RESUMO
OBJECTIVE: Intraventricular hemorrhage (IVH) of prematurity occurs in 20-38% of infants born < 28 weeks gestational age and 15% of infants born in 28-32 weeks gestational age. Treatment has evolved from conservative management and CSF diversion of temporizing and shunting procedures to include strategies aimed at primarily clearing intraventricular blood products. Neuroendoscopic lavage (NEL) aims to decrease the intraventricular blood burden under the same anesthetic as temporizing CSF diversion measures in cases of hydrocephalus from IVH of prematurity. Given the variety of neuroendoscopes, we sought to review the literature and practical considerations to help guide neuroendoscope selection when planning NEL. METHODS: We conducted a systematic review of the literature on neuroendoscopic lavage in IVH of prematurity to examine data on the choice of neuroendoscope and outcomes regarding shunt rate. We then collected manufacturer data on neuroendoscopic devices, including inflow and outflow mechanisms, working channel specifications, and tools compatible with the working channel. We paired this information with the advantages and disadvantages reported in the literature and observations from the experiences of pediatric neurosurgeons from several institutions to provide a pragmatic evaluation of international clinical experience with each neuroendoscope in NEL. RESULTS: Eight studies were identified; four neuroendoscopes have been used for NEL as reported in the literature. These include the Karl Storz Flexible Neuroendoscope, LOTTA® system, GAAB system, and Aesculap MINOP® system. The LOTTA® and MINOP® systems were similar in setup and instrument options. Positive neuroendoscope features for NEL include increased degrees of visualization, better visualization with the evolution of light and camera sources, the ability to sterilize with autoclave processes, balanced inflow and outflow mechanisms via separate channels, and a working channel. Neuroendoscope disadvantages for NEL may include special sterilization requirements, large outer diameter, and limitations in working channels. CONCLUSIONS: A neuroendoscope integrating continuous irrigation, characterized by measured inflow and outflow via separate channels and multiple associated instruments, appears to be the most commonly used technology in the literature. As neuroendoscopes evolve, maximizing clear visualization, adequate inflow, measured outflow, and large enough working channels for paired instrumentation while minimizing the footprint of the outer diameter will be most advantageous when applied for NEL in premature infants.
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Recém-Nascido Prematuro , Neuroendoscopia , Irrigação Terapêutica , Humanos , Neuroendoscopia/métodos , Neuroendoscopia/instrumentação , Recém-Nascido , Irrigação Terapêutica/métodos , Irrigação Terapêutica/instrumentação , Hemorragia Cerebral/cirurgia , Hemorragia Cerebral Intraventricular/cirurgia , Neuroendoscópios , Doenças do Prematuro/cirurgia , Doenças do Prematuro/terapiaRESUMO
INTRODUCTION: Vagus nerve stimulation (VNS) is an adjunctive treatment in children with intractable epilepsy. When lead replacement becomes necessary, the old leads are often truncated and retained and new leads are implanted at a newly exposed segment of the nerve. Direct lead removal and replacement are infrequently described, with outcomes poorly characterized. We aimed to describe our experience with feasibility of VNS lead removal and replacement in pediatric patients. METHODS: Retrospective review examined 14 patients, at a single, tertiary-care, children's hospital, who underwent surgery to replace VNS leads, with complete removal of the existing lead from the vagus nerve and placement of a new lead on the same segment of the vagus nerve, via blunt and sharp dissection without use of electrocautery. Preoperative characteristics, stimulation parameters, and outcomes were collected. RESULTS: Mean age at initial VNS placement was 7.6 years (SD 3.5, range 4.5-13.4). Most common etiologies of epilepsy were genetic (5, 36%) and cryptogenic (4, 29%). Lead replacement was performed at a mean of 6.0 years (SD 3.8, range 2.1-11.7) following initial VNS placement. Reasons for revision included VNS lead breakage or malfunction. There were no perioperative complications, including surgical site infection, voice changes, dysphagia, or new deficits postoperatively. Stimulation parameters after replacement surgery at last follow-up were similar compared to preoperatively, with final stimulation parameters ranging from 0.25 mA higher to 1.5 mA lower to maintain baseline seizure control. The mean length of follow-up was 7.9 years (SD 3.5, range 3.1-13.7). CONCLUSION: Removal and replacement of VNS leads are feasible and can be safely performed in children. Further characterization of surgical technique, associated risk, impact on stimulation parameters, and long-term outcomes are needed to inform best practices in VNS revision.
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Epilepsia Resistente a Medicamentos , Epilepsia , Estimulação do Nervo Vago , Humanos , Criança , Pré-Escolar , Adolescente , Epilepsia/terapia , Epilepsia Resistente a Medicamentos/cirurgia , Convulsões , Estimulação do Nervo Vago/métodos , Estudos Retrospectivos , Nervo Vago , Resultado do TratamentoRESUMO
PURPOSE: The transition from pediatric to adult care can be complex and difficult to navigate for adolescents with cerebral palsy (CP). We aimed to assess the current state of transitional care for young persons with CP and delineate guidelines for best practice with opportunities for intervention. METHODS: A systematic review was conducted using PRISMA guidelines to search PubMed, Embase, and Scopus databases. Articles were screened for relevance via title and abstract prior to full-text review. RESULTS: Of 3151 resultant articles, 27 observational studies were included. Fourteen (52%) studies assessed clinical outcomes of patients with CP during and post-transition. Transition-associated poor outcomes included housing instability, unemployment, difficulty forming relationships, increased hospital admission rates, and decreased use of rehabilitation services. Factors associated with improved outcomes included family participation, promotion of self-efficacy, and meeting the adult team before transition. Nine (33%) studies conducted interviews with transition-age persons with CP. Key themes were a lack of transition preparedness, difficulty navigating the adult system, gaps in seamless care, and limited accessibility to specialists and environments suitable for patients with complex care needs. Four (15%) studies examined features of current transition services. Perceived barriers included poor communication within health service teams, limited adult providers accepting CP patients, and the lack of financial resources for specialized care. There was no standardized transition tool or approach. CONCLUSION: These findings underscore the importance of a planned transition process in optimizing long-term medical and psychosocial outcomes for persons with CP. Further research, including translational, team-based, and community-engaged research, are needed.
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Paralisia Cerebral , Transição para Assistência do Adulto , Cuidado Transicional , Adulto , Adolescente , Humanos , Criança , Paralisia Cerebral/reabilitaçãoRESUMO
OBJECTIVES: Refractory epilepsy is a diagnosis of recurrent seizures that requires multiple resources for optimal chronic management. The disease negatively impacts the lives of affected patients and families and poses an economic burden to the health care system. This study compares hospital costs between pediatric patients treated with antiseizure medications (ASMs) only and ASMs plus vagus nerve stimulation (VNS). METHODS: Patients 0-17 years of age who were diagnosed with refractory epilepsy between January 1, 2011 and December 31, 2016, were identified from the Children's Hospital Association's Pediatric Health Information System (PHIS) database. Patients treated with ASMs only or ASMs plus VNS were included in the study and were followed 1 year prior and 2 years after meeting pre-determined criteria for refractory epilepsy. The difference-in-difference (DID) approach along with the two-part model was used to compare the changes in mean hospital costs captured in the PHIS database over time between the two cohorts. RESULTS: One thousand one hundred thirteen patients treated with ASMs plus VNS and 3471 patients treated with ASMs only were included. At a follow-up time of 2 years, for the ASMs-only cohort, the adjusted all-cause and epilepsy-related mean annual total costs increased by $14 715 (95% confidence interval [CI]: $12 375-$17 055) and $18 437 (95% CI: $15 978-$20 896), respectively. By comparison, the adjusted all-cause and epilepsy-related mean annual total costs of the ASMs plus VNS cohort increased by $12 838 (95% CI: $8171-$17 505) and $15 183 (95% CI: $10 253-$20 113), respectively. Compared to ASMs only, ASMs plus VNS generated a cost savings of $3254 for epilepsy-related annual costs per year after the index date. SIGNIFICANCE: Compared to ASMs alone, ASMs plus VNS is a treatment modality associated with lower annual hospital costs over time. Our study shows that VNS is a cost-beneficial treatment for a national cohort of pediatric patients with refractory epilepsy.
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Epilepsia Resistente a Medicamentos , Epilepsia , Estimulação do Nervo Vago , Criança , Epilepsia Resistente a Medicamentos/etiologia , Epilepsia Resistente a Medicamentos/terapia , Epilepsia/tratamento farmacológico , Custos Hospitalares , Humanos , Resultado do Tratamento , Nervo Vago , Estimulação do Nervo Vago/efeitos adversosRESUMO
BACKGROUND: Greater ventriculomegaly in preterm infants with intraventricular hemorrhage (IVH) has been associated with worse neurodevelopmental outcomes in infancy. We aim to explore the relationship between ventriculomegaly and school-age functional outcome. METHODS: Retrospective review of preterm infants with Grade III/IV IVH from 2006 to 2020. Frontal-occipital horn ratio (FOHR) was measured on imaging throughout hospitalization and last available follow-up scan. Pediatric Cerebral Performance Category (PCPC) scale was used to assess functional outcome at ≥4 years. Ordinal logistic regression was used to determine the relationship between functional outcome and FOHR at the time of Neurosurgery consult, neurosurgical intervention, and last follow-up scan while adjusting for confounders. RESULTS: One hundred and thirty-four infants had Grade III/IV IVH. FOHR at consult was 0.62 ± 0.12 and 0.75 ± 0.13 at first intervention (p < 0.001). On univariable analysis, maximum FOHR, FOHR at the last follow-up scan, and at Neurosurgery consult predicted worse functional outcome (p < 0.01). PVL, longer hospital admission, and gastrotomy/tracheostomy tube also predicted worse outcome (p < 0.05). PVL, maximum FOHR, and FOHR at consult remained significant on multivariable analysis (p < 0.05). Maximum FOHR of 0.61 is a fair predictor for moderate-severe impairment (AUC 75%, 95% CI: 62-87%). CONCLUSIONS: Greater ventricular dilatation and PVL were independently associated with worse functional outcome in Grade III/IV IVH regardless of neurosurgical intervention. IMPACT: Ventriculomegaly measured by frontal-occipital horn ratio (FOHR) and periventricular leukomalacia are independent correlates of school-age functional outcomes in preterm infants with intraventricular hemorrhage regardless of need for neurosurgical intervention. These findings extend the known association between ventriculomegaly and neurodevelopmental outcomes in infancy to functional outcomes at school age. FOHR is a fair predictor of school-age functional outcome, but there are likely other factors that influence functional status, which highlights the need for prospective studies to incorporate other clinical and demographic variables in predictive models.
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Doenças Fetais , Hidrocefalia , Doenças do Prematuro , Leucomalácia Periventricular , Hemorragia Cerebral/complicações , Criança , Feminino , Humanos , Hidrocefalia/diagnóstico por imagem , Lactente , Recém-Nascido , Recém-Nascido Prematuro , Estudos ProspectivosRESUMO
BACKGROUND: Benefits from early surgical intervention in preterm infants with intraventricular hemorrhage (IVH) prior to symptomatic ventriculomegaly must be weighed against risks of surgery. We calculated thresholds of common ventriculomegaly indices at a late-intervention institution to predict subsequent symptomatic ventriculomegaly requiring neurosurgery. METHODS: We retrospectively reviewed neuroimaging and neurosurgical outcomes in preterm infants with grade III/IV IVH between 2007 and 2020. Frontal-occipital horn ratio (FOHR), frontal-temporal horn ratio (FTHR), anterior horn width (AHW), and ventricular index (VI) were measured. Area under the receiver operating curve (AUC) for predicting intervention (initiated after progressive symptomatic ventriculomegaly) was calculated for diagnostic scan, scans during weeks 1-4, and maximum measurement prior to intervention. Threshold values that optimized sensitivity and specificity were derived. RESULTS: A total of 1254 scans in 132 patients were measured. In all, 37 patients had a neurosurgical intervention. All indices differed between those with and without intervention from the first diagnostic scan (p < 0.001). AUC of maximum measurement was 97.1% (95% CI 94.6-99.7) for FOHR, 97.7% (95% CI 95.6-99.8) for FTHR, 96.6% (95% CI 93.9-99.4) for AHW, and 96.8% (95% CI 94.0-99.5) for VI. Calculated thresholds were FOHR 0.66, FTHR 0.62, AHW 15.5 mm, and VI 8.4 mm > p97 (sensitivities >86.8%, specificities >90.1%). CONCLUSION: Ventriculomegaly indices were greater for patients who developed progressive persistent ventriculomegaly from the first diagnostic scan and predicted neurosurgical intervention. IMPACT: We derived thresholds of common ventriculomegaly indices (ventricular index, anterior frontal horn width, fronto-occipital horn and fronto-temporal horn index) to best predict the development of progressive symptomatic post-hemorrhage hydrocephalus in preterm infants with intraventricular hemorrhage. While current thresholds were established by a priori expert consensus, we report the first data-driven derivation of ventriculomegaly thresholds across all indices for the prediction of symptomatic hydrocephalus. Data-derived thresholds will more precisely weigh the risks and benefits of early intervention.
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Doenças Fetais , Hidrocefalia , Doenças do Prematuro , Lactente , Feminino , Humanos , Recém-Nascido , Recém-Nascido Prematuro , Estudos Retrospectivos , Dilatação , Hidrocefalia/diagnóstico por imagem , Hemorragia Cerebral/complicações , Hemorragia Cerebral/diagnóstico por imagem , Ventrículos CerebraisRESUMO
BACKGROUND: Traumatic brain injury (TBI) disproportionately affects children within low- and middle-income countries (LMICs). Prehospital emergency care can mitigate secondary brain injury and improve outcomes. Here, we systematically review clinical practice guidelines (CPGs) for pediatric TBI with the goal to inform LMICs prehospital care. METHODS: A systematic search was conducted in PubMed/Medline, Embase, and Web of Science databases. We appraised evidence-based CPGs addressing prehospital management of pediatric TBI using the Appraisal of Guidelines for Research & Evaluation (AGREE) tool. CPGs were rated as high-quality if ≥ 5 (out of 6) AGREE domains scored > 60%. RESULTS: Of the 326 articles identified, 10 CPGs were included in analysis. All 10 were developed in HICs, and 4 were rated as high-quality. A total of 154 pediatric prehospital recommendations were grouped into three subcategories, initial assessment (35.7%), prehospital treatment (38.3%), and triage (26.0%). Of these, 79 (51.3%) were evidence-based with grading, and 31 (20.1%) were consensus-based without direct evidence. CONCLUSION: Currently available CPGs for prehospital pediatric TBI management were all developed in HICs. Four CPGs have high-quality, and recommendations from these can serve as frameworks for LMICs or resource-limited settings. Context-specific evaluation and implementation of evidence-based recommendations allow LMIC settings to respond to the public health crisis of pediatric TBI and address gaps in trauma care systems.
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Lesões Encefálicas Traumáticas , Lesões Encefálicas , Serviços Médicos de Emergência , Lesões Encefálicas Traumáticas/terapia , Criança , Consenso , Bases de Dados Factuais , HumanosRESUMO
INTRODUCTION: Premature neonates have a high risk of intraventricular hemorrhage (IVH) at birth, the blood products of which activate inflammatory cascades that can cause hydrocephalus and long-term neurological morbidities and sequelae. However, there is no consensus for one treatment strategy. While the mainstay of treatment involves CSF diversion to reduce intracranial pressure, a number of interventions focus on blood product removal at various stages including extraventricular drains (EVD), intra-ventricular thrombolytics, drainage-irrigation-fibrinolytic therapy (DRIFT), and neuroendoscopic lavage (NEL). METHODS: We performed a systematic review and meta-analysis to compare the risks and benefits commonly associated with active blood product removal treatment strategies. We searched MEDLINE, Embase, Scopus, Cochrane Library, and CINAHL databases through Dec 2020 for articles reporting on outcomes of EVDs, thrombolytics, DRIFT, and NEL. Outcomes of interest were rate of conversion to ventriculoperitoneal shunt (VPS), infection, mortality, secondary hemorrhage, and cognitive disability. RESULTS: Of the 10,398 articles identified in the search, 23 full-text articles representing 22 cohorts and 530 patients were included for meta-analysis. These articles included retrospective, prospective, and randomized controlled studies on the use of EVDs (n = 7), thrombolytics (n = 8), DRIFT therapy (n = 3), and NEL (n = 5). Pooled rates of reported outcomes for EVD, thrombolytics, DRIFT, and NEL for ventriculoperitoneal shunt (VPS) placement were 51.1%, 43.3%, 34.3%, and 54.8%; for infection, 15.4%, 12.5%, 4.7%, and 11.0%; for mortality, 20.0%, 11.6%, 6.0%, and 4.9%; for secondary hemorrhage, 5.8%, 7.8%, 20.0%, and 6.9%; for cognitive impairment, 52.6%, 50.0%, 53.7%, and 50.9%. Meta-regression using type of treatment as a categorical covariate showed no effect of treatment modality on rate of VPS conversion or cognitive disability. CONCLUSION: There was a significant effect of treatment modality on secondary hemorrhage and mortality; however, mortality was no longer significant after adjusting for year of publication. Re-hemorrhage rate was significantly higher for DRIFT (p < 0.001) but did not differ among the other modalities. NEL also had lower mortality relative to EVD (p < 0.001) and thrombolytics (p = 0.013), which was no longer significant after adjusting for year of publication. Thus, NEL appears to be safer than DRIFT in terms of risk of hemorrhage, and not different than other blood-product removal strategies in terms of mortality. Outcomes-in terms of shunting and cognitive impairment-did not differ. Later year of publication was predictive of lower rates of mortality, but not the other outcome variables. Further prospective and randomized studies will be necessary to directly compare NEL with other temporizing procedures.
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Hidrocefalia , Doenças do Prematuro , Hemorragia Cerebral/complicações , Hemorragia Cerebral/cirurgia , Humanos , Hidrocefalia/complicações , Hidrocefalia/cirurgia , Recém-Nascido , Doenças do Prematuro/cirurgia , Estudos Retrospectivos , Derivação Ventriculoperitoneal/efeitos adversos , Derivação Ventriculoperitoneal/métodosRESUMO
A 6-year-old girl presented with a 1-week history of progressive upper and lower extremity weakness and bilateral upper extremity dysesthesia. Imaging demonstrated a 4.7 × 1.2-cm enhancing intramedullary lesion in the cervical spine from level C2 to C5 with associated cystic components and syringomyelia. The patient underwent a C2-C5 laminoplasty, with gross total resection of the intramedullary lesion. Histological analysis showed small to medium-sized epithelioid cells, with predominantly a solid architecture focally infiltrating into the adjacent spinal cord tissue. Focal papillary differentiation was present along with peri-vascular pseudorosettes, mucin microcysts, and globules of dense collagen. Focal anaplasia was noted with mitosis (5/10 HPF), focal necrosis, and elevated Ki67 10-15%. These findings were consistent with a myxopapillary ependymoma with anaplastic features. CSF cytology was negative for tumor cells. MYCN amplification was not present. She was treated with targeted proton-beam radiation therapy. This is the fourth case of an intramedullary anaplastic myxopapillary ependymoma to date, and the first case in the cervical spine reported in the literature.
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Ependimoma , Laminoplastia , Neoplasias da Medula Espinal , Vértebras Cervicais/diagnóstico por imagem , Vértebras Cervicais/patologia , Vértebras Cervicais/cirurgia , Criança , Ependimoma/diagnóstico por imagem , Ependimoma/patologia , Ependimoma/cirurgia , Feminino , Humanos , Neoplasias da Medula Espinal/diagnóstico por imagem , Neoplasias da Medula Espinal/patologia , Neoplasias da Medula Espinal/cirurgiaRESUMO
INTRODUCTION: Ventriculosubgaleal shunts (VSGSs) require fewer cerebrospinal (CSF) aspirations than ventricular access devices (VADs) for temporization of posthemorrhagic ventricular dilatation (PHVD) in preterm infants. Cost of postoperative CSF aspiration has not been quantified. METHODS: We reviewed CSF aspiration and laboratory studies obtained in preterm infants with PHVD and VAD at our institution between 2009 and 2020. Cost per aspiration was calculated for materials, labs, and Medicare fee schedule for ventricular puncture through implanted reservoir. We searched PubMed, Cochrane Library, Embase, CINAHL, and Web of Science for meta-analysis of pooled mean number of CSF aspirations and proportion of patients requiring aspiration. RESULTS: Thirty-five preterm infants with PHVD had VAD placed with 22.2 ± 18.4 aspirations per patient. Labs were obtained after every aspiration per local protocol. Cost per aspiration at our institution was USD 935.51. Of 269 published studies, 77 reported on VAD, 29 VSGS, and 13 both. Five studies on VAD (including the current study) had a pooled mean of 25.8 aspirations per patient (95% CI: 16.7-34.8). One study on VSGS reported a mean of 1.6 ± 1.7 aspirations. Three studies on VAD (including the current study) had a pooled proportion of 97.4% of patients requiring aspirations (95% CI: 87.9-99.5). Four studies on VSGS had a pooled proportion of 36.5% requiring aspirations (95% CI: 26.9-47.2). Frequency of lab draws ranged from weekly to daily. Based on costs at our institution, mean number of aspirations, and proportion of patients requiring aspirations, cost difference ranged between USD 4,243 and 23,235 per patient and USD 500,903 and 2.36 million per 100 patients depending on frequency of taps and Medicare locality. DISCUSSION/CONCLUSION: Lower number of CSF aspirations using VSGS can be associated with considerably lower cost compared to VAD.
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Hidrocefalia , Doenças do Prematuro , Idoso , Hemorragia Cerebral/complicações , Hemorragia Cerebral/cirurgia , Ventrículos Cerebrais/cirurgia , Derivações do Líquido Cefalorraquidiano , Dilatação , Humanos , Hidrocefalia/complicações , Hidrocefalia/cirurgia , Lactente , Recém-Nascido , Recém-Nascido Prematuro , Doenças do Prematuro/cirurgia , Medicare , Estudos Retrospectivos , Estados UnidosRESUMO
OBJECTIVE: To determine the relationship between timing of initiation of temporizing neurosurgical treatment and rates of ventriculoperitoneal shunt (VPS) and neurodevelopmental impairment in premature infants with post-hemorrhagic ventricular dilatation (PHVD). STUDY DESIGN: We searched MEDLINE, EMBASE, CINAHL, Web of Science, the Cochrane Database of Systematic Reviews, and the Cochrane Center Register of Controlled Trials for studies that reported on premature infants with PHVD who underwent a temporizing neurosurgical procedure. The timing of the temporizing neurosurgical procedure, gestational age, birth weight, outcomes of conversion to VPS, moderate-to-severe neurodevelopmental impairment, infection, temporizing neurosurgical procedure revision, and death at discharge were extracted. RESULTS: Sixty-two full-length articles and 6 conference abstracts (n = 2533 patients) published through November 2020 were included. Pooled rate for conversion to VPS was 60.5% (95% CI, 54.9-65.8), moderate-severe neurodevelopmental impairment 34.8% (95% CI, 27.4-42.9), infection 8.2% (95% CI, 6.7-10.1), revision 14.6% (95% CI, 10.4-20.1), and death 12.9% (95% CI, 10.2-16.4). The average age at temporizing neurosurgical procedure was 24.2 ± 11.3 days. On meta-regression, older age at temporizing neurosurgical procedure was a predictor of conversion to VPS (P < .001) and neurodevelopmental impairment (P < .01). Later year of publication predicted increased survival (P < .01) and external ventricular drains were associated with more revisions (P = .001). Tests for heterogeneity reached significance for all outcomes and a qualitative review showed heterogeneity in the study inclusion and diagnosis criteria for PHVD and initiation of temporizing neurosurgical procedure. CONCLUSIONS: Later timing of temporizing neurosurgical procedure predicted higher rates of conversion to VPS and moderate-severe neurodevelopmental impairment. Outcomes were often reported relative to the number of patients who underwent a temporizing neurosurgical procedure and the criteria for study inclusion and the initiation of temporizing neurosurgical procedure varied across institutions. There is need for more comprehensive outcome reporting that includes all infants with PHVD regardless of treatment.
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Hemorragia Cerebral , Doenças do Prematuro , Idoso , Hemorragia Cerebral/cirurgia , Dilatação , Humanos , Lactente , Recém-Nascido , Doenças do Prematuro/cirurgia , Procedimentos Neurocirúrgicos , Revisões Sistemáticas como AssuntoRESUMO
Lennox-Gastaut syndrome (LGS) is a severe form of childhood onset epilepsy in which patients require multiple medications and may be candidates for palliative surgical intervention. In this meta-analysis, we sought to evaluate the impact of palliative vagus nerve stimulation (VNS), corpus callosotomy (CC), and resective surgery (RS) by analyzing their impact on seizure control, antiepileptic drug (AED) usage, quality of life (QOL), behavior, cognition, prognostic factors, and complications. A systematic search of PubMed MEDLINE, Scopus, and Cochrane Database of Systematic Reviews was performed to find articles that met the following criteria: (1) prospective/retrospective study with original data, (2) at least one LGS surgery patient aged less than 18 years, and (3) information on seizure frequency reduction (measured as percentage, Engel class, or qualitative comment). Seizures were analyzed quantitatively in a meta-analysis of proportions and a random-effects model, whereas other outcomes were analyzed qualitatively. Forty studies with 892 LGS patients met the selection criteria, with 19 reporting on CC, 17 on VNS, four on RS, two on RS + CC, one on CC + VNS, and one on deep brain stimulation. CC seizure reduction rate was 74.1% (95% confidence interval [CI] = 64.5%-83.7%), and VNS was 54.6% (95% CI = 42.9%-66.3%), which was significantly different (p < .001). RS seizure reduction was 88.9% (95% CI = 66.1%-99.7%). Many VNS patients reported alertness improvements, and most had no major complications. VNS was most effective for atonic/tonic seizures; higher stimulation settings correlated with better outcomes. CC patients reported moderate cognitive and QOL improvements; disconnection syndrome, transient weakness, and respiratory complications were noted. Greater callosotomy extent correlated with better outcomes. AED usage most often did not change after surgery. RS showed considerable QOL improvements for patients with localized seizure foci. In the reported literature, CC appeared to be more effective than VNS for seizure reduction. VNS may provide a similar or higher level of QOL improvement with lower aggregate risk of complications. Patient selection, anatomy, and seizure type will inform decision-making.
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Gerenciamento Clínico , Síndrome de Lennox-Gastaut/fisiopatologia , Síndrome de Lennox-Gastaut/cirurgia , Psicocirurgia/métodos , Humanos , Síndrome de Lennox-Gastaut/diagnóstico , Estudos Prospectivos , Estudos RetrospectivosRESUMO
OBJECTIVE: Drug-resistant epilepsy (DRE) during the first few months of life is challenging and necessitates aggressive treatment, including surgery. Because the most common causes of DRE in infancy are related to extensive developmental anomalies, surgery often entails extensive tissue resections or disconnection. The literature on "ultra-early" epilepsy surgery is sparse, with limited data concerning efficacy controlling the seizures, and safety. The current study's goal is to review the safety and efficacy of ultra-early epilepsy surgery performed before the age of 3 months. METHODS: To achieve a large sample size and external validity, a multinational, multicenter retrospective study was performed, focusing on epilepsy surgery for infants younger than 3 months of age. Collected data included epilepsy characteristics, surgical details, epilepsy outcome, and complications. RESULTS: Sixty-four patients underwent 69 surgeries before the age of 3 months. The most common pathologies were cortical dysplasia (28), hemimegalencephaly (17), and tubers (5). The most common procedures were hemispheric surgeries (48 procedures). Two cases were intentionally staged, and one was unexpectedly aborted. Nearly all patients received blood products. There were no perioperative deaths and no major unexpected permanent morbidities. Twenty-five percent of patients undergoing hemispheric surgeries developed hydrocephalus. Excellent epilepsy outcome (International League Against Epilepsy [ILAE] grade I) was achieved in 66% of cases over a median follow-up of 41 months (19-104 interquartile range [IQR]). The number of antiseizure medications was significantly reduced (median 2 drugs, 1-3 IQR, p < .0001). Outcome was not significantly associated with the type of surgery (hemispheric or more limited resections). SIGNIFICANCE: Epilepsy surgery during the first few months of life is associated with excellent seizure control, and when performed by highly experienced teams, is not associated with more permanent morbidity than surgery in older infants. Thus surgical treatment should not be postponed to treat DRE in very young infants based on their age.
Assuntos
Epilepsia Resistente a Medicamentos , Epilepsia , Malformações do Desenvolvimento Cortical , Idoso , Epilepsia Resistente a Medicamentos/cirurgia , Eletroencefalografia , Epilepsia/cirurgia , Estudos de Viabilidade , Humanos , Lactente , Estudos Retrospectivos , Resultado do TratamentoRESUMO
OBJECTIVES: Refractory epilepsy imposes a substantial burden on affected patients, families, and healthcare system. In terms of treating seizures in children, vagus nerve stimulation (VNS) has been proved to be comparable to that of antiepileptic drugs (AEDs). This study compared healthcare resource utilization between pediatric patients treated with AEDs only and AEDs plus VNS. METHODS: Pediatric patients diagnosed with refractory epilepsy between the 1st of January 2011 and the 31st of December 2016 were identified from the Pediatric Health Information System Database. Patients treated with AEDs only or AEDs plus VNS were included in the study and were followed up from one year before to two years after the date when defined criteria for refractory epilepsy were met. The difference-in-difference approach along with the hurdle model was used to compare the changes in healthcare resource utilization over time between patients treated with AEDs only and AEDs plus VNS. RESULTS: The study included 1502 patients treated with AEDs plus VNS and 4541 patients treated with AEDs only. There was a difference in post-index all-cause and epilepsy-related inpatient visits compared to the pre-index period: inpatient hospitalizations were decreased in the AEDs plus VNS cohort, and increased in the AEDs only cohort. There was no significant difference in the pre-index to post-index change for all-cause and epilepsy-related emergency department visits between the two treatment cohorts. For outpatient encounters in the initial post-index period, patients treated with AEDs plus VNS had significantly higher increase in all-cause and epilepsy-related outpatient visits compared to the AEDs only cohort. CONCLUSIONS: Compared to those treated with AEDs only, pediatric patients with refractory epilepsy treated with AEDs plus VNS have fewer inpatient visits and more outpatient visits within a 2-year follow-up. Given the lower acuity of care in outpatient versus inpatient settings, this study can inform treatment choices for children with refractory epilepsy.
Assuntos
Epilepsia Resistente a Medicamentos , Epilepsia , Estimulação do Nervo Vago , Anticonvulsivantes/uso terapêutico , Criança , Epilepsia Resistente a Medicamentos/tratamento farmacológico , Epilepsia/tratamento farmacológico , Humanos , Aceitação pelo Paciente de Cuidados de Saúde , Estudos Retrospectivos , Resultado do Tratamento , Nervo VagoRESUMO
Children with neurofibromatosis type I (NF1) have a higher predisposition for low-grade astrocytomas of the optic pathway, commonly referred to as optic pathway gliomas (OPGs). OPGs can result in visual deterioration. Treatment outcomes in OPG-NF1 management are often reported around tumor stabilization. We sought to compare vision outcomes associated with different OPG treatment strategies to inform about this important functional metric. A meta-analysis exploring the different modalities to treat children with OPG-NF1 was conducted following Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines using multiple databases. Of the 113 articles identified in the search, 23 full text articles, representing 564 patients, were included for review. These articles included retrospective, prospective, and randomized controlled studies on observation (n=9), chemotherapy (n=19), radiation therapy (n=6), and surgery (n=7). Of the patients undergoing observation, 87% (60/69) demonstrated stable acuity. In the chemotherapy studies, 27.3% (72/264) demonstrated improved acuity/visual field and/or visual-evoked potential amplitudes, 39.4% (104/264) stable acuity, and 33.3% (88/264) deterioration. Both the radiation and surgical treatments reported worsening acuity at 90.9% (10/11) and 73.3% (11/15), respectively. Causal associations are not known. Indications for and timing of treatment choice warrant larger scale study to provide further understanding.
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Neurofibromatose 1/terapia , Glioma do Nervo Óptico/terapia , Criança , Gerenciamento Clínico , Humanos , Neurofibromatose 1/fisiopatologia , Neurofibromatose 1/radioterapia , Neurofibromatose 1/cirurgia , Glioma do Nervo Óptico/fisiopatologia , Glioma do Nervo Óptico/radioterapia , Glioma do Nervo Óptico/cirurgia , Resultado do Tratamento , Visão Ocular/efeitos dos fármacos , Visão Ocular/efeitos da radiação , Acuidade Visual/efeitos dos fármacos , Acuidade Visual/efeitos da radiaçãoRESUMO
OBJECT: While it has been extensively studied in adults, the role of spinal angiography in children with suspected spinal vascular malformations is not fully characterized. With special implications regarding technique, radiation dose, and pathology, we sought to review our single-center experience with pediatric spinal vascular pathology and use a systematic review of the literature to further identify its role in pediatrics. METHODS: A retrospective chart review was conducted under IRB approval for all patients age 0-18 years old who underwent spinal angiography at our institution between 2007 and 2018 for concern for spinal vascular pathology. A simultaneous systematic review was conducted via dedicated search terms in two distinct databases and reviewed to identify all studies referring to spinal angiography or angiograms in pediatric patients. RESULTS: Six patients were included. Three patients (50%) had vascular malformations confirmed on diagnostic angiography and underwent subsequent endovascular treatment. Two patients (33.3%), one with hematomyelia and one with spinal cord infarction, had negative diagnostic angiograms. One patient (16.7%) had a spinal tumor and underwent angiography for further evaluation preoperatively. Spinal angiography was used to aid in diagnosis, preoperative planning, and treatment in these cases. It was demonstrated to be safe in this patient population, with no untoward events, minimal radiation dose, and possible therapeutic applications in select cases. The systematic review identified 11 studies regarding pediatric spinal angiography. These ranged from single case reports to case series of up to 38 patients and highlighted the role of spinal angiography in diagnosis, endovascular treatment, preoperative planning, and postoperative follow-up. CONCLUSIONS: Spinal angiography may be used in a variety of scenarios to better understand the architecture of vascular spinal lesions and facilitate endovascular intervention. While its application in both adult and pediatric pathology is limited to select cases, spinal angiography remains a key diagnostic procedure when approaching vascular lesions or tumors of the spine, assessing for etiology of spinal cord infarcts, and in the evaluation of unexplained hemorrhage in the spinal canal.
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Malformações Vasculares do Sistema Nervoso Central , Embolização Terapêutica , Pediatria , Adolescente , Adulto , Angiografia , Malformações Vasculares do Sistema Nervoso Central/terapia , Criança , Pré-Escolar , Humanos , Lactente , Recém-Nascido , Estudos Retrospectivos , Medula Espinal/diagnóstico por imagem , Coluna Vertebral/diagnóstico por imagemRESUMO
INTRODUCTION: Ventriculoperitoneal shunts (VPS) is commonly performed by pediatric neurosurgeons and there is no consensus in management of VPS infection as it relates to diagnosis and treatment. OBJECTIVE: We utilized an international practitioner-based survey to study the variability in VPS infection diagnostic and therapeutic measures. METHODS: A survey gauging practice patterns of pediatric neurosurgeons regarding VPS and its complication management was distributed. Survey endpoints were analyzed by VPS case volume and pediatric-focused case volume regarding diagnostic measures, use of cerebrospinal fluid (CSF) profile, microbiology, and treatment. RESULTS: A total of 439 surveys were distributed, with a response rate of 31%. Responders ranged from Americas (44.9%), European (31.4%), Asian (18.6%), African (2.5%), to Australian continents (2.5%). Practitioners were stratified based on number and percentage pediatric VPS performed. Institutions performing highest VPS and percentage pediatric case volumes had lower rate of VPS infection. Shunt tap was the most widely used diagnostic study. Overall CSF profile did not affect decision making towards VPS internalization, except for leukocyte count ≤ 20 × 109/L. Practitioners utilized 3 negative cultures prior to VPS internalization. Discrepancies in surgical management were noted amongst centers with high versus low VPS volume and proportion of pediatric-focused case volume. Practice patterns were not noted to be organism dependent. Antibiotic-impregnated shunts were utilized in the Americas and Europe over other regions but only in one third of all initial VPS or as a preventive strategy after a VPS infection has been resolved respectively. DISCUSSION: Survey results from 6 continents in VPS management revealed patterns of lower infection in high-volume centers, 3 negative cultures prior to internalization and aggressive surgical VPS infection management in high-volume institutions.
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Derivações do Líquido Cefalorraquidiano , Hidrocefalia , Austrália , Derivações do Líquido Cefalorraquidiano/efeitos adversos , Criança , Europa (Continente) , Humanos , Hidrocefalia/cirurgia , Próteses e Implantes , Derivação Ventriculoperitoneal/efeitos adversosRESUMO
AIM: To describe 30-day outcomes after intrathecal baclofen (ITB) pump placement in children and identify risk factors for readmission, reoperation, and perioperative complication using the National Surgical Quality Improvement Program-Pediatric (NSQIP-P) database. METHOD: Patients aged 0 to 18 years who underwent ITB pump placement (2012-2014) comprised the study cohort defined in the database. Multivariate regression analysis was performed using preoperative and perioperative data from the American College of Surgeons' NSQIP-P database. Outcomes of interest within 30 days of surgery were (1) unplanned reoperation; (2) unplanned readmission; and (3) composite postoperative event, including complication, reoperation, and/or readmission. RESULTS: We identified 423 surgeries; 246 (58.2%) patients were male, 177 (41.8%) patients were female. Median age was 12 years and 11 months. Median operative time was 70 minutes (interquartile range 56-97min). Mean length of stay was 3.8 days. The patient population had a high number of medical comorbidities. The overall readmission rate was 7.3%; mean postoperative admission date was 14.1 days after surgery. Of readmitted patients, 64.5% underwent reoperation. The most common indication for reoperation was surgical site infection. Female sex was associated with decreased risk of readmission (odds ratio [OR] 0.25, 95% confidence interval [CI] 0.09-0.65; p=0.01); American Society of Anesthesiologists Classification of greater than or equal to 3 was associated with decreased risk of unplanned return to surgery (OR 0.26, 95% CI 0.11-0.66; p=0.04); length of stay greater than or equal to 3 days at index surgery was associated with increased risk of composite 30-day perioperative event (OR 2.33, 95% CI 1.29-4.20; p=0.01). INTERPRETATION: Our data provide national perspectives on 30-day perioperative outcomes for ITB pump placement in children. Results illustrate NSQIP-P database collection methodology and highlight opportunities for quality improvement in clinical practice. WHAT THIS PAPER ADDS: Seven percent of patients who underwent intrathecal baclofen pump placement required readmission within 30 days. The most common indication for reoperation was surgical site infection.
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Baclofeno/administração & dosagem , Bombas de Infusão Implantáveis/efeitos adversos , Bombas de Infusão Implantáveis/estatística & dados numéricos , Relaxantes Musculares Centrais/administração & dosagem , Espasticidade Muscular/tratamento farmacológico , Avaliação de Resultados em Cuidados de Saúde/estatística & dados numéricos , Readmissão do Paciente/estatística & dados numéricos , Complicações Pós-Operatórias/epidemiologia , Reoperação/estatística & dados numéricos , Adolescente , Criança , Feminino , Humanos , Infusão Espinal , Masculino , Complicações Pós-Operatórias/etiologia , Estudos Retrospectivos , Fatores de Risco , Infecção da Ferida Cirúrgica/epidemiologiaRESUMO
PURPOSE: The aim of this study was to use the multicenter American College of Surgeons National Surgical Quality Improvement Program-Pediatric (NSQIP-P) to evaluate and identify risk factors for 30-day adverse events in children undergoing epilepsy surgery. METHODS: Using the 2015 NSQIP-P database, we identified children (age 0-18 years) undergoing pediatric epilepsy surgery and analyzed NSQIP-defined complications, unplanned reoperations, and unplanned readmissions. Multivariable logistic regression analysis was performed using perioperative data to identify risk factors for adverse events within 30 days of the index procedure. RESULTS: Two hundred eight pediatric patients undergoing epilepsy surgery were identified for the year 2015 in the NSQIP-P database. The majority of patients were male (51.8%) and white (72.9%). The median age was 10 years. Neurological and neuromuscular comorbidities were seen in 62.5% of patients. Surgical blood loss and transfusion was the most common overall NSQIP-defined event (15.7%) and was reported in 40% with hemispherectomy. Nineteen patients (6.8%) had an unplanned reoperation and 20 patients (7.1%) had an unplanned readmission. Multivariable logistic regression analysis showed that African American patients (OR 3.26, 95% CI 1.29-8.21, p = 0.01) and hemispherectomy (OR 3.05, 95% CI 1.4-6.65, p = 0.01) were independently associated with NSQIP-defined complications. Patients undergoing hemispherectomy (OR 4.11, 95% CI 1.48-11.42, p = 0.01) were also at significantly higher risk of unplanned readmission after pediatric epilepsy surgery. CONCLUSIONS: Data from the 2015 NSQIP-P database showed that hemispherectomy was significantly associated with higher perioperative events in children undergoing epilepsy surgery. Quality improvement initiatives for hemispherectomy should target surgical blood loss and wound-related complications. Racial disparities in access to cranial pediatric epilepsy surgery and perioperative complications were also highlighted in the present study.
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Bases de Dados Factuais/tendências , Epilepsia/epidemiologia , Epilepsia/cirurgia , Procedimentos Neurocirúrgicos/tendências , Readmissão do Paciente/tendências , Complicações Pós-Operatórias/epidemiologia , Adolescente , Criança , Pré-Escolar , Epilepsia/diagnóstico , Feminino , Humanos , Masculino , Procedimentos Neurocirúrgicos/efeitos adversos , Complicações Pós-Operatórias/diagnóstico , Fatores de Risco , Fatores de Tempo , Resultado do TratamentoRESUMO
PURPOSE: It is common to evaluate children who have sustained minor head trauma with computed tomography (CT) of the head. Scalp swelling, in particular, has been associated with intracranial injury. A subset of patients, however, present in delayed fashion, often days after the head trauma, as soft tissue edema progresses and their caregiver notices scalp swelling. We explore the value of further workup in this setting. METHODS: We conducted a retrospective review of a prospectively collected cohort of children ≤24 months of age presenting to the Texas Children's Hospital with scalp swelling more than 24 h following a head trauma. Cases were collected over a 2-year study period from June 1, 2014 to May 31, 2016. RESULTS: Seventy-six patients comprising 78 patient encounters were included in our study. The mean age at presentation was 8.8 months (range 3 days-24 months). All patients had noncontrast CT of the head as part of their evaluation by emergency medicine, as well as screening for nonaccidental trauma (NAT) by the Child Protection Team. The most common finding on CT head was a linear/nondisplaced skull fracture (SF) with associated extra-axial hemorrhage (epidural or subdural hematoma), which was found in 31/78 patient encounters (40%). Of all 78 patient encounters, 43 patients (55%) were discharged from the emergency room (ER), 17 patients (22%) were admitted for neurologic monitoring, and 18 patients (23%) were admitted solely to allow further NAT evaluation. Of those patients admitted, none experienced a neurologic decline and all had nonfocal neurologic exams on discharge. No patient returned to the ER in delayed fashion for a neurologic decline. Of all the patient encounters, no patient required surgery. CONCLUSIONS: Pediatric patients ≤24 months of age presenting to the ER in delayed fashion with scalp swelling after minor head trauma-who were otherwise nonfocal on examination-did not require surgical intervention and did not experience any neurologic decline. Further radiographic investigation did not alter neurosurgical management in these patients; however, it should be noted that workup for child abuse and social care may have been influenced by CT findings, suggesting the need for the future development of a clinical decision-making tool to help safely avoid CT imaging in this setting.