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Bilateral pallidotomy for acquired or heredodegenerative generalized dystonia in children

Kanwaljeet Garg, Manmohan Singh, Raghu Samala, Roopa Rajan, Sheffali Gulati, and Vinay Goyal

OBJECTIVE

Dystonias are relatively rare disorders characterized by sustained or intermittent muscle contractions causing abnormal movements or postures. Generalized dystonia is a therapeutic challenge because medications are unable to control dystonia adequately in most patients. These patients may be candidates for surgical therapy. The commonly used surgical procedures in these patients are pallidotomy and deep brain stimulation. Limited studies are available on the role of pallidotomy in children with acquired/heredodegenerative generalized dystonia. The objective of this study was to describe the authors’ experience with bilateral pallidotomy in this group of patients.

METHODS

The authors retrospectively reviewed all pediatric patients (less than 18 years of age) with acquired/heredodegenerative generalized dystonia who underwent bilateral simultaneous pallidotomy at their center between January 2014 and January 2021. Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) scores before and after surgery were recorded. Complications arising after the procedure were recorded as well.

RESULTS

Ten patients (8 male and 2 female) with a mean (range) age of 11.1 (5–17) years were included in this study. The mean duration between disease onset and surgical intervention was 3.9 years. Two patients presented in status dystonicus. The mean ± SD (range) preoperative BFMDRS score of the patients without status dystonicus (n = 8) was 80 ± 18.9 (59.5–108). The mean ± SD BFMDRS score at the time of discharge from the hospital after surgery was 58.8 ± 37.9. Three patients had more than 20% change in BFMDRS score at the time of discharge from the hospital. The mean improvement was 25.5% at the end of 1 year. Of 5 surviving patients in the non–status dystonicus group, 3 patients had more than 40% change in BFMDRS score while the other 2 patients developed recurrence at the last follow-up (4.5 years). Status dystonicus abated after bilateral pallidotomy in both patients. Permanent bulbar complications were seen in 2 patients.

CONCLUSIONS

Bilateral pallidotomy may result in clinically significant improvement in children with acquired/heredodegenerative generalized dystonia, although the benefits should be closely weighed against the risk of irreversible bulbar dysfunction. It is a viable option for children in resource-limited settings.

In Neurosurgical Focus Volume 53 (2022): Issue 4 (Oct 2022): Pediatric Functional Disease

Open access

Challenging access during flow diversion treatment of a giant cavernous ICA aneurysm

Visish M. Srinivasan, Joelle N. Hartke, Joshua S. Catapano, Ethan A. Winkler, Ashutosh P. Jadhav, Felipe C. Albuquerque, and Andrew F. Ducruet

A man in his 60s presented with severe ophthalmoparesis and loss of visual acuity in his right eye. He was found to have a giant aneurysm of the cavernous internal carotid artery (ICA). Treatment with a flow diverter was recommended. The aneurysm caused matricidal outflow restriction of the ICA. Microwire and microcatheter access through the aneurysm was challenging, requiring multiple wires, stentriever reduction, and more. Eventually, a construct of 3 Pipeline embolization devices was created across the aneurysm. Troubleshooting access across giant aneurysms is an important part of treatment. Informed consent was obtained for the procedure and for publication.

The video can be found here: https://stream.cadmore.media/r10.3171/2022.7.FOCVID2258

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms

Open access

Combined deconstructive and reconstructive treatment of a giant vertebrobasilar fenestration aneurysm

Lorenzo Rinaldo, Soliman Oushy, and Giuseppe Lanzino

Aneurysms associated with a vertebrobasilar fenestration are rare lesions and can grow to a giant size, presenting significant therapeutic challenges. Endovascular treatment of these aneurysms has traditionally been with coiling; however, flow diverter placement within the fenestration arms has recently proven to be a viable treatment strategy. The authors present a case of a giant vertebrobasilar fenestration aneurysm in a patient presenting with a cranial nerve VI palsy. The lesion was treated by using a combination of flow diverter placement and vertebral artery sacrifice. The nuances of flow diversion therapy for these aneurysms and the management of intra- and postoperative complications are discussed.

The video can be found here: https://stream.cadmore.media/r10.3171/2022.7.FOCVID2256

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms

Free access

Comparison of outcomes after stereoelectroencephalography and subdural grid monitoring in pediatric tuberous sclerosis complex

Thomas Larrew, Jesse Skoch, S. Katie Z. Ihnen, Ravindra Arya, Katherine D. Holland, Jeffrey R. Tenney, Paul S. Horn, James L. Leach, Darcy A. Krueger, Hansel M. Greiner, and Francesco T. Mangano

OBJECTIVE

Patients with tuberous sclerosis complex (TSC) epilepsy present with unique clinical challenges such as early seizure onset and high rates of intractability and multifocality. Although there are numerous studies about the safety and efficacy of stereoelectroencephalography (SEEG), this topic has not been studied in TSC patients who have distinct epilepsy profiles. The authors investigated subdural grid (SDG) and SEEG monitoring to determine whether these procedures lead to similar seizure and safety outcomes and to identify features unique to this pediatric population.

METHODS

TSC patients who underwent SDG or SEEG placement and a second epilepsy surgery during the period from 2007 to 2021 were included in this single-center retrospective cohort analysis. Various patient, hospitalization, and epilepsy characteristics were collected.

RESULTS

A total of 50 TSC patients were included in this study: 30 were included in the SDG cohort and 20 in the SEEG cohort. Baseline weekly seizure count did not significantly differ between the 2 groups (p = 0.412). The SEEG group had a greater mean baseline number of antiepileptic drugs (AEDs) (3.0 vs 2.0, p = 0.003), higher rate of previous surgical interventions (25% vs 0%, p = 0.007), and larger proportion of patients who underwent bilateral monitoring (50% vs 13.3%, p = 0.005). Despite this, there was no significant difference in seizure freedom between the SDG and SEEG cohorts. The mean reduction in seizure count was 84.9% and 47.8% of patients were seizure free at last follow-up (mean 79.4 months). SEEG trended toward being a safer procedure than SDG monitoring, with a shorter mean ICU stay (0.7 days vs 3.9 days, p < 0.001), lower blood transfusion rate (0% vs 13.3%, p = 0.140), and lower surgical complication rate (0% vs 10%, p = 0.265).

CONCLUSIONS

In the comparison of the SDG and SEEG cohorts, the SEEG group included patients who appeared to receive more aggressive management and have a higher rate of multifocality, more prior surgical interventions, more AEDs at baseline, and a higher rate of bilateral invasive monitoring. Despite this, the SEEG cohort had similar seizure outcomes and a trend toward increased safety. Based on these findings, SEEG appears to allow for monitoring of a wider breadth of TSC patients given its minimally invasive nature and its relative simplicity for monitoring numerous regions of the brain.

In Neurosurgical Focus Volume 53 (2022): Issue 4 (Oct 2022): Pediatric Functional Disease

Free access

Deep brain stimulation in children and youth: perspectives of patients and caregivers gleaned through Twitter

Lior M. Elkaim, Farbod Niazi, Jordan J. Levett, Rakan Bokhari, Carolina Gorodetsky, Sara Breitbart, Fahad Alotaibi, Abdulelah A. Alluhaybi, Alexander G. Weil, Aria Fallah, Naif M. Alotaibi, and George M. Ibrahim

OBJECTIVE

This study aims to glean patient and caregiver perspectives surrounding deep brain stimulation (DBS) in children and youth through an analysis of patterns of social media usage.

METHODS

The authors performed a comprehensive search of the Twitter Application Programming Interface (API) database for all tweets about DBS use in children and youth, with no date restriction. Data pertaining to each tweet were extracted for analysis. Results were analyzed using qualitative and quantitative methodologies. These included thematic analysis of tweets, accounts, and descriptive statistics. Sentiment analysis of extracted tweets was also performed. A multivariable regression model was used to identify predictors of higher engagement metrics (likes, retweets, and quotes).

RESULTS

A comprehensive search of the Twitter database yielded 877 tweets from 816 unique accounts meeting study inclusion criteria. Most tweets were from patients or caregivers, researchers, or news media outlets. The most common themes among analyzed tweets were research discussing novel findings (45.2%) or personal experiences of patients or caregivers (27.4%). Sentiment analysis showed that 54.5% of tweets were positive, 35.1% were neutral, and 10.4% were negative. The presence of pictures or videos increased the tweet engagement count by an average of 10.5 (95% CI 7.3–13.6). Tweets about personal patient experiences (β = 6, 95% CI 0.95–12) and tweets tagging other accounts (β = 3.2, 95% CI 0.63–5.8) were also significantly associated with higher engagement metrics.

CONCLUSIONS

The current study is the first to assess patient and caregiver perspectives surrounding pediatric DBS through a comprehensive analysis of social media usage. Given the nascent field, social media presents an opportunity to share experiences and promote patient and healthcare professional education surrounding pediatric DBS.

In Neurosurgical Focus Volume 53 (2022): Issue 4 (Oct 2022): Pediatric Functional Disease

Open access

Flow diversion for cerebral aneurysms

Joseph A. Carnevale, Jacob L. Goldberg, Gary Kocharian, Andrew L. A. Garton, Alexander Ramos, Justin Schwarz, Srikanth Reddy Boddu, Y. Pierre Gobin, and Jared Knopman

The treatment of cerebral aneurysms includes open microsurgical options (e.g., clipping, trapping/bypass) and evolving endovascular techniques. Following the landmark trials that propelled endovascular treatment to the forefront, flow diversion has shown high aneurysm cure rates with minimal complications. Flow diversion stents are placed in the parent vessel, redirecting blood flow from the aneurysm, promoting reendothelization across the neck, and resulting in complete occlusion of the aneurysm. As a result, flow diversion has become increasingly used as the primary treatment for unruptured aneurysms; however, its applications are being pushed to new frontiers. Here, the authors present three cases showcasing the treatment of intracranial aneurysms with flow diversion.

The video can be found here: https://stream.cadmore.media/r10.3171/2022.7.FOCVID2253

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms

Open access

Flow diversion of a dissecting PICA aneurysm

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

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms

Free access

Focal cortical dysplasia pathology: diagnostic difficulty, classification, and utility for pathogenesis

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.

In Neurosurgical Focus Volume 53 (2022): Issue 4 (Oct 2022): Pediatric Functional Disease

Open access

FRED flow diversion with LVIS protection of large posterior communicating artery aneurysm: the "FRELVIS" technique

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

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms

Open access

Introduction. Flow diversion for cerebral aneurysms

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

In Neurosurgical Focus: Video Volume 7: Issue 2 (Oct 2022): Flow Diversion for Cerebral Aneurysms