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The natural history of postoperative hydrocephalus after pediatric hemispherectomy for medically refractory epilepsy: an institutional experience

Victor M. Lu, Shelly Wang, and John Ragheb

OBJECTIVE

Hemispherectomy is a complex surgical intervention for medically refractory epilepsy, and its surgical sequelae continue to be defined. The incidence, timing, and predictors of postoperative hydrocephalus are not well understood. Correspondingly, the aim of this study was to define the natural history of the development of hydrocephalus after hemispherectomy based on the authors’ institutional experience.

METHODS

The authors performed a retrospective review of their departmental database for all relevant cases between 1988 and 2018. Demographic and clinical results were abstracted and analyzed using regression analyses to identify predictors of postoperative hydrocephalus.

RESULTS

Of the 114 patients who satisfied selection criteria, there were 53 females (46%) and 61 males (53%) with mean ages of 2.2 and 6.5 years at first seizure and at hemispherectomy, respectively. There were 16 patients (14%) with a history of previous seizure surgery. In terms of surgery, the mean estimated blood loss was 441 ml, with a mean operative time of 7 hours, and 81 patients (71%) required intraoperative transfusions. A planned postoperative external ventricular drain (EVD) was placed in 38 patients (33%). The most common procedural complications were infection and hematoma, occurring in 7 patients (6%) each. Overall, there were 13 patients (11%) with postoperative hydrocephalus requiring permanent CSF diversion, occurring at a median of 1 year (range 0.1–5 years) after surgery. On multivariable analysis, a postoperative EVD (OR 0.12, p < 0.01) was significantly associated with a decreased likelihood of postoperative hydrocephalus, whereas previous surgery history (OR 4.32, p = 0.03) and postoperative infection complication (OR 5.14, p = 0.04) were significantly associated with increased likelihood of postoperative hydrocephalus.

CONCLUSIONS

Postoperative hydrocephalus mandating permanent CSF diversion following hemispherectomy can be expected in approximately 1 in 10 cases, presenting months after surgery on average. A postoperative EVD appears to reduce this likelihood, whereas postoperative infection and previous history of seizure surgery were shown to statistically increase this likelihood. These parameters should be carefully considered in the management of pediatric hemispherectomy for medically refractory epilepsy.

In Journal of Neurosurgery: Pediatrics Publish Before Print

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Safety of vagus nerve stimulation and responsive neurostimulation used in combination for multifocal and generalized onset epilepsy in pediatric patients

Cameron P. Beaudreault, Eris Spirollari, Alexandria F. Naftchi, Vishad Sukul, Ankita Das, Sima Vazquez, Steven M. Wolf, Patricia E. McGoldrick, and Carrie R. Muh

OBJECTIVE

The aim of this study was to assess the safety and efficacy of combined active responsive neurostimulation (RNS) and vagus nerve stimulation (VNS) therapies in pediatric patients with drug-resistant epilepsy.

METHODS

A single-center retrospective chart review was conducted on pediatric patients implanted with the RNS System with a concomitant active VNS System (VNS+RNS) between 2015 and 2021. Patients with at least 1 month of overlapping concomitant VNS and RNS treatment were included. Patients who had an RNS device implanted after 21 years of age, those who had responsive neurostimulators implanted after their VNS was inactivated, or those in whom the VNS battery died and was not replaced before RNS System implantation were excluded.

RESULTS

Seven pediatric VNS+RNS patients were identified, and their courses of treatment were evaluated. All patients tolerated concurrent VNS and RNS treatment well, no device-device interactions were identified, and no major treatment-related adverse effects were noted. The median follow-up after RNS System implantation was 1.2 years. By electroclinical criteria, all 7 patients achieved 75%–99% reductions in the frequency of disabling seizures after RNS System implantation. By patient and caregiver report, 2 patients (28.6%) had 75%–99% reductions in the frequency of their disabling seizures, 2 patients (28.6%) achieved 50%–74% reductions, 2 patients achieved 1%–24% reduction in frequency of disabling seizures, and 1 patient (14.3%) experienced a 1%–24% increase in seizure frequency. The available VNS magnet swipe data identified 2 patients with 75%–99% reductions in seizure frequency as measured by magnet swipes, one with 25%–49% reductions and the other with 1%–24% increases in seizure frequency as measured by magnet swipes.

CONCLUSIONS

This study demonstrated that RNS and VNS therapies can safely be used simultaneously in pediatric patients. RNS may potentially augment the therapeutic effects of VNS treatment. Patients in whom a response to VNS has been suboptimal should still be considered for RNS therapy.

In Journal of Neurosurgery: Pediatrics Publish Before Print

Open access

Carbon fiber lumbopelvic reconstruction following sacral giant cell tumor resection: illustrative case

Jon Raso, Jialun Chi, Lawal A. Labaran, Charles Frank, and Francis H. Shen

BACKGROUND

The use of carbon fiber or polyetheretherketone spine constructs has proven to be a safe and effective alternative to standard metal alloy. The mechanical properties of carbon fiber while unique provide a construct that is comparable in strength to previous titanium-based constructs and have additionally shown greater fatigue resistance. These constructs have been especially useful for the mechanical stabilization of the spine following tumor resection. The subsequent interference seen when imaging a patient with a traditional metallic construct is reduced and allows for improved tumor surveillance after the procedure, and a more accurate delivery of radiotherapy when indicated.

OBSERVATIONS

This case report details the treatment of a 25-year-old female diagnosed with a sacral giant cell tumor. The authors discuss the use of a carbon fiber-reinforced polyetheretherketone for lumbopelvic reconstruction.

LESSONS

Carbon fiber-reinforced polyetheretherketone with its radiolucency and rigidity is a reliable option for complex spinal reconstruction after tumor resection.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 11 (Mar 2023)

Open access

Spinal epidural tuberculoma with osseous involvement: illustrative case

Hershel W. Cannon, Michael Weaver, Anand Kaul, and Ahmed Lazim

BACKGROUND

A tuberculosis infection of the central nervous system can present as a localized, intraspinal tuberculoma. These lesions may cause spinal cord compression requiring early identification and surgical decompression to limit deleterious neurological sequelae.

OBSERVATIONS

A 28-year-old female with a history of opioid use disorder presented with low-back pain in the setting of trauma with progressive bilateral lower extremity radiculopathy and paraparesis. T1- and T2-weighted magnetic resonance imaging sequences of the spine demonstrated a heterogeneously hyperintense extra-axial epidural mass at T11 with mass effect. Biopsy of the lesion revealed benign soft tissue with necrosis and caseating granulomatous inflammation consistent with tuberculoma. The patient underwent laminectomy and debulking of mass for decompression and was subsequently began antitubercular treatment with good neurological outcome.

LESSONS

To the best of the authors’ knowledge, there are only a handful of microbiologically and radiographically confirmed cases of spinal epidural tuberculoma in English literature. These lesions are rare and difficult to clinically and radiographically characterize in the absence of systemic pulmonary TB symptoms. Tuberculoma is an important differential for a spinal epidural mass, particularly because resection with systemic antitubercular treatment results in symptom resolution.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 11 (Mar 2023)

Open access

Transvenous embolization of the direct carotid-cavernous fistula via the pterygoid plexus: illustrative case

Yuki Matsuda, Masafumi Hiramatsu, Kenji Sugiu, Tomohito Hishikawa, Jun Haruma, Kazuhiko Nishi, Yoko Yamaoka, Yuki Ebisudani, Ryu Kimura, Hisanori Edaki, and Isao Date

BACKGROUND

Endovascular treatment is the mainstay of treatment for carotid-cavernous fistulas, but endovascular approaches vary widely. The authors report a rare case of a direct carotid-cavernous fistula with cranial nerve symptoms caused by rupture of a giant aneurysm in which selective transvenous embolization via the pterygoid plexus was performed.

OBSERVATIONS

An 81-year-old man presented with headache and various progressive cranial nerve symptoms due to a direct carotid-cavernous fistula caused by a ruptured giant aneurysm. All the draining veins visualized on preoperative examination immediately before the treatment were occluded except for the pterygoid plexus. Therefore, the authors chose the dilated pterygoid plexus to approach the shunted pouch at the cavernous sinus and achieve shunt obliteration by selective embolization with coils and n-butyl cyanoacrylate.

LESSONS

Careful study of the three-dimensional rotational images in the preoperative examination is important when considering the various approaches to surgery. The pterygoid plexus can be an effective venous approach route to reach the cavernous sinus area.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 11 (Mar 2023)

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Additional growth of the spine and changes in spinal alignment after posterior spinal fusion in adolescent idiopathic scoliosis

Zhipeng Deng, Liang Wang, Yueming Song, Linnan Wang, Xi Yang, Limin Liu, and Lei Wang

OBJECTIVE

Previous studies have evaluated growth in spinal height immediately following surgical posterior correction of idiopathic scoliosis, yet have not reported on further spinal growth following surgery. The aims of this study were to investigate the characteristics of spinal growth after scoliosis surgery and determine whether they affect spinal alignment.

METHODS

The study included 91 patients (mean age 13.93 years) who underwent spinal fusion using pedicle screws for the treatment of adolescent idiopathic scoliosis (AIS). The study population included 70 female and 21 male patients. The height of the spine (HOS), length of the spine (LOS), and spinal alignment parameters were measured on anteroposterior and lateral radiographs. A stepwise multiple linear regression analysis was used to examine the variables that affected HOS gain from growth. The patients were divided into two groups, the growth group and the nongrowth group, according to whether the HOS gain from growth exceeded 1 cm, to analyze the effect of spinal growth on its alignment.

RESULTS

The mean (± SD) HOS gain from growth was 0.88 ± 0.66 (range −0.46 to 3.21) cm, with 40.66% of patients exhibiting growth ≥ 1 cm. This increase was significantly related to young age, male sex, and a small Risser stage (sex: b = −0.532, p < 0.001, male = 1, female = 2; Risser stage: b = −0.185, p < 0.001; age: b = −0.125, p = 0.011; adjusted R2 = 0.442). The variation in LOS was similar to that of HOS. Thoracic kyphosis and upper instrumented vertebra–lowest instrumented vertebra Cobb angle were reduced in both groups, with a greater reduction observed in the growth group. Patients with an increase in HOS < 1 cm showed a larger lumbar lordosis and a greater tendency for the sagittal vertical axis (SVA) to shift backward and the pelvic tilt to decrease (anteverted pelvis) than in the growth group.

CONCLUSIONS

The spine still has growth potential after corrective fusion surgery for AIS, and 40.66% of the patients in this study continued to vertically grow by 1 cm or more. Unfortunately, the height changes cannot be accurately predicted by currently measured parameters. Changes in the sagittal alignment of the spine may affect the vertical growth increment.

In Journal of Neurosurgery: Spine Publish Before Print

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Applying objective metrics to neurosurgical skill development with simulation and spaced repetition learning

Faith C. Robertson, Christopher J. Stapleton, Jean-Valery C. E. Coumans, Federico Nicolosi, Manuela Vooijs, Sarah Blitz, Francesco Guerrini, Giannantonio Spena, Carlo Giussani, Cesare Zoia, and Brian V. Nahed

OBJECTIVE

Surgical skills laboratories augment educational training by deepening one’s understanding of anatomy and allowing the safe practice of technical skills. Novel, high-fidelity, cadaver-free simulators provide an opportunity to increase access to skills laboratory training. The neurosurgical field has historically evaluated skill by subjective assessment or outcome measures, as opposed to process measures with objective, quantitative indicators of technical skill and progression. The authors conducted a pilot training module with spaced repetition learning concepts to evaluate its feasibility and impact on proficiency.

METHODS

The 6-week module used a simulator of a pterional approach representing skull, dura mater, cranial nerves, and arteries (UpSurgeOn S.r.l.). Neurosurgery residents at an academic tertiary hospital completed a video-recorded baseline examination, performing supraorbital and pterional craniotomies, dural opening, suturing, and anatomical identification under a microscope. Participation in the full 6-week module was voluntary, which precluded randomizing by class year. The intervention group participated in four additional faculty-guided trainings. In the 6th week, all residents (intervention and control) repeated the initial examination with video recording. Videos were evaluated by three neurosurgical attendings who were not affiliated with the institution and who were blinded to participant grouping and year. Scores were assigned via Global Rating Scales (GRSs) and Task-based Specific Checklists (TSCs) previously built for craniotomy (cGRS, cTSC) and microsurgical exploration (mGRS, mTSC).

RESULTS

Fifteen residents participated (8 intervention, 7 control). The intervention group included a greater number of junior residents (postgraduate years 1–3; 7/8) compared to the control group (1/7). External evaluators had internal consistency within 0.5% (kappa probability > Z of 0.00001). The total average time improved by 5:42 minutes (p < 0.003; intervention, 6:05, p = 0.07; control, 5:15, p = 0.001). The intervention group began with lower scores in all categories and surpassed the comparison group in cGRS (10.93 to 13.6/16) and cTSC (4.0 to 7.4/10). Percent improvements for the intervention group were cGRS 25% (p = 0.02), cTSC 84% (p = 0.002), mGRS 18% (p = 0.003), and mTSC 52% (p = 0.037). For controls, improvements were cGRS 4% (p = 0.19), cTSC 0.0% (p > 0.99), mGRS 6% (p = 0.07), and mTSC 31% (p = 0.029).

CONCLUSIONS

Participants who underwent a 6-week simulation course showed significant objective improvement in technical indicators, particularly individuals who were early in their training. Small, nonrandomized grouping limits generalizability regarding degree of impact; however, introducing objective performance metrics during spaced repetition simulation would undoubtedly improve training. A larger multiinstitutional randomized controlled study will help elucidate the value of this educational method.

In Journal of Neurosurgery Publish Before Print

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Association of novel mutation in TRPV4 with familial nonsyndromic craniosynostosis with complete penetrance and variable expressivity

Tenzin Gayden, Gabriel Crevier-Sorbo, Wajih Jawhar, Christine Saint-Martin, Robert Eveleigh, Mirko S. Gilardino, Natascia Anastasio, Yannis Trakadis, Angelia V. Bassenden, Albert M. Berghuis, Nada Jabado, and Roy W. R. Dudley

OBJECTIVE

The aim of this study was to characterize a novel pathogenic variant in the transient receptor potential vanilloid 4 (TRPV4) gene, causing familial nonsyndromic craniosynostosis (CS) with complete penetrance and variable expressivity.

METHODS

Whole-exome sequencing was performed on germline DNA of a family with nonsyndromic CS to a mean depth coverage of 300× per sample, with greater than 98% of the targeted region covered at least 25×. In this study, the authors detected a novel variant, c.469C>A in TRPV4, exclusively in the four affected family members. The variant was modeled using the structure of the TRPV4 protein from Xenopus tropicalis. In vitro assays in HEK293 cells overexpressing wild-type TRPV4 or TRPV4 p.Leu166Met were used to assess the effect of the mutation on channel activity and downstream MAPK signaling.

RESULTS

The authors identified a novel, highly penetrant heterozygous variant in TRPV4 (NM_021625.4:c.469C>A) causing nonsyndromic CS in a mother and all three of her children. This variant results in an amino acid change (p.Leu166Met) in the intracellular ankyrin repeat domain distant from the Ca2+-dependent membrane channel domain. In contrast to other TRPV4 mutations in channelopathies, this variant does not interfere with channel activity as identified by in silico modeling and in vitro overexpression assays in HEK293 cells.

CONCLUSIONS

Based on these findings, the authors hypothesized that this novel variant causes CS by modulating the binding of allosteric regulatory factors to TRPV4 rather than directly modifying its channel activity. Overall, this study expands the genetic and functional spectrum of TRPV4 channelopathies and is particularly relevant for the genetic counseling of CS patients.

In Journal of Neurosurgery: Pediatrics Publish Before Print

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Cerebellar tonsil reduction for surgical treatment of Chiari malformation type I in children

Bruno P. Braga, Eric Y. Montgomery, Bradley E. Weprin, Angela V. Price, Brett A. Whittemore, Mark N. Pernik, Frederick Sklar, Rafael De Oliveira Sillero, and Dale M. Swift

OBJECTIVE

The goal of this study was to review the efficacy and safety of different surgical techniques used for treatment of Chiari malformation type I (CM-I) in children.

METHODS

The authors retrospectively reviewed 437 consecutive children surgically treated for CM-I. Procedures were classified into four groups: bone decompression (posterior fossa decompression [PFD]) and duraplasty (PFD with duraplasty [PFDD]), PFDD with arachnoid dissection (PFDD+AD), PFDD with tonsil coagulation of at least one cerebellar tonsil (PFDD+TC), and PFDD with subpial tonsil resection of at least one tonsil (PFDD+TR). Efficacy was measured as a greater than 50% reduction in the syrinx by length or anteroposterior width, patient-reported improvement in symptoms, and rate of reoperation. Safety was measured as the rate of postoperative complications.

RESULTS

The mean patient age was 8.4 years (range 3 months to 18 years). In total, 221 (50.6%) patients had syringomyelia. The mean follow-up was 31.1 months (range 3–199 months), and there was no statistically significant difference between groups (p = 0.474). Preoperatively, univariate analysis showed that non-Chiari headache, hydrocephalus, tonsil length, and distance from the opisthion to brainstem were associated with the surgical technique used. Multivariate analysis demonstrated that hydrocephalus was independently associated with PFD+AD (p = 0.028), tonsil length was independently associated with PFD+TC (p = 0.001) and PFD+TR (p = 0.044), and non-Chiari headache was inversely associated with PFD+TR (p = 0.001). In the treatment groups postoperatively, symptoms improved in 57/69 (82.6%) PFDD patients, 20/21 (95.2%) PFDD+AD patients, 79/90 (87.8%) PFDD+TC patients, and 231/257 (89.9%) PFDD+TR patients, and differences between groups were not statistically significant. Similarly, there was no statistically significant difference in postoperative Chicago Chiari Outcome Scale scores between groups (p = 0.174). Syringomyelia improved in 79.8% of PFDD+TC/TR patients versus only 58.7% of PFDD+AD patients (p = 0.003). PFDD+TC/TR remained independently associated with improved syrinx outcomes (p = 0.005) after controlling for which surgeon performed the operation. For those patients whose syrinx did not resolve, no statistically significant differences between surgery groups were observed in the length of follow-up or time to reoperation. Overall, there was no statistically significant difference between groups in postoperative complication rates, including aseptic meningitis and CSF- and wound-related issues, or reoperation rates.

CONCLUSIONS

In this single-center retrospective series, cerebellar tonsil reduction, by either coagulation or subpial resection, resulted in superior reduction of syringomyelia in pediatric CM-I patients, without increased complications.

In Journal of Neurosurgery: Pediatrics Publish Before Print

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Cerebral ischemia: a frequent complication of large traumatic epidural hematoma in infants

Matthieu Landart, Sandro Benichi, Lelio Guida, Marilena Lazarescu, Marie Bourgeois, Hélène Sauvé-Martin, Raphael Levy, Charles-Joris Roux, Syril James, Timothée de Saint-Denis, Estelle Vergnaud, Giovanna Paternoster, Kevin Beccaria, and Thomas Blauwblomme

OBJECTIVE

Epidural hematoma (EDH) has rarely been studied specifically in infants. The objective of this study was to investigate the outcomes of patients aged < 18 months (infants) with EDH.

METHODS

The authors conducted a single-center retrospective study of 48 infants aged less than 18 months who underwent an operation for a supratentorial EDH in the last decade. Clinical, radiological, and biological variables were used in a statistical analysis to identify factors predictive of radiological and clinical outcome.

RESULTS

Forty-seven patients were included in the final analysis. Seventeen children (36%) had cerebral ischemia on postoperative imaging, either due to stroke (cerebral herniation) or by local compression. Factors associated with ischemia after multivariate logistic regression were the presence of an initial neurological deficit (76% vs 27%, p = 0.03), low platelet count (mean 192 vs 267 per mm3, p = 0.01), low fibrinogen level (mean 1.4 vs 2.2 g/L, p = 0.04) and long intubation time (mean 65.7 vs 10.1 hours, p = 0.03). Cerebral ischemia on MRI was predictive of a poor clinical outcome.

CONCLUSIONS

Infants with EDH have a low mortality rate but a high risk of cerebral ischemia, along with long-term neurological sequelae.

In Journal of Neurosurgery: Pediatrics Publish Before Print