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Benjamin Davidson, Lior M. Elkaim, Nir Lipsman and George M. Ibrahim

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Jetan H. Badhiwala, Brij Karmur, Lior M. Elkaim, Naif M. Alotaibi, Benjamin R. Morgan, Nir Lipsman, Philippe De Vloo, Suneil K. Kalia, Andres M. Lozano and George M. Ibrahim

OBJECTIVE

Although deep brain stimulation (DBS) is an accepted treatment for childhood dystonia, there is significant heterogeneity in treatment response and few data are available to identify ideal surgical candidates.

METHODS

Data were derived from a systematic review and individual patient data meta-analysis of DBS for dystonia in children that was previously published. Outcomes were assessed using the Burke-Fahn-Marsden Dystonia Rating Scale for movement (BFMDRS-M) and for disability (BFMDRS-D). The authors used partial least squares, bootstrapping, and permutation statistics to extract patterns of contributions of specific preoperative characteristics to relationship with distinct outcomes, in all patients and in patients with primary and secondary dystonia separately.

RESULTS

Of 301 children undergoing DBS for dystonia, 167 had primary dystonia, 125 secondary dystonia, and 9 myoclonus dystonia. Three dissociable preoperative phenotypes (latent variables) were identified and associated with the following: 1) BFMDRS-M at last follow-up; 2) relative change in BFMDRS-M score; and 3) relative change in BFMDRS-D score. The phenotype of patients with secondary dystonia, with a high BFMDRS-M score and truncal involvement, undergoing DBS at a younger age, was associated with a worse postoperative BFMDRS-M score. Children with primary dystonia involving the trunk had greater improvement in BFMDRS-M and -D scores. Those with primary dystonia of shorter duration and proportion of life with disease, undergoing globus pallidus DBS, had greater improvements in BFMDRS-D scores at long-term follow-up.

CONCLUSIONS

In a comprehensive, data-driven, multivariate analysis of DBS for childhood dystonia, the authors identified novel and dissociable patient phenotypes associated with distinct outcomes. The findings of this report may inform surgical candidacy for DBS.

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Marie-Andrée Coulombe, Lior M. Elkaim, Naif M. Alotaibi, Daniel A. Gorman, Alexander G. Weil, Aria Fallah, Suneil K. Kalia, Nir Lipsman, Andres M. Lozano and George M. Ibrahim

OBJECTIVE

Gilles de la Tourette syndrome (GTS) is a disorder characterized by motor and vocal tics. Although by definition the onset of GTS is before age 18 years, clinical trials of deep brain stimulation (DBS) have been conducted only in adults. Using individual participant data (IPD) meta-analysis methodology, the current study investigated the safety and efficacy of DBS as a treatment for GTS in children and youth.

METHODS

A systematic review with no date or language restrictions was performed according to the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) statement. Three electronic databases were searched: PubMed, EMBASE, and Web of Science. From 843 articles screened, the IPD of 58 children and youth (ages 12–21 years) extracted from 21 articles were collected and analyzed. A mixed-effects univariable analysis followed by multivariable hierarchical regression was performed using change in the Yale Global Tic Severity Scale (YGTSS) score as the primary outcome and reported measures of comorbidities as secondary outcomes.

RESULTS

The authors’ results showed an average improvement of 57.5% ± 24.6% across studies on the YGTSS. They also found that comorbid depression and stimulation pulse width each correlated negatively with outcome (p < 0.05). In patients with less severe GTS, greater improvements were evident following thalamic stimulation. More than one-quarter (n = 16, 27.6%) of participants experienced side effects, the majority of which were minor.

CONCLUSIONS

DBS in the pediatric population may be an effective option with a moderate safety profile for treatment of GTS in carefully selected children and youth. Large, prospective studies with long-term follow-up are necessary to understand how DBS influences tic symptoms and may alter the natural course of GTS in children.

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Harrison J. Westwick, Lior M. Elkaim, Sami Obaid, Aria Fallah, Albert Tu, George M. Ibrahim and Alexander G. Weil

OBJECTIVE

The lack of access to primary neurosurgical care presents a significant burden for patients in low- and middle-income countries (LMICs). Efforts to decrease the inequities in access to surgical care have become an increasing focus of initiatives and advocacy within the international neurosurgery community. Although residents may represent highly active members of international teams, the extent of their availability for and interest in global neurosurgery opportunities remains unclear. The objectives of this study were to assess the degree of participation, the level of interest, and the perceived barriers for residents of Canadian neurosurgery programs in engaging in neurosurgery initiatives in LMICs.

METHODS

A cross-sectional survey was administered to 144 residents from all accredited Canadian neurosurgery residency programs. The survey was used to determine the level of participation and interest in global neurosurgery initiatives.

RESULTS

The survey response rate from residents at Canadian neurosurgery programs was 45/144 (31%), although not all respondents answered every question; thus, the denominator for each question is not always 45. Most neurosurgery residents (n = 30, 68%) were unaware of any global neurosurgery opportunities available during residency. Despite 64% of residents having an interest in participation (strongly agree/agree; n = 28), with most residents planning on participating as attending staff (strongly agree/agree; n = 28, 64%), only 3 residents (7%) had taken part in LMIC neurosurgery initiatives. The most cited perceived limitations for participation were lack of time (n = 37, 84%) and finances (n = 26, 59%).

CONCLUSIONS

Among surveyed residents from Canadian neurosurgery programs, there was a low participation rate in global neurosurgery initiatives and limited knowledge of available opportunities, despite relatively high interest. Many respondents identified time and financial constraints as barriers to participation. Many residents felt that interest could increase were there a structured program dedicated to global neurosurgery available in their residency program.

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Alexander G. Weil, Aria Fallah, Shelly Wang, George M. Ibrahim, Lior M. Elkaim, Prasanna Jayakar, Ian Miller, Sanjiv Bhatia, Toba N. Niazi and John Ragheb

OBJECTIVE

Although hemispherectomy is an effective treatment for children with intractable hemispheric epilepsy syndromes, as many as 40% of patients eventually develop seizure recurrence. The causes of seizure recurrence in these patients are incompletely understood. The authors sought to evaluate the efficacy of hemispherectomy at their center and determine whether contralateral MRI abnormalities can predict seizure recurrence.

METHODS

A retrospective review of consecutive hemispherectomies performed at Miami Children’s Hospital between January 2000 and June 2014 was performed. Time-to-event analysis was performed. The “event” was defined as any seizures following resective epilepsy surgery (not including seizures in the first postoperative week and auras). Several preoperative variables were analyzed to determine their suitability to predict seizure recurrence following surgery.

RESULTS

Sixty-nine patients (44 boys) with a mean age of 8.2 ± 5.9 years (range 0.1–20.8 years) underwent 72 hemispherectomies; 67 of these were functional hemispherectomies, while another 5 were completion of a previous functional hemispherectomy (2 completions of functional hemispherectomies, 3 anatomical hemispherectomies). The duration of epilepsy was 5.8 ± 5.5 years with 66 cases (91.7%) having daily seizures. Etiology included stroke (n = 28), malformation of cortical development (n = 11), hemimegalencephaly (n = 11), encephalitis (n = 13), and other (n = 7). Engel class I outcome was achieved in 59 (86%) and 56 (81%) patients at 1 and 2 years of follow-up, respectively. The mean time to seizure recurrence was 33.5 ± 31.1 months. In univariate analyses, the absence of contralateral abnormalities on MRI (HR 4.09, 95% CI 1.41–11.89, p = 0.009) was associated with a longer duration of seizure freedom. The presence of contralateral MRI abnormalities was associated with contralateral ictal seizures on preoperative scalp EEG (p = 0.002). Fifteen patients experienced 20 complications (20/72, 27.8%), including the development of hydrocephalus necessitating CSF diversion in 9 cases (13%), hygroma in 1, hemispheric edema in 1, aseptic meningitis in 2, postoperative hemorrhage in 2, infection in 2, ischemic stroke in 2, and blood transfusion–contracted hepatitis C in 1 case.

CONCLUSIONS

Patients with bihemispheric abnormalities, as evidenced by contralateral MRI abnormalities, have a higher risk of earlier seizure recurrence following functional hemispherectomy.