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Open access

Tyler Lazaro, Viren Vasandani, Ariadna Robledo, Nisha Gadgil, and Peter Kan

A 47-year-old female with a history of a ruptured left posterior inferior cerebellar artery (PICA) aneurysm, status post coil embolization and retreatment for recurrence, presented with evidence of a recurrent dissecting PICA aneurysm. Given that these aneurysms are considered high risk and have a greater propensity for rupture than anterior circulation aneurysms, retreatment was recommended. With the patient’s strong preference for endovascular therapy, flow diversion with a Silk Vista Baby was performed. Given the low-profile design of the device, a radial artery approach and coaxial technique were used to deploy the flow diverter. The device was successfully placed, with complete obliteration of the aneurysm after 1 year.

The video can be found here: https://stream.cadmore.media/r10.3171/2022.7.FOCVID2247

Free access

Ozge Kapar, Zahide Mail Gurkan, Muge Dolgun, Altay Sencer, Candan Gürses, and Bilge Bilgic

OBJECTIVE

In the histopathological examination of treatment-resistant epilepsy, focal cortical dysplasia (FCD) is the most common diagnosis in the pediatric group. FCD is classified histopathologically according to the International League Against Epilepsy (ILAE) classification. In the last decade since the ILAE classification has been released, molecular genetic studies have revealed mTOR pathway–related mutations as a major etiology. The objective of this study was to determine the incidence of FCD in treatment-resistant epilepsy patients, explore histomorphological and immunohistochemical features, examine clinicopathological correlation, demonstrate mTOR pathway activation using a pS6 antibody immunohistochemically, and try to introduce a candidate for possible targeted therapies.

METHODS

Paraffin blocks and slides of tissue from patients with treatment-resistant epilepsy were reexamined retrospectively. Histopathological subtypes of FCD were determined according to the ILAE classification. NeuN and neurofilament H (NF-H) staining were performed, and additionally a pS6 antibody was used to demonstrate mTOR pathway activation.

RESULTS

In 32 cases diagnosed with FCD, or 17.5% of 183 surgical epilepsy materials, there were no significant differences in the statistical analysis of clinical variables between the ILAE FCD subtypes. Recommended antibody NeuN revealed microcolumnar alignment in the FCD type Ia and IIIa groups and the loss of lamination in the type Ib group. Another recommended antibody, NF-H, was not found to be useful in discriminating between normal and dysmorphic neurons. pS6 expression, showing mTOR pathway activation, was observed in dysmorphic neurons and balloon cells in all FCD type II cases.

CONCLUSIONS

Significant pS6 expression in FCD type II represents the genomic nature of the disease noted in the literature. Nevertheless, the known MTOR gene and mTOR pathway–related mutations remain behind proportionally to explain the mTOR pathway activation in all FCD type II cases. Clinicopathologically and genetically integrated classification and usage of mTOR pathway inhibitors in treatment are expected as a recent evolution.

Open access

Steven B. Housley, Justin M. Cappuzzo, Muhammad Waqas, Andre Monteiro, Elad I. Levy, and Adnan H. Siddiqui

Treatment of wide-necked posterior communicating artery (PCoA) aneurysms is extremely challenging, especially in fetal posterior cerebral artery (PCA) configurations. This technical video demonstrates the nuances of an innovative use of flow diversion to treat a recurrent wide-necked PCoA aneurysm. This middle-aged patient presented with recurrence of a previously ruptured, coil-embolized PCoA aneurysm. Initial attempts at Comaneci-assisted coiling were unsuccessful because the coil herniated into the middle cerebral artery (MCA). Therefore, a low-profile visualized intraluminal support (LVIS) was placed in the fetal PCA across the aneurysm ostium and a flow diverter was placed in the internal carotid artery and MCA to constitute a Y-construct.

The video can be found here: https://stream.cadmore.media/r10.3171/2022.7.FOCVID2262

Open access

Peter T. Kan, Elad I. Levy, Felipe C. Albuquerque, and Mandy Jo Binning

Free access

P. David Adelson, George M. Ibrahim, Erin N. Kiehna, and Chima Oluigbo

Free access

Arad Iranmehr, Mohammad Amin Dabbagh Ohadi, Mohammadreza Chavoshi, Amin Jahanbakhshi, and Konstantin V. Slavin

OBJECTIVE

Hypothalamic hamartoma (HH) is a rare, nonmalignant, heterotopic developmental malformation that consists of a mixture of normal neurons and glial cells. Resection of HHs has been associated with high rates of mortality and morbidity. Therefore, minimally invasive ablation methods could be the best treatment option for HH. The most frequently used minimally invasive options for HH ablation are radiofrequency thermocoagulation (RFT), laser ablation (LA), and stereotactic radiosurgery.

METHODS

To investigate three minimally invasive procedures in the treatment of refractory seizures related to HH, the authors conducted a systematic search in March 2022 in the MEDLINE, Embase, Scopus, and Web of Science databases in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Seizure freedom was the primary outcome of interest. The authors defined seizure freedom as Engel class I or International League Against Epilepsy class 1 or 2 or as the reported term “seizure freedom.” The secondary outcome was long-term complications reported in studies. Both random- and fixed-effects models were used to calculate the pooled proportion of seizure freedom and complication rate with 95% confidence intervals. A modified version of the Joanna Briggs Institute (JBI) Critical Appraisal to assess the risk of bias was used.

RESULTS

The authors included 15 studies with 422 patients (RFT, n = 190; LA, n = 171; and Gamma Knife Radiosurgery [GKRS], n = 61). Generally, the mean incidences of overall seizure freedom after minimally invasive procedures were 77% (95% CI 0.74–0.81) and 68% (95% CI 0.57–0.79) using fixed- and random-effects models, respectively. The mean incidence of overall seizure freedom after RFT was 69% (95% CI 0.63–0.75), and the mean incidences of overall seizure freedom after LA and GKRS were 87% (95% CI 0.82–0.92) and 44% (95% CI 0.32–0.57), respectively. The total complication rate with minimally invasive procedures was 13% (95% CI 0.01–0.26). The complication rate in each treatment was as follows: 5% (95% CI 0.0–0.12) for RFT, 20% (95% CI 0.0–0.47) for LA, and 22% (95% CI 0–0.65) for GKRS. Meta-regression analysis showed an association between older age and higher complication rates in the LA group.

CONCLUSIONS

In this meta-analysis, LA showed superiority in seizure freedom over the other two methods. The complication rate associated with RFT was less than those in the other two methods; however, this difference was not statistically significant.

Free access

Jun Wang, Xinyi Luo, Chenghan Chen, Jiahong Deng, Hao Long, Kaijun Yang, and Songtao Qi

OBJECTIVE

In this systematic review the authors aimed to evaluate the effectiveness and superiority of radiomics in detecting tiny epilepsy lesions and to conduct original research in the use of radiomics for preliminary prediction of postoperative seizures in patients with dysembryoplastic neuroepithelial tumor (DNET).

METHODS

The PubMed and Web of Science databases were searched from the earliest record, January 1, 2018, to December 29, 2021, for reports of the detection of epilepsy using radiomics, and the resulting articles were carefully checked according to the PRISMA 2020 guidelines. The authors then conducted original research by evaluating MR images in 18 patients, who were then separated into two groups, the epilepsy recurrence group (ERG) and the epilepsy nonrecurrence group. The tumor region and the edema region were segmented manually by 3D Slicer. The radiomics data were extracted from MR images by using “Slicer Radiomics” running on Mac OS X. Tumor regions were observed with T1-weighted imaging, and edema with FLAIR imaging. Radiomics features with significant differences were selected through comparison according to epilepsy relapses performed with the Mann-Whitney U-test. The edema and tumor regions were also compared within groups to identify their distinctive features. Radiomics features were tested to verify their ability to predict recurrence epilepsy by receiver operating characteristic curve.

RESULTS

This systematic review located 9 original articles related to epilepsy and radiomics published from 2018 to 2021. The reported studies demonstrated that radiomics is useful for detecting tiny epilepsy lesions. Among the radiomics features used, the predictive ability of the area under the curve was more than 0.8. The heterogeneity of the peritumoral edema region was found to be higher in the ERG.

CONCLUSIONS

Satellite lesions in the peritumoral edema region of DNET patients may cause epilepsy recurrence, and radiomics is an emerging method to detect and evaluate these epilepsy-associated lesions.

Free access

Sandeep Kandregula, Danielle Terrell, Robbie Beyl, Anne Freelin, Bharat Guthikonda, Christina Notarianni, and Jamie Toms

OBJECTIVE

Racial and ethnic disparities in healthcare have gained significant importance since the Institute of Medicine published its report on disparities in healthcare. There is a lack of evidence on how race and ethnicity affect access to advanced treatment of pediatric medically intractable epilepsy. In this context, the authors analyzed the latest Kids’ Inpatient Database (KID) for racial/ethnic disparities in access to surgical treatment of epilepsy.

METHODS

The authors queried the KID for the years 2016 and 2019 for the diagnosis of medically intractable epilepsy.

RESULTS

A total of 29,292 patients were included in the sample. Of these patients, 8.9% (n = 2610) underwent surgical treatment/invasive monitoring. The mean ages in the surgical treatment and nonsurgical treatment groups were 11.73 years (SD 5.75 years) and 9.5 years (SD 6.16 years), respectively. The most common insurance in the surgical group was private/commercial (55.9%) and Medicaid in the nonsurgical group (47.7%) (p < 0.001). White patients accounted for the most common population in both groups, followed by Hispanic patients. African American patients made up 7.9% in the surgical treatment group compared with 12.9% in the nonsurgical group. African American (41.1%) and Hispanic (29.9%) patients had higher rates of emergency department (ED) utilization compared with the White population (24.6%). After adjusting for all covariates, the odds of surgical treatment increased with increasing age (OR 1.06, 95% CI 1.053–1.067; p < 0.001). African American race (OR 0.513, 95% CI 0.443–0.605; p < 0.001), Hispanic ethnicity (OR 0.681, 95% CI 0.612–0.758; p < 0.001), and other races (OR 0.789, 95% CI 0.689–0.903; p = 0.006) had lower surgical treatment odds compared with the White population. Medicaid/Medicare was associated with lower surgical treatment odds than private/commercial insurance (OR 0.603, 0.554–0.657; p < 0.001). Interaction analysis revealed that African American (OR 0.708, 95% CI 0.569–0.880; p = 0.001) and Hispanic (OR 0.671, 95% CI 0.556–0.809; p < 0.001) populations with private insurance had lower surgical treatment odds than White populations with private insurance. Similarly, African American patients, Hispanic patients, and patients of other races with nonprivate insurance also had lower surgical treatment odds than their White counterparts after adjusting for all other covariates.

CONCLUSIONS

Based on the KID, African American and Hispanic populations had lower surgical treatment rates than their White counterparts, with higher utilization of the ED for pediatric medically intractable epilepsy.

Free access

Kendall Curtis, Jasmine L. Hect, Emily Harford, William P. Welch, and Taylor J. Abel

OBJECTIVE

Responsive neurostimulation (RNS) is a promising treatment for pediatric patients with drug-resistant epilepsy for whom resective surgery is not an option. The relative indications and risk for pediatric patients undergoing RNS therapy require further investigation. Here, the authors report their experience with RNS implantation and therapy in pediatric patients.

METHODS

The authors performed a retrospective chart review to identify patients implanted with RNS depth or strip electrodes for the treatment of drug-resistant epilepsy at their institution between 2020 and 2022. Patient demographics, surgical variables, and patient seizure outcomes (Engel class and International League Against Epilepsy [ILAE] reporting) were evaluated.

RESULTS

The authors identified 20 pediatric patients ranging in age from 8 to 21 years (mean 15 [SD 4] years), who underwent RNS implantation, including depth electrodes (n = 15), strip electrodes (n = 2), or both (n = 3). Patient seizure semiology, onset, and implantation strategy were heterogeneous, including bilateral centromedian nucleus (n = 5), mesial temporal lobe (n = 4), motor cortex or supplementary motor area (n = 7), or within an extratemporal epileptogenic zone (n = 4). There were no acute complications of RNS implantation (hemorrhage or stroke) or device malfunctions. One patient required rehospitalization for postoperative infection. At the longest follow-up (mean 10 [SD 7] months), 13% patients had Engel class IIB, 38% had Engel class IIIA, 6% had Engel class IIIB, 19% had Engel class IVA, 19% had Engel class IVB, and 6% had Engel class IVC outcomes. Using ILAE metrics, 6% were ILAE class 3, 25% were ILAE class 4, and 69% were ILAE class 5.

CONCLUSIONS

This case series supports current literature suggesting that RNS is a safe and potentially effective surgical intervention for pediatric patients with drug-resistant epilepsy. The authors report comparable rates of serious adverse events to current RNS literature in pediatric and adult populations. Seizure outcomes may continue to improve with follow-up as stimulation strategy is refined and the chronic neuromodulatory effect evolves, as previously described in patients with RNS. Further large-scale, multicenter case series of RNS in pediatric patients with drug-resistant epilepsy are required to determine long-term pediatric safety and effectiveness.

Free access

Nicole Falls, Jorge I. Arango, and P. David Adelson

OBJECTIVE

Medically refractory epilepsy remains a therapeutic challenge when resective surgery is not a practical option and indirect neurostimulation efficacy may be limited. In these instances responsive neurostimulation (RNS) has been used in adults, with good outcomes in most patients. However, the utility of RNS in children and young adults has not been systematically explored. In this study, the authors present a single institution’s experience with RNS in pediatric patients.

METHODS

A single-center retrospective chart review of patients who underwent RNS implantation at Phoenix Children’s Hospital during the 4-year period between January 2018 and December 2021 was performed.

RESULTS

Following evaluation for epilepsy surgery, 22 patients underwent RNS implantation using different anatomical targets depending on the predetermined epileptic focus/network. In the cohort, 59% of patients were male, the mean age at implantation was 16.4 years (range 6–22 years), and the mean follow-up time was 2.7 years (range 1.0–4.3 years). All patients had a preoperative noninvasive evaluation that included MRI, video-electroencephalography, and resting-state functional MRI. Additionally, 13 patients underwent invasive monitoring with stereo-electroencephalography to help determine RNS targets. All patients had variable positive responses with reduction of seizure frequency and/or intensity. Overall, seizure frequency reduction of > 50% was seen in the majority (86%) of patients. There were two complications: one patient experienced transitory weakness and one generator failed, requiring replacement. A patient died of sudden unexpected death in epilepsy 3 years after implantation despite being seizure free during the previous year.

CONCLUSIONS

RNS used in children with medically refractory epilepsy improved seizure control after implantation, with decreases in seizure frequency > 50% from preoperative baseline in the majority of patients. Preliminary findings indicate that functional MRI and stereo-electroencephalography were helpful for RNS targeting and that RNS can be used safely even in young children.