECoG signal by implementing a simple band-pass filter to estimate broadband spectral change. Following a brief (less than 10-second) period to characterize baseline activity, χ-band activity was integrated while 7 epileptic patients with implanted ECoG electrodes performed a verb-generation task.

Results

While the patients were performing verb-generation or noun-reading tasks, cortical activation was consistently identified in primary mouth motor area, superior temporal gyrus, and Broca and Wernicke association areas. Maps were robust after a mean time of 47seconds (using an “activation overlap” measure). Correlation with electrocortical stimulation was not complete and was stronger for noun reading than verb generation.

Conclusions

Broadband ECoG changes can be captured online to identify eloquent cortex. This demonstrates the existence of a powerful new tool for functional mapping in the operative and chronic implant setting.

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Abhineet Chowdhary, Taylor J. Abel, and Anthony M. Avellino

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Taylor J. Abel, Brian D. Dalm, Andrew J. Grossbach, Adam W. Jackson, Teri Thomsen, and Jeremy D. W. Greenlee

Lesch-Nyhan disease (LND) is an X-linked hereditary disorder caused by a deficiency of hypoxanthine-guanine phosphoribosyltransferase. This syndrome is characterized by hyperuricemia, self-mutilation, cognitive impairment, and movement disorders such as spasticity and dystonia. The authors describe the case of a 15-year-old boy who underwent bilateral placement of globus pallidus internus (GPi) deep brain stimulation (DBS) electrodes for the treatment of generalized dystonia. His self-mutilating behavior gradually disappeared several weeks after the start of GPi stimulation. The dystonia and self-mutilating behavior returned on the left side only after a right lead fracture. This case is the first reported instance of LND treated with DBS in which the stimulation was interrupted and the self-mutilation returned in a lateralized fashion. The findings indicate that the neurobehavioral aspect of LND is lateralized and that contralateral GPi stimulation is responsible for lateralized improvement in self-injurious behavior.

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Taylor J. Abel, René Varela Osorio, Ricardo Amorim-Leite, Francois Mathieu, Philippe Kahane, Lorella Minotti, Dominique Hoffmann, and Stephan Chabardes

OBJECTIVE

Robot-assisted stereoelectroencephalography (SEEG) is gaining popularity as a technique for localization of the epileptogenic zone (EZ) in children with pharmacoresistant epilepsy. Here, the authors describe their frameless robot-assisted SEEG technique and report preliminary outcomes and relative complications in children as compared to results with the Talairach frame–based SEEG technique.

METHODS

The authors retrospectively analyzed the results of 19 robot-assisted SEEG electrode implantations in 17 consecutive children (age < 17 years) with pharmacoresistant epilepsy, and compared these results to 19 preceding SEEG electrode implantations in 18 children who underwent the traditional Talairach frame–based SEEG electrode implantation. The primary end points were seizure-freedom rates, operating time, and complication rates.

RESULTS

Seventeen children (age < 17 years) underwent a total of 19 robot-assisted SEEG electrode implantations. In total, 265 electrodes were implanted. Twelve children went on to have EZ resection: 4 demonstrated Engel class I outcomes, whereas 2 had Engel class II outcomes, and 6 had Engel class III–IV outcomes. Of the 5 patients who did not have resection, 2 underwent thermocoagulation. One child reported transient paresthesia associated with 2 small subdural hematomas, and 3 other children had minor asymptomatic intracranial hemorrhages. There were no differences in complication rates, rates of resective epilepsy surgery, or seizure freedom rates between this cohort and the preceding 18 children who underwent Talairach frame–based SEEG. The frameless robot-assisted technique was associated with shorter operating time (p < 0.05).

CONCLUSIONS

Frameless robot-assisted SEEG is a safe and effective means of identifying the EZ in children with pharmacoresistant partial epilepsy. Robot-assisted SEEG is faster than the Talairach frame–based method, and has equivalent safety and efficacy. The former, furthermore, facilitates more electrode trajectory possibilities, which may improve the localization of epileptic networks.

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Taylor J. Abel, Abhineet Chowdhary, Mahesh Thapa, Joseph C. Rutledge, Joseph Gruss, Scott Manning, and Anthony M. Avellino

The growth of ectopic glioneuronal tissue in the middle cranial fossa region is an uncommon event, with very few cases reported in the literature. In this paper the authors document 4 cases of ectopic glioneuronal tissue in the middle cranial fossa in children and briefly describe the clinical course and pathology. All of the children presented within the first 6 months of life. Two children presented with facial masses, 1 with airway obstruction, and another with proptosis of the right eye. Each child underwent a customized surgery dependent on the location and characteristics of the harbored lesion. Ectopic glioneuronal masses in the middle cranial fossa are rare and benign congenital tumors, and affected newborns can present with airway obstruction, feeding difficulties, and facial deformity depending on the lesion location. Determining an appropriate surgical approach and strategy is a significant challenge and may involve a multidisciplinary team of craniofacial plastic surgeons, otolaryngologists, and neurosurgeons. Although these lesions share clinical and anatomical similarities, because of their histopathological heterogeneity, it is unlikely that they represent a single pathological entity. The long-term outcome in these children is still unknown and is an area for future study. The pathogenesis of these lesions also remains unknown and may be revealed in future research.

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Han Yan, Eric Toyota, Melanie Anderson, Taylor J. Abel, Elizabeth Donner, Suneil K. Kalia, James Drake, James T. Rutka, and George M. Ibrahim

OBJECTIVE

Drug-resistant epilepsy (DRE) presents a therapeutic challenge in children, necessitating the consideration of multiple treatment options. Although deep brain stimulation (DBS) has been studied in adults with DRE, little evidence is available to guide clinicians regarding the application of this potentially valuable tool in children. Here, the authors present the first systematic review aimed at understanding the safety and efficacy of DBS for DRE in pediatric populations, emphasizing patient selection, device placement and programming, and seizure outcomes.

METHODS

The systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines and recommendations. Relevant articles were identified from 3 electronic databases (MEDLINE, Embase, and Cochrane CENTRAL) from their inception to November 17, 2017. Inclusion criteria of individual studies were 1) diagnosis of DRE; 2) treatment with DBS; 3) inclusion of at least 1 pediatric patient (age ≤ 18 years); and 4) patient-specific data. Exclusion criteria for the systematic review included 1) missing data for age, DBS target, or seizure freedom; 2) nonhuman subjects; and 3) editorials, abstracts, review articles, and dissertations.

RESULTS

This review identified 21 studies and 40 unique pediatric patients (ages 4–18 years) who received DBS treatment for epilepsy. There were 18 patients with electrodes placed in the bilateral or unilateral centromedian nucleus of the thalamus (CM) electrodes, 8 patients with bilateral anterior thalamic nucleus (ATN) electrodes, 5 patients with bilateral and unilateral hippocampal electrodes, 3 patients with bilateral subthalamic nucleus (STN) and 1 patient with unilateral STN electrodes, 2 patients with bilateral posteromedial hypothalamus electrodes, 2 patients with unilateral mammillothalamic tract electrodes, and 1 patient with caudal zona incerta electrode placement. Overall, 5 of the 40 (12.5%) patients had an International League Against Epilepsy class I (i.e., seizure-free) outcome, and 34 of the 40 (85%) patients had seizure reduction with DBS stimulation.

CONCLUSIONS

DBS is an alternative or adjuvant treatment for children with DRE. Prospective registries and future clinical trials are needed to identify the optimal DBS target, although favorable outcomes are reported with both CM and ATN in children.