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Laser interstitial thermal therapy compared with open resection for treating subependymal giant cell astrocytoma

Diane J. Aum, Rebecca A. Reynolds, Sean D. McEvoy, Michael Wong, Jarod L. Roland, and Matthew D. Smyth

OBJECTIVE

Subependymal giant cell astrocytomas (SEGAs) are WHO grade 1 tumors associated with tuberous sclerosis that classically arise from the ventricular wall near the caudate groove and foramen of Monro. Laser interstitial thermal therapy (LITT) is a minimally invasive surgical technique, which works by heating a stereotactically placed laser fiber to ablative temperatures under MRI thermometry monitoring. In this paper, the authors present LITT as a surgical alternative to open resection of SEGAs.

METHODS

Twelve patients with SEGAs who underwent 16 procedures between 2007 and 2022 at a single institution were retrospectively reviewed. These patients underwent either open resection or LITT. Clinical data, imaging, recurrence rate, further treatments, and related complications were analyzed.

RESULTS

Among the 16 procedures, 9 were open resection and 7 were LITT. An external ventricular drain was placed in 66% (6/9) of open procedures and 57.1% (4/7) of LITT cases. A septostomy was performed in 56% (5/9) of open procedures and 29% (2/7) of LITT cases. Complication rates were higher in open cases than in LITT procedures (44% vs 0%, p < 0.05). Complications included hydrocephalus, transient venous ischemia, wound infection, and bone flap migration. The median length of hospital stay was 4 days (IQR 3.3–5.5 days) for open cases and 4 days (IQR 3.0–7.0 days) for LITT procedures. Recurrence or progression occurred after 3 open cases and 2 LITT cases (33% vs 33%, p = 0.803). For the recurrences, 2 open cases underwent stereotactic radiosurgery, 1 open case underwent LITT, and 1 LITT case underwent repeat LITT. Among the LITT cases, only the patients with no decrease in tumor size by 6 months experienced tumor progression afterward. The 2 LITT cases with progression were the only ones with calcification present on preoperative imaging. The median follow-up times for cases assessed for progression were 8.4 years (IQR 3.8–14.4 years) for open resection and 3.9 years (IQR 3.4–5.1 years) for LITT.

CONCLUSIONS

The small size of this case series limits generalizability or adequate comparison of safety. However, this series adds to the literature supporting LITT as a less invasive surgical alternative to open resection of SEGAs and demonstrates that LITT has similar recurrence and/or progression rates to open resection. Additional studies with more data are necessary for comprehensive comparisons between open resection and LITT for treating SEGA.

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An online calculator using machine learning for predicting survival in pediatric patients with medulloblastoma

Cathleen C. Kuo, Andre Monteiro, Jaims Lim, Nolan J. Brown, Matthew J. Recker, Moleca M. Ghannam, Julian L. Gendreau, Veetai Li, and Renée M. Reynolds

OBJECTIVE

Medulloblastoma is the most common malignant intracranial tumor affecting the pediatric population. Despite advancements in multimodal treatment over the past 2 decades yielding a 5-year survival rate > 75%, children who survive often have substantial neurological and cognitive sequelae. The authors aimed to identify risk factors and develop a clinically friendly online calculator for prognostic estimation in pediatric patients with medulloblastoma.

METHODS

Pediatric patients with a histopathologically confirmed medulloblastoma were extracted from the Surveillance, Epidemiology, and End Results database (2000–2018) and split into training and validation cohorts in an 80:20 ratio. The Cox proportional hazards model was used to identify the univariate and multivariate survival predictors. Subsequently, a calculator with those factors was developed to predict 2-, 5-, and 10-year overall survival as well as median survival months for pediatric patients with medulloblastoma. The performance of the calculator was determined by discrimination and calibration.

RESULTS

One thousand seven hundred fifty-nine pediatric patients with medulloblastoma met the prespecified inclusion criteria. Age, sex, race, ethnicity, median household income, county attribute, laterality, anatomical location, tumor grade, tumor size, surgery status, radiotherapy, and chemotherapy were variables included in the calculator (https://spine.shinyapps.io/Peds_medullo/). The concordance index was 0.769 in the training cohort and 0.755 in the validation cohort, denoting clinically useful predictive accuracy. Good agreement between the predicted and observed outcomes was demonstrated by the calibration plots.

CONCLUSIONS

An easy-to-use prognostic calculator for a large cohort of pediatric patients with medulloblastoma was established. Future efforts should focus on improving granularity of population-based registries and externally validating the proposed calculator.

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Concurrent impact of de novo mutations on cranial and cortical development in nonsyndromic craniosynostosis

Emre Kiziltug, Phan Q. Duy, Garrett Allington, Andrew T. Timberlake, Riki Kawaguchi, Aaron S. Long, Mariana N. Almeida, Michael L. DiLuna, Seth L. Alper, Michael Alperovich, Daniel H. Geschwind, and Kristopher T. Kahle

OBJECTIVE

Nonsyndromic craniosynostosis (nsCS), characterized by premature cranial suture fusion, is considered a primary skull disorder in which impact on neurodevelopment, if present, results from the mechanical hindrance of brain growth. Despite surgical repair of the cranial defect, neurocognitive deficits persist in nearly half of affected children. Therefore, the authors performed a functional genomics analysis of nsCS to determine when, where, and in what cell types nsCS-associated genes converge during development.

METHODS

The authors integrated whole-exome sequencing data from 291 nsCS proband-parent trios with 29,803 single-cell transcriptomes of the prenatal and postnatal neurocranial complex to inform when, where, and in what cell types nsCS-mutated genes might exert their pathophysiological effects.

RESULTS

The authors found that nsCS-mutated genes converged in cranial osteoprogenitors and pial fibroblasts and their transcriptional networks that regulate both skull ossification and cerebral neurogenesis. Nonsyndromic CS–mutated genes also converged in inhibitory neurons and gene coexpression modules that overlapped with autism and other developmental disorders. Ligand-receptor cell-cell communication analysis uncovered crosstalk between suture osteoblasts and neurons via the nsCS-associated BMP, FGF, and noncanonical WNT signaling pathways.

CONCLUSIONS

These data implicate a concurrent impact of nsCS-associated de novo mutations on cranial morphogenesis and cortical development via cell- and non–cell-autonomous mechanisms in a developmental nexus of fetal osteoblasts, pial fibroblasts, and neurons. These results suggest that neurodevelopmental outcomes in nsCS patients may be driven more by mutational status than surgical technique.

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Cervical saccular limited dorsal myeloschisis, so-called "cervical myelomeningocele": long-term follow-up of a single-center series and systematic review

Seyed Farzad Maroufi, Mohammad Amin Dabbagh Ohadi, Iman Hosseinnejad, Keyvan Tayebi Meybodi, Alireza Takzare, Bahar Ashjaei, Farideh Nejat, and Zohreh Habibi

OBJECTIVE

Cervical saccular limited dorsal myeloschisis (LDM), previously so-called "cervical myelomeningocele," is a rare spinal dysraphism. Although the pathogenesis of true myelomeningocele is primary neurulation failure, LDM results from a delayed abnormality during the final stages of neurulation. The aim of the study was to evaluate the outcome of these patients and to assess the correlation of outcomes with the level and type of lesion. Also, pooled data from the literature on similar lesions were systematically reviewed.

METHODS

A retrospective study was conducted at Children’s Medical Center (CMC), Tehran, Iran. Information of patients who underwent surgery between 2004 and 2020 (i.e., the recent series) was extracted and combined with data from a previously published series from the same center that were obtained between 2000 and 2003 (CMC series). The literature was reviewed for all published cases, to be combined with the CMC series for further analyses.

RESULTS

Twenty-two patients were included in the recent series. Combined with 16 previously published cases, 38 patients with a mean ± SD age at surgery of 11.75 ± 28.64 months were included in the CMC series. The rates of neurological deficit, hydrocephalus, and Chiari malformation type II in the CMC series were 26.32%, 39.47%, and 28.95%, respectively. The lesions were at the upper levels in 17 (44.7%) and lower cervical levels in 21 (55.3%) patients, with 31 cases (81.58%) diagnosed with stalk-type lesions and 7 cases (18.42%) with myelocystocele-type lesions. At final follow-up, 31 patients (81.57%) achieved sphincter continence, and all 36 accessible patients were ambulated, consisting of 28 (73.68%) independent and 8 (21.05%) dependent ambulation patients. The rates of Chiari malformation type II and hydrocephalus were insignificantly higher in patients with upper-level lesions, but those of neurological deficit, ambulation, and sphincter continence were not associated with level. The rates of hydrocephalus (p < 0.01), Chiari type II malformation (p < 0.01), and neurological deficit (p = 0.04) were significantly higher in the myelocystocele group. In the systematic review, 24.77% of patients had neurological deficit. Binary logistic regression showed that older age at surgery (p = 0.03) and associated spinal anomalies (p = 0.04) were significant predictors of deficits. Chiari type II malformation was significantly (p < 0.001) and hydrocephalus was marginally (p = 0.06) more common in patients with myelocystocele-type lesions. The rate of Chiari malformation type II was higher in patients with upper-level lesions (p = 0.02).

CONCLUSIONS

Patients with cervical saccular LDM had better outcome compared with those patients with true myelomeningocele in more distal areas. According to the current series, most patients obtained ambulation and voiding continence, regardless of the level or type of lesion. Hydrocephalus, Chiari type II malformation, and neurological deficit were more common in patients with myelocystocele-type lesions.

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Association of cerebrospinal fluid parameters with treatment and complications among children with cerebrospinal fluid shunt infections: a multicenter study

Michael T. Hawkes, Alastair McAlpine, Michelle Barton, Adrianna Ranger, Archana Balamohan, H. Dele Davies, Gwenn Skar, Marie-Astrid Lefebvre, Ahmed Almadani, Dolores Freire, Nicole Le Saux, Jennifer Bowes, Jocelyn A. Srigley, Patrick Passarelli, John Bradley, Sarah Khan, Rupeena Purewal, Isabelle Viel-Thériault, Joan L. Robinson, and for the Paediatric Investigators Collaborative Network on Infections in Canada (PICNIC)

OBJECTIVE

Cerebrospinal fluid (CSF) white blood cell (WBC) count, neutrophil percentage, protein concentration, and glucose level are typically measured at diagnosis and serially during the treatment of CSF shunt infections. The objective of this retrospective cohort study was to describe the longitudinal profile of CSF parameters in children with CSF shunt infections and assess their association with treatment and outcome.

METHODS

Participants were children treated at 11 tertiary pediatric hospitals in Canada and the United States for CSF shunt infection, from July 1, 2013, through June 30, 2019, with hardware removal, external ventricular drain placement, intravenous antibiotics, and subsequent permanent shunt reinsertion. The relationship between CSF parameters and a complicated course (a composite outcome representing children with at least one of the following: contiguous soft-tissue infection, worsening hydrocephalus, CSF leak, intracranial bleed, brain abscess, venous thrombosis, reinfection after insertion of the new shunt, other complication, ICU admission, or death) was analyzed.

RESULTS

A total of 109 children (median age 2.8 years, 44% female) were included in this study. CSF pleocytosis, elevated protein, and hypoglycorrhachia had sensitivities of 69%, 47%, and 38% for the diagnosis of culture-confirmed CSF shunt infection, respectively. The longitudinal profile of the neutrophil percentage followed a monotonic trend, decreasing by 1.5% (95% CI 1.0%–2.0%, p < 0.0001) per day over the course of treatment. The initial WBC count differed significantly between pathogens (p = 0.011), but the proportion of neutrophils, protein concentration, and glucose level did not, and was lowest with Cutibacterium acnes. The duration of antibiotic treatment and the time to shunt reinsertion were longer in patients with a higher initial neutrophil percentage. Fifty-eight patients (53%) had one or more complications during their admission. A neutrophil percentage > 44% (Youden index) in the initial CSF sample was associated with a 1.8-fold (95% CI 1.2- to 2.8-fold) higher relative risk of a complicated course. In a random-intercept, random-slope linear mixed-effects model, the longitudinal neutrophil trajectory differed significantly between patients with and without complications (p = 0.030).

CONCLUSIONS

A higher proportion of neutrophils in the CSF at diagnosis was associated with a complicated clinical course. Other CSF parameters were associated with treatment and outcome; however, wide variability in values may limit their clinical utility.

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Clinical outcomes of MR-guided laser interstitial thermal therapy corpus callosum ablation in drug-resistant epilepsy: a systematic review and meta-analysis

Jasmine L. Hect, Emily Harford, Seyed Farzad Maroufi, Mary Lou Klem, Alireza Mansouri, and Taylor J. Abel

OBJECTIVE

The goal of this systematic review and meta-analysis was to provide an updated analysis of studies investigating outcomes, morbidity, and mortality associated with MR-guided laser interstitial thermal therapy (MRgLITT) corpus callosum ablation (CCA).

METHODS

Study inclusion criteria for screening required that studies report on human subjects only, including patients aged 1–52 years diagnosed with drug-resistant epilepsy who underwent CCA. Sixteen articles published between 2016 and 2023 were included for the systematic review and analysis, including 4 case reports, 11 case series, and 1 case-control study. Altogether, 85 pediatric and adult patients undergoing CCA were included in the systematic review (46 patients younger and 39 patients older than 21 years). The main outcome of seizure freedom was measured using the decrease in the frequency of atonic seizures following surgery, percentage of atonic seizure freedom following surgery, and percentage of overall seizure freedom following surgery. These measurements were made using data from the last follow-up for patients with at least 6 months of follow-up post-CCA.

RESULTS

The extent of CCA differed across the pooled cohorts, including anterior two-thirds CCA (38.89%, n = 35) and posterior one-third CCA for completion of a prior partial CCA (22.22%, n = 20), complete CCA (27.78%, n = 25), or CCA of residual white matter in the case of subtotal initial ablation (5.56%, n = 5). Overall, 12.94% of the patients undergoing CCA experienced operational complications. The most common operative complications across 90 CCA operations were probe malpositioning (n = 6), hemorrhage (n = 5), off-target extension of splenium ablation to the thalamus (n = 1), infection (n = 1), and postoperative CSF leak (n = 1). Neurological deficits following CCA were reported as transient in 18.82% and permanent in 4.71% of patients across all studies. The most common neurological deficits were disconnection syndrome (n = 4) or transient hemiplegia (supplementary motor area–like syndrome; n = 4). The 6-month overall seizure freedom rate was 18.87% of 53 patients, and the atonic seizure freedom rate was 46.28% of 52 patients postoperatively. CCA resulted in an average decrease in atonic seizure rate from 8.30 to 1.65 atonic seizures per day (average decrease 80.12%).

CONCLUSIONS

CCA is associated with an acceptable complication profile, and most patients experience a meaningful reduction in target seizure semiologies. Accurate MRgLITT probe placement is likely important for maximizing CCA while avoiding collateral damage. Avoidable complications of CCA include off-target ablation (and associated deficits), hemorrhage, and future surgery for residual CCA to palliate continued seizures.

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Effect of thin-split encephalomyosynangiosis on transient neurological events in revascularization surgery for pediatric patients with moyamoya disease

Kazuki Ishii, Fumiaki Kanamori, Yoshio Araki, Kenji Uda, Kinya Yokoyama, Takashi Mamiya, Kai Takayanagi, Shunsaku Goto, Masahiro Nishihori, Takashi Izumi, and Ryuta Saito

OBJECTIVE

Transient neurological events (TNEs) are among the most important events after revascularization surgery in pediatric patients with moyamoya disease (MMD). Although hemodynamic changes and crying are representative factors of TNEs, brain compression by encephalomyosynangiosis (EMS) is another important cause of TNEs. Therefore, the authors assumed that making the EMS as thin as possible reduces the frequency of TNEs. However, thin-split EMS can lead to insufficient development of collateral vessels. This study aimed to evaluate the effects of thin-split EMS in combined revascularization surgery on postoperative outcomes in pediatric patients with MMD.

METHODS

The authors retrospectively included 56 consecutive combined revascularization surgeries in the anterior cerebral circulation in pediatric patients with MMD. These surgeries were classified into the former group and thin-split EMS group. The temporal muscle was halved in the former EMS group and split as thinly as possible in the thin-split EMS group. The authors performed between-group comparisons of postoperative stroke events and TNEs during the acute period and the development of collateral flow and stroke events during the chronic period.

RESULTS

Former and thin-split EMS procedures were performed in 37 and 19 patients, respectively. TNEs without crying or hemodynamic changes were observed significantly less frequently in the thin-split EMS group than in the former EMS group (0 [0.0%] in the thin-split EMS group vs 9 [24.3%] in the former EMS group, p = 0.021). There were no significant between-group differences in the development of indirect bypass during the chronic period (good: 34 [91.9%] and poor: 3 [8.1%] in the former EMS group vs good: 16 [84.2%] and poor: 3 [15.8%] in the thin-split EMS group; p = 0.397). Additionally, there were no significant between-group differences in the incidence of chronic clinical events, including death or stroke.

CONCLUSIONS

Thin-split EMS can reduce TNEs that do not accompany crying or hemodynamic changes. Furthermore, the procedure has sufficient prevention effects against stroke events during the chronic postoperative period in pediatric patients with MMD.

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Letter to the Editor. Intraparenchymal stent for the treatment of hydrocephalus

Fardad T. Afshari

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Machine learning models for predicting seizure outcome after MR-guided laser interstitial thermal therapy in children

Omar Yossofzai, Scellig S. D. Stone, Joseph R. Madsen, Shelly Wang, John Ragheb, Ismail Mohamed, Robert J. Bollo, Dave Clarke, M. Scott Perry, Alexander G. Weil, Jeffrey S. Raskin, Jonathan Pindrik, Raheel Ahmed, Sandi K. Lam, Aria Fallah, Cassia Maniquis, Andrea Andrade, George M. Ibrahim, James Drake, James T. Rutka, Jignesh Tailor, Nicholas Mitsakakis, and Elysa Widjaja

OBJECTIVE

MR-guided laser interstitial thermal therapy (MRgLITT) is associated with lower seizure-free outcome but better safety profile compared to open surgery. However, the predictors of seizure freedom following MRgLITT remain uncertain. This study aimed to use machine learning to predict seizure-free outcome following MRgLITT and to identify important predictors of seizure freedom in children with drug-resistant epilepsy.

METHODS

This multicenter study included children treated with MRgLITT for drug-resistant epilepsy at 13 epilepsy centers. The authors used clinical data, diagnostic investigations, and ablation features to predict seizure-free outcome at 1 year post-MRgLITT. Patients from 12 centers formed the training cohort, and patients in the remaining center formed the testing cohort. Five machine learning algorithms were developed on the training data by using 10-fold cross-validation, and model performance was measured on the testing cohort. The models were developed and tested on the complete feature set. Subsequently, 3 feature selection methods were used to identify important predictors. The authors then assessed performance of the parsimonious models based on these important variables.

RESULTS

This study included 268 patients who underwent MRgLITT, of whom 44.4% had achieved seizure freedom at 1 year post-MRgLITT. A gradient-boosting machine algorithm using the complete feature set yielded the highest area under the curve (AUC) on the testing set (AUC 0.67 [95% CI 0.50–0.82], sensitivity 0.71 [95% CI 0.47–0.88], and specificity 0.66 [95% CI 0.50–0.81]). Logistic regression, random forest, support vector machine, and neural network yielded lower AUCs (0.58–0.63) compared to the gradient-boosting machine but the findings were not statistically significant (all p > 0.05). The 3 feature selection methods identified video-EEG concordance, lesion size, preoperative seizure frequency, and number of antiseizure medications as good prognostic features for predicting seizure freedom. The parsimonious models based on important features identified by univariate feature selection slightly improved model performance compared to the complete feature set.

CONCLUSIONS

Understanding the predictors of seizure freedom after MRgLITT will assist with prognostication.

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Combined endovascular and skull base surgical management of pediatric craniocervical pathology: a case series

Jonathan P. Scoville, Matthew C. Findlay, Evan Joyce, Nikita Alexiades, Elena Kurudza, Philipp Taussky, and Douglas L. Brockmeyer

OBJECTIVE

Pathological bony abnormalities of the craniocervical region in children sometimes require surgical intervention as part of their management. Rarely, abnormal skeletal or vascular anatomy can render traditional surgical techniques ineffective because of the risk of injury to the vertebral artery. To mitigate these risks, a combined endovascular and skull base approach was devised. The authors describe their experience using vertebral artery sacrifice as an adjunctive surgical method to reduce the risk of inadvertent vertebral artery injury during surgical correction of pediatric craniocervical deformity.

METHODS

Three patients underwent vertebral artery sacrifice for structural craniocervical pathologies (1 male, 2 females; ages 12, 14, and 3 years). One patient presented with basilar invagination odontogenic brainstem compression, and the other 2 patients presented with congenital cervical fusion. All patients underwent endovascular left vertebral artery sacrifice after passing balloon test occlusion.

RESULTS

No adverse effects from the vertebral artery sacrifice were observed. At the last follow-ups (35, 30, and 32 months), all 3 patients had a satisfactory outcome with no adverse effects as a result of their sacrificed artery.

CONCLUSIONS

Endovascular vertebral artery sacrifice followed by skull base approaches can be used to effectively and safely treat craniocervical pathology from a variety of pediatric skeletal abnormalities.