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Nikhil Bellamkonda, H. Westley Phillips, Jia-Shu Chen, Alexander M. Tucker, Cassia Maniquis, Gary W. Mathern, and Aria Fallah

OBJECTIVE

Rasmussen encephalitis (RE) is a rare inflammatory neurological disorder typically involving one hemisphere and resulting in drug-resistant epilepsy and progressive neurological decline. Here, the authors present seizure outcomes in children who underwent epilepsy surgery for RE at a single institution.

METHODS

The records of consecutive patients who had undergone epilepsy surgery for RE at the UCLA Mattel Children’s Hospital between 1982 and 2018 were retrospectively reviewed. Basic demographic information, seizure history, procedural notes, and postoperative seizure and functional outcome data were analyzed.

RESULTS

The cohort included 44 patients, 41 of whom had sufficient data for analysis. Seizure freedom was achieved in 68%, 48%, and 22% of the patients at 1, 5, and 10 years, respectively. The median time to the first seizure for those who experienced seizure recurrence after surgery was 39 weeks (IQR 11–355 weeks). Anatomical hemispherectomy, as compared to functional hemispherectomy, was independently associated with a longer time to postoperative seizure recurrence (HR 0.078, p = 0.03). There was no statistically significant difference in postoperative seizure recurrence between patients with complete hemispherectomy and those who had less-than-hemispheric surgery. Following surgery, 68% of the patients could ambulate and 84% could speak regardless of operative intervention.

CONCLUSIONS

A large proportion of RE patients will have seizure relapse after surgery, though patients with anatomical hemispherectomies may have a longer time to postoperative seizure recurrence. Overall, the long-term data in this study suggest that hemispheric surgery can be seen as palliative treatment for seizures rather than a cure for RE.

Restricted access

Kristian Aquilina, Catherine Hobbs, Shobha Cherian, Alexander Tucker, Helen Porter, Andrew Whitelaw, and Marianne Thoresen

Object

The combination of intraventricular hemorrhage (IVH) and posthemorrhagic ventricular dilation (PHVD) remains an important cause of disability in children surviving prematurity. Currently, there is no clear agreement on the management of neonatal IVH, apart from the eventual insertion of a shunt to control PHVD. Cerebrospinal fluid (CSF) shunts are associated with a relatively high complication rate in this population. The development of new treatment options requires greater understanding of the pathophysiological mechanisms of IVH and PHVD, as well as an opportunity to monitor closely their effects on the immature brain. The authors have developed a neonatal large animal model of IVH with long-term survival, allowing the full development of PHVD.

Methods

Fourteen piglets that were 3 to 24 hours old were randomized to receive slow injections of autologous blood, autologous blood with elevated hematocrit, or artificial CSF after induction of general anesthesia. A fourth group served as controls. All animals underwent surgery to form an artificial fontanelle at the bregma. Physiological parameters, including intracranial pressure and electroencephalography, were monitored during injection.

Results

Serial cranial ultrasonography studies performed during the 23- to 44-day survival period demonstrated progressive ventricular dilation in the animals injected with blood. Ventricular volumes, measured with image analysis software, confirmed the highest dilation after injection of blood with an elevated hematocrit. Histological evaluation showed fibrosis in the basal subarachnoid space of hydrocephalic piglets.

Conclusions

This piglet model closely replicates human neonatal IVH and PHVD. It allows detailed physiological and ultrasonographic monitoring over a prolonged survival period. It is suitable for evaluation of noninvasive as well as surgical options in the management of IVH and PHVD.

Open access

Christopher L. Kalmar, Jordan W. Swanson, Sameer Shakir, Alexander M. Tucker, Benjamin C. Kennedy, Phillip B. Storm, Gregory G. Heuer, Scott P. Bartlett, Jesse A. Taylor, and Shih-Shan Lang

Spring-mediated cranioplasty is a useful treatment modality for correcting scaphocephalic head shape in sagittal craniosynostosis because it is less invasive than whole-vault cranioplasty and offers durable morphologic outcomes. Herein, the authors provide a multimedia demonstration of alternative operative approaches for spring-mediated cranioplasty for sagittal craniosynostosis.

The video can be found here: https://vimeo.com/511256259

Restricted access

Shih-Shan Lang, Amber Valeri, Phillip B. Storm, Gregory G. Heuer, Alexander M. Tucker, Benjamin C. Kennedy, Benjamin W. Kozyak, Anjuli Sinha, Todd J. Kilbaugh, and Jimmy W. Huh

OBJECTIVE

Single-ventricle congenital heart disease (CHD) in pediatric patients with Glenn and Fontan physiology represents a unique physiology requiring the surgical diversion of the systemic venous return from the superior vena cava (Glenn) and then the inferior vena cava (Fontan) directly to the pulmonary arteries. Because many of these patients are on chronic anticoagulation therapy and may have right-to-left shunts, arrhythmias, or lymphatic disorders that predispose them to bleeding and/or clotting, they are at risk of experiencing neurological injury requiring intubation and positive pressure ventilation, which can significantly hamper pulmonary blood flow and cardiac output. The aim of this study was to describe the complex neurological and cardiopulmonary interactions of these pediatric patients after acute central nervous system (CNS) injury.

METHODS

The authors retrospectively analyzed the records of pediatric patients who had been admitted to a quaternary children’s hospital with CHD palliated to bidirectional Glenn (BDG) or Fontan circulation and acute CNS injury and who had undergone intubation and mechanical ventilation. Patients who had been admitted from 2005 to 2019 were included in the study. Clinical characteristics, surgical outcomes, cardiovascular and pulmonary data, and intracranial pressure data were collected and analyzed.

RESULTS

Nine pediatric single-ventricle patients met the study inclusion criteria. All had undergone the BDG procedure, and the majority (78%) were status post Fontan palliation. The mean age was 7.4 years (range 1.3–17.3 years). At the time of acute CNS injury, which included traumatic brain injury, intracranial hemorrhage, and cerebral infarct, the median time interval from the most recent cardiac surgical procedure was 3 years (range 2 weeks–11 years). Maintaining normocarbia to mild hypercarbia for most patients during intubation periods did not cause neurological deterioration, and hemodynamic profiles were more favorable as compared to periods of hypocarbia. Hypocarbia was associated with unfavorable hemodynamics but was necessary to decrease intracranial hypertension. Most patients were managed using low mean airway pressure (MAWP) in order to minimize the impact on preload and cardiac output.

CONCLUSIONS

The authors highlight the complex neurological and cardiopulmonary interactions with respect to partial pressure of arterial CO2 (PaCO2) and MAWP when pediatric CHD patients with single-ventricle physiology require mechanical ventilation. The study data demonstrated that tight control of PaCO2 and minimizing MAWP with the goal of early extubation may be beneficial in this population. A multidisciplinary team of pediatric critical care intensivists, cardiac intensivists and anesthesiologists, and pediatric neurosurgeons and neurologists are recommended to ensure the best possible outcomes.

Free access

Jasmine A. T. DiCesare, Alexander M. Tucker, Irene Say, Kunal Patel, Todd H. Lanman, Frank J. Coufal, Justin Millard, Jeffrey E. Deckey, Siddharth Shetgeri, and Duncan Q. McBride

Cervical spondylosis is one of the most commonly treated conditions in neurosurgery. Increasingly, cervical disc replacement (CDR) has become an alternative to traditional arthrodesis, particularly when treating younger patients. Thus, surgeons continue to gain a greater understanding of short- and long-term complications of arthroplasty. Here, the authors present a series of 4 patients initially treated with Mobi-C artificial disc implants who developed postoperative neck pain. Dynamic imaging revealed segmental kyphosis at the level of the implant. All implants were locked in the flexion position, and all patients required reoperation. This is the first reported case series of symptomatic segmental kyphosis after CDR.

Open access

Christopher L. Kalmar, Jordan W. Swanson, Sameer Shakir, Alexander M. Tucker, Benjamin C. Kennedy, Phillip B. Storm, Gregory G. Heuer, Scott P. Bartlett, Jesse A. Taylor, and Shih-Shan Lang

Cranial spring hardware is generally removed 3 months after placement for spring-mediated cranioplasty. Spring removal is performed as an outpatient procedure under general anesthesia in approximately 15 minutes through the incision locations of the index procedure. Herein, the authors provide a multimedia demonstration of cranial spring hardware removal after spring-mediated cranioplasty for sagittal craniosynostosis.

The video can be found here: https://vimeo.com/511179695

Restricted access

Christopher L. Kalmar, Zachary D. Zapatero, Mychajlo S. Kosyk, Anna R. Carlson, Scott P. Bartlett, Gregory G. Heuer, Alexander M. Tucker, Jesse A. Taylor, Shih-Shan Lang, and Jordan W. Swanson

OBJECTIVE

Children with multiple prematurely fused cranial sutures and those undergoing surgical correction later in life appear to experience worse neurocognitive outcomes, but it is unclear whether higher intracranial pressure (ICP) is implicated in this process. The purpose of this study was to elucidate the effect of age at intervention and number of involved cranial sutures on ICP, as well as to assess which cranial suture closure may be more associated with elevated ICP.

METHODS

The prospective craniofacial database at the authors’ institution was queried for patients undergoing initial corrective surgery for craniosynostosis in whom intraoperative measurement of ICP was obtained prior to craniectomy. Age, involved sutures, and syndromic status were analyzed in the context of measured ICP by using multiple linear regression.

RESULTS

Fifty patients met the inclusion criteria. Age at procedure (p = 0.028, β = +0.060 mm Hg/month) and multiple-suture involvement (p = 0.010, β = +4.175 mm Hg if multisuture) were both significantly implicated in elevated ICP. The actual number of major sutures involved was significantly correlated to ICP (p = 0.001; β = +1.687 mm Hg/suture). Among patients with single-suture involvement, there was an overall significant difference of median ICP across the suture types (p = 0.008), with metopic having the lowest (12.5 mm Hg) and sagittal having the highest (16.0 mm Hg). Patients with multiple-suture involvement had significantly higher ICP (p = 0.003; 18.5 mm Hg). Patients with craniofacial syndromes were 79.3 times more likely to have multiple-suture involvement (p < 0.001). Corrective surgery for craniosynostosis demonstrated significant intraoperative reduction of elevated ICP (all p < 0.050).

CONCLUSIONS

Syndromic status, older age at intervention for craniosynostosis, and multiple premature fusion of cranial sutures were associated with significantly higher ICP.

Restricted access

Shih-Shan Lang, Alexander M. Tucker, Craig Schreiber, Phillip B. Storm, Hongyan Liu, Yimei Li, Rebecca Ichord, Lauren A. Beslow, Neda I. Sedora-Roman, Mougnyan Cox, Hussein Nasser, Arastoo Vossough, Michael J. Fisher, Todd J. Kilbaugh, and Jimmy W. Huh

OBJECTIVE

Digital subtraction angiography (DSA) is commonly performed after pial synangiosis surgery for pediatric moyamoya disease to assess the degree of neovascularization. However, angiography is invasive, and the risk of ionizing radiation is a concern in children. In this study, the authors aimed to identify whether arterial spin labeling (ASL) can predict postoperative angiogram grading. In addition, they sought to determine whether patients who underwent ASL imaging without DSA had similar postoperative outcomes when compared with patients who received ASL imaging and postoperative DSA.

METHODS

The medical records of pediatric patients who underwent pial synangiosis for moyamoya disease at a quaternary children’s hospital were reviewed during a 10-year period. ASL-only and ASL+DSA cohorts were analyzed. The frequency of preoperative and postoperative symptoms was analyzed within each cohort. Three neuroradiologists assigned a visual ASL grade for each patient indicating the change from the preoperative to postoperative ASL perfusion sequences. A postoperative neovascularization grade was also assigned for patients who underwent DSA.

RESULTS

Overall, 21 hemispheres of 14 patients with ASL only and 14 hemispheres of 8 patients with ASL+DSA were analyzed. The groups had similar rates of MRI evidence of acute or chronic stroke preoperatively (61.9% in the ASL-only group and 64.3% in the ASL+DSA group). In the entire cohort, transient ischemic attack (TIA) (p = 0.027), TIA composite (TIA or unexplained neurological symptoms; p = 0.0006), chronic headaches (p = 0.035), aphasia (p = 0.019), and weakness (p = 0.001) all had decreased frequency after intervention. The authors found a positive association between revascularization observed on DSA and the visual ASL grading (p = 0.048). The visual ASL grades in patients with an angiogram indicating robust neovascularization demonstrated improved perfusion when compared with the ASL grades of patients with a poor neovascularization.

CONCLUSIONS

Noninvasive ASL perfusion imaging had an association with postoperative DSA neoangiogenesis following pial synangiosis surgery in children. There were no significant postoperative stroke differences between the ASL-only and ASL+DSA cohorts. Both cohorts demonstrated significant improvement in preoperative symptoms after surgery. Further study in larger cohorts is necessary to determine whether the results of this study are validated in order to circumvent the invasive catheter angiogram.