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Diagnostic Accuracy of Ocular Ultrasonography in Identifying Raised Intracranial Pressure among Pediatric Population. 眼超声诊断小儿颅内压升高的准确性。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 DOI: 10.1159/000530921
Vignan Kappagantu, Tej Prakash Sinha, Deepak Agrawal, Nayer Jamshed, Akshay Kumar, Atin Kumar, R M Pandey, Bharath Gopinath, Vidhya Bhushan, Atul Kumar Tiwari, Sanjeev Kumar Bhoi

Introduction: Role of CT scan, MRI, ophthalmoscopy, direct monitoring by a transducer probe in identifying raised intracranial pressure (ICP) in emergency department is limited. There are few studies correlating elevated optic nerve sheath diameter (ONSD) measured by point of care ultrasound (POCUS) with raised ICP in pediatrics emergencies. We studied the diagnostic accuracy of ONSD, crescent sign, and optic disc elevation in identifying increased ICP in pediatrics.

Methods: Prospective observational study was done between April 2018 and August 2019 after ethics approval. Out of 125 subjects, 40 patients without clinical features of raised ICP were recruited as external controls and 85 with clinical features of raised ICP as study subjects. Their demographic profile, clinical examination, and ocular ultrasound findings were noted. This was followed by CT scan. Out of 85 patients, 43 had raised ICP (cases) and 42 had normal ICP (disease controls). Diagnostic accuracy of ONSD in identifying raised ICP was evaluated using STATA.

Results: The mean ONSD in case group was 5.5 ± 0.6 mm, 4.9 ± 0.5 mm in disease control group and external control group was 4.8 ± 0.3 mm. Cut-off of ONSD for raised ICP at ≥4.5 mm had a sensitivity and specificity of 97.67% and 10.98%, while ≥5.0 mm showed a sensitivity and specificity of 86.05% and 71.95%. Crescent sign and optic disc elevation had good correlation with increased ICP.

Conclusion: ONSD ≥5 mm by POCUS identified raised ICP in pediatric population. Crescent sign and optic disc elevation may function as additional POCUS signs in identifying raised ICP.

导读:CT扫描、MRI、眼镜检查、传感器探头直接监测在急诊科识别颅内压升高中的作用有限。在儿科急诊中,很少有研究将点护理超声(POCUS)测量的视神经鞘直径(ONSD)升高与ICP升高联系起来。我们研究了ONSD、新月形征象和视盘抬高对儿科颅内压增高的诊断准确性。方法:经伦理批准,于2018年4月至2019年8月进行前瞻性观察研究。125名受试者中,40名无升高ICP临床特征的患者作为外部对照,85名有升高ICP临床特征的患者作为研究对象。记录了患者的人口统计资料、临床检查和眼部超声检查结果。随后进行了CT扫描。85例患者中,43例ICP升高(病例),42例ICP正常(疾病对照)。使用STATA评估ONSD诊断颅内压升高的准确性。结果:病例组平均ONSD为5.5±0.6 mm,疾病对照组平均ONSD为4.9±0.5 mm,外对照组平均ONSD为4.8±0.3 mm。ONSD截断值对≥4.5 mm升高的ICP的敏感性和特异性分别为97.67%和10.98%,≥5.0 mm的敏感性和特异性分别为86.05%和71.95%。月牙征和视盘升高与颅内压增高有良好的相关性。结论:ONSD≥5 mm的POCUS可识别儿童ICP升高。新月征象和视盘升高可以作为额外的POCUS征象来识别升高的ICP。
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引用次数: 0
A New Measure of Posterior Morphology in Sagittal Craniosynostosis: The Occipital Bullet Index. 矢状颅后部形态的新测量方法:枕骨子弹指数
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-09-13 DOI: 10.1159/000533168
Griffin P Bins, Deborah Cull, Ryan G Layton, Samuel Kogan, Larry Zhou, Blake Dunson, Lisa R David, Christopher M Runyan

Introduction: Sagittal craniosynostosis (SC) is associated with scaphocephaly, an elongated narrow head shape. Assessment of regional severity in the scaphocephalic head is limited by the use of serial computed tomographic (CT) imaging or complex computer programing. Three-dimensional measurements of cranial surface morphology provide a radiation-free alternative for assessing cranial shape. This study describes the creation of an occipital bulleting index (OBI), a novel tool using surface morphology to assess the regional severity in patients with SC.

Methods: Surface imaging from CT scans or 3D photographs of 360 individuals with SC and 221 normocephalic individuals were compared to identify differences in morphology. Cartesian grids were created on each individual's surface mesh using equidistant axial and sagittal planes. Area under the curve (AUC) analyses were performed to identify trends in regional morphology and create measures capturing population differences.

Results: The largest differences were located in the medial regions posteriorly. Using these population trends, a measure was created to maximize AUC. The OBI has an AUC of 0.72 with a sensitivity of 74% and a specificity of 61%. When the frontal bossing index is applied in tandem, the two have a sensitivity of 94.7% and a specificity of 93.1%. Correlation between the two scores in individuals with SC was found to be negligible with an intraclass correlation coefficient of 0.018. Severity was found to be independent of age under 24 months, sex, and imaging modality.

Conclusions: This index creates a tool for differentiating control head shapes from those with SC and has the potential to allow for objective evaluation of the regional severity, outcomes of different surgical techniques, and tracking shape changes in individuals over time, without the need for radiation.

简介矢状颅畸形(SC)与肩胛畸形(一种狭长的头型)有关。由于使用序列计算机断层扫描(CT)成像或复杂的计算机程序,对肩胛畸形头部区域严重程度的评估受到限制。颅骨表面形态的三维测量为评估颅骨形状提供了一种无辐射的替代方法。本研究描述了枕骨弹起指数(OBI)的创建过程,这是一种利用表面形态学评估 SC 患者区域严重程度的新型工具:方法:对 360 名 SC 患者和 221 名正常颅脑患者的 CT 扫描或 3D 照片进行表面成像比较,以确定形态上的差异。使用等距轴向和矢状面在每个人的表面网格上创建笛卡尔网格。进行了曲线下面积(AUC)分析,以确定区域形态的趋势,并建立捕捉群体差异的指标:结果:差异最大的区域位于后方的内侧区域。利用这些群体趋势,创建了一种测量方法,以最大限度地提高 AUC。OBI 的 AUC 为 0.72,灵敏度为 74%,特异度为 61%。如果同时应用额部翘曲指数,两者的灵敏度为 94.7%,特异度为 93.1%。在 SC 患者中,两个评分之间的相关性微乎其微,类内相关系数为 0.018。严重程度与 24 个月以下的年龄、性别和成像方式无关:该指数是区分对照组头型和SC组头型的工具,可用于客观评估区域严重程度、不同手术技术的效果,以及跟踪个体随时间推移的头型变化,且无需放射线。
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引用次数: 0
Serial Neuroendoscopic Lavage for the Treatment of Elevated Cerebrospinal Fluid Protein Levels in Infants with Gram-Negative Rod Ventriculitis. 连续神经内镜灌洗治疗患有革兰氏阴性杆菌脑室炎的婴儿脑脊液蛋白水平升高。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-09-13 DOI: 10.1159/000534083
Jasmine L Hect, Roberta K Sefcik, Kamil W Nowicki, Joel Katz, Stephanie Greene

Introduction: Gram-negative rod (GNR) bacterial ventriculitis is a rare complication of shunt-dependent hydrocephalus, often requiring an extended and invasive treatment course. Accumulation of purulent material, as well as empyema and septation formation, limits circulation of antibiotics and infection clearance. Supplementation of standard care with neuroendoscopic-guided intraventricular lavage with lactated Ringer solution and fenestration of septations may facilitate infection clearance and simplify the eventual shunt construct required. Here, the utility of serial lavage for ventriculitis is described in a population of shunt-dependent neonates and infants at high risk for morbidity and mortality.

Methods: Five infants with shunt-dependent hydrocephalus and subsequent GNR ventriculitis were treated with standard care measures with the addition of serial neuroendoscopic lavage. A retrospective chart review was performed to collect patient characteristics, shunt dependency, and shunt revisions within a year of ventriculitis resolution.

Results: Patients demonstrated a mean 74% decrease in cerebrospinal fluid (CSF) protein following each neuroendoscopic lavage and trended toward a shorter time to infection clearance in comparison to previously published literature. Patients required 0-2 shunt revisions at 1-year follow-up following hospitalization for shunt-related ventriculitis (mean 0.8 +/- 0.8).

Conclusions: Serial neuroendoscopic lavage is an effective technique, used alone or in combination with fenestration of septations, to reduce the CSF protein and bacterial load in the treatment of ventriculitis, decreasing time until eradication of infection. Serial lavage may reduce the risk of future shunt malfunction, simplify the future shunt construct, and decrease duration of infection.

导言:革兰氏阴性杆菌(GNR)细菌性脑室炎是分流依赖性脑积水的一种罕见并发症,通常需要长时间的侵入性治疗。脓性物质的积聚以及气肿和隔膜的形成限制了抗生素的循环和感染的清除。在神经内镜引导下用乳酸林格液进行脑室内灌洗,并对隔膜进行栅栏缝合,作为标准治疗的补充,可促进感染清除,并简化最终所需的分流结构。在此,我们将对依赖分流术的新生儿和高发病率和死亡率风险的婴儿进行描述,说明连续灌洗治疗脑室炎的效用:方法:对五名患有分流依赖性脑积水并继发 GNR 脑室炎的婴儿采用标准护理措施进行治疗,并增加了连续神经内镜灌洗。研究人员对病历进行了回顾性分析,以收集患者特征、分流依赖性以及脑室炎缓解后一年内的分流改造情况:结果:每次神经内镜灌洗后,患者脑脊液(CSF)蛋白平均下降74%,与之前发表的文献相比,感染清除时间呈缩短趋势。因分流相关脑室炎住院的患者在1年的随访中需要进行0-2次分流改造(平均0.8 +/- 0.8):结论:连续神经内镜灌洗是治疗脑室炎的一种有效技术,可单独使用或与鼻腔穿刺术结合使用,以减少脑脊液蛋白和细菌负荷,缩短根除感染的时间。连续灌洗可降低分流管未来发生故障的风险,简化未来的分流管构建,并缩短感染持续时间。
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引用次数: 0
Peri-Insular Hemispherotomy: A Systematic Review and Institutional Experience. 岛周半球切开术:系统回顾和机构经验。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 DOI: 10.1159/000529098
Charles F Yates, Stephen Malone, Kate Riney, Ubaid Shah, Martin J Wood

Introduction: Peri-insular hemispherotomy (PIH) is a hemispheric separation technique under the broader hemispherotomy group, a surgical treatment for patients with intractable epilepsy. Hemispherotomy techniques such as the PIH, vertical parasagittal hemispherotomy (VPH), and modified-lateral hemispherotomy are commonly assessed together, despite significant differences in anatomical approach and patient selection. We aim to describe patient selection, outcomes, and complications of PIH in its own right.

Methods: A systematic review of the literature, in accordance with the Preferred Reporting Items of Systematic Reviews and Meta-Analyses (PRISMA) guidelines, was conducted, with searches of the PubMed and Embase databases. A local series including patients receiving PIH and followed up at the Queensland Children's Hospital between 2014 and 2020 was included.

Results: Systematic review of the literature identified 393 patients from 13 eligible studies. Engel class 1 outcomes occurred in 82.4% of patients, while 8.6% developed post-operative hydrocephalus. Hydrocephalus was most common in the youngest patient cohorts. Developmental pathology was present in 114 (40.8%) patients, who had fewer Engel 1 outcomes compared to those with acquired pathology (69.1% vs. 83.7%, p = 0.0167). The local series included 13 patients, 11/13 (84.6%) had Engel class 1 seizure outcomes. Post-operative hydrocephalus occurred in 2 patients (15.4%), and 10/13 (76.9%) patients had worsened neurological deficit.

Conclusion: PIH delivers Engel 1 outcomes for over 4 in 5 patients selected for this procedure, greater than described in combined hemispherectomy analyses. It is an effective technique in patients with developmental and acquired pathologies, despite general preference of VPH in this patient group. Finally, very young patients may have significant seizure and cognitive benefits from PIH; however, hydrocephalus is most common in this group warranting careful risk-benefit assessment. This review delivers a dedicated PIH outcomes analysis to inform clinical and patient decision-making.

简介:岛叶周围半球切开术(PIH)是广义半球切开术组下的一种半球分离技术,是一种治疗难治性癫痫的手术方法。尽管解剖入路和患者选择存在显著差异,但通常将PIH、垂直副矢状半球切开术(VPH)和改良外侧半球切开术等半球切开术一起进行评估。我们的目的是描述患者的选择,结果,并在其本身的权利的PIH并发症。方法:根据系统评价和荟萃分析(PRISMA)指南的首选报告项目对文献进行系统评价,并检索PubMed和Embase数据库。包括2014年至2020年期间在昆士兰儿童医院接受PIH并随访的当地系列患者。结果:系统回顾文献,从13项符合条件的研究中确定了393例患者。82.4%的患者出现Engel 1级结局,8.6%的患者出现术后脑积水。脑积水在最年轻的患者队列中最常见。114例(40.8%)患者存在发育性病理,与获得性病理患者相比,其Engel 1结果较少(69.1% vs. 83.7%, p = 0.0167)。局部纳入13例患者,11/13(84.6%)发生Engel 1级癫痫发作。术后发生脑积水2例(15.4%),10/13例(76.9%)患者神经功能缺损加重。结论:选择PIH手术的患者中,超过4 / 5的患者达到Engel 1结果,高于联合半球切除术分析。这是一种有效的技术,在患者的发展和获得性病理,尽管普遍倾向于VPH在这组患者。最后,非常年轻的患者可能从PIH中获得显著的癫痫发作和认知益处;然而,脑积水在这一组中最常见,需要仔细的风险-收益评估。本综述提供了一个专门的PIH结果分析,为临床和患者决策提供信息。
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引用次数: 0
Developing Predictive Models to Anticipate Shunt Complications in 33,248 Pediatric Patients with Shunted Hydrocephalus Utilizing Machine Learning. 利用机器学习开发预测模型,预测33248例分流性脑积水患儿的分流并发症。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-06-30 DOI: 10.1159/000531754
Shane Shahrestani, Nathan Shlobin, Julian L Gendreau, Nolan J Brown, Alexander Himstead, Neal A Patel, Noah Pierzchajlo, Sachiv Chakravarti, Darrin Jason Lee, Peter A Chiarelli, Carli L Bullis, Jason Chu

Introduction: Hydrocephalus is a common pediatric neurosurgical pathology, typically treated with a ventricular shunt, yet approximately 30% of patients experience shunt failure within the first year after surgery. As a result, the objective of the present study was to validate a predictive model of pediatric shunt complications with data retrieved from the Healthcare Cost and Utilization Project (HCUP) National Readmissions Database (NRD).

Methods: The HCUP NRD was queried from 2016 to 2017 for pediatric patients undergoing shunt placement using ICD-10 codes. Comorbidities present upon initial admission resulting in shunt placement, Johns Hopkins Adjusted Clinical Groups (JHACG) frailty-defining criteria, and Major Diagnostic Category (MDC) at admission classifications were obtained. The database was divided into training (n = 19,948), validation (n = 6,650), and testing (n = 6,650) datasets. Multivariable analysis was performed to identify significant predictors of shunt complications which were used to develop logistic regression models. Post hoc receiver operating characteristic (ROC) curves were created.

Results: A total of 33,248 pediatric patients aged 6.9 ± 5.7 years were included. Number of diagnoses during primary admission (OR: 1.05, 95% CI: 1.04-1.07) and initial neurological admission diagnoses (OR: 3.83, 95% CI: 3.33-4.42) positively correlated with shunt complications. Female sex (OR: 0.87, 95% CI: 0.76-0.99) and elective admissions (OR: 0.62, 95% CI: 0.53-0.72) negatively correlated with shunt complications. ROC curve for the regression model utilizing all significant predictors of readmission demonstrated area under the curve of 0.733, suggesting these factors are possible predictors of shunt complications in pediatric hydrocephalus.

Conclusion: Efficacious and safe treatment of pediatric hydrocephalus is of paramount importance. Our machine learning algorithm delineated possible variables predictive of shunt complications with good predictive value.

引言:脑积水是一种常见的儿科神经外科病理学,通常通过脑室分流术进行治疗,但约30%的患者在术后第一年内出现分流术失败。因此,本研究的目的是利用从医疗保健成本和利用项目(HCUP)国家研究数据库(NRD)检索的数据来验证儿科分流并发症的预测模型。方法:从2016年到2017年,使用ICD-10代码查询接受分流安置的儿科患者的HCUP NRD。获得初次入院时出现的合并症导致分流、约翰斯·霍普金斯调整临床组(JHACG)虚弱定义标准和入院时的主要诊断类别(MDC)分类。数据库分为训练(n=19948)、验证(n=6650)和测试(n=665)数据集。进行多变量分析以确定分流并发症的重要预测因素,并用于开发逻辑回归模型。创建了自组织接收器工作特性(ROC)曲线。结果:共纳入33248名儿童患者,年龄6.9±5.7岁。初次入院期间的诊断数(OR:1.05,95%CI:1.04-1.07)和初次神经系统入院诊断数(OR:3.83,95%CI:3.33-4.42)与分流并发症呈正相关。女性(OR:0.87,95%CI:0.76-0.99)和选择性入院(OR:0.62,95%CI:0.53-0.72)与分流并发症呈负相关。利用所有再入院的重要预测因素的回归模型的ROC曲线显示曲线下面积为0.733,表明这些因素可能是儿童脑积水分流并发症的预测因素。结论:有效、安全的治疗小儿脑积水至关重要。我们的机器学习算法描绘了可预测分流并发症的可能变量,具有良好的预测价值。
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引用次数: 0
Intracranial Empyemas in the COVID-19 Era: A New Phenomenon? A Paediatric Case Series and Review of the Literature. 新冠肺炎时代的颅内积脓:一种新现象?儿科病例系列及文献复习。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-06-30 DOI: 10.1159/000531753
Benjamin J Hall, John C Duddy, Katerina Apostolopoulou, Raenette David, Arthur Kurzbuch, Abhishek Nadkarni, Sandhya Trichinopoly Krishna, Ben Cooper, Hayley Gouldbourne, Dawn Hennigan, William Dawes, Jonathan Ellenbogen, Christopher Parks, Benedetta Pettorini, Ajay Sinha, Conor Mallucci

Introduction: We present the largest series of paediatric intracranial empyemas occurring after COVID-19 infection to date, and discuss the potential implications of the pandemic on this neurosurgical pathology.

Methods: Patients admitted to our centre between January 2016 and December 2021 with a confirmed radiological diagnosis of intracranial empyema were retrospectively reviewed, excluding non-otorhinological source cases. Patients were grouped according to onset before or after onset of the COVID-19 pandemic and COVID-19 status. A literature review of all post-COVID-19 intracranial empyemas was performed. SPSS v27 was used for statistical analysis.

Results: Sixteen patients were diagnosed with intracranial empyema: n = 5 prior to 2020 and n = 11 after, resulting in an average annual incidence of 0.3% prior to onset of the pandemic and 1.2% thereafter. Of those diagnosed since the pandemic, 4 (25%) were confirmed to have COVID-19 on recent PCR test. Time from COVID-19 infection until empyema diagnosis ranged from 15 days to 8 weeks. Mean age for post-COVID-19 cases was 8.5 years (range: 7-10 years) compared to 11 years in non-COVID cases (range: 3-14 years). Streptococcus intermedius was grown in all cases of post-COVID-19 empyema, and 3 of 4 (75%) post-COVID-19 cases developed cerebral sinus thromboses, compared to 3 of 12 (25%) non-COVID-19 cases. All cases were discharged home with no residual deficit.

Conclusion: Our post-COVID-19 intracranial empyema series demonstrates a greater proportion of cerebral sinus thromboses than non-COVID-19 cases, potentially reflecting the thrombogenic effects of COVID-19. Incidence of intracranial empyema at our centre has increased since the start of the pandemic, causes of which require further investigation and multicentre collaboration.

简介:我们介绍了迄今为止新冠肺炎感染后发生的最大系列儿科颅内脓胸,并讨论了大流行对这种神经外科病理的潜在影响。方法:对2016年1月至2021年12月期间入住我中心并经放射学诊断为颅内积脓的患者进行回顾性分析,不包括非耳鼻源病例。根据新冠肺炎大流行开始前或之后的发病情况和新冠肺炎状态对患者进行分组。对所有COVID-19后颅内积脓进行了文献回顾。采用SPSS v27软件进行统计分析。结果:16名患者被诊断为颅内积脓:2020年前n=5,2020年后n=11,导致疫情爆发前和疫情爆发后的年均发病率分别为0.3%和1.2%。自疫情以来确诊的人中,有4人(25%)在最近的PCR检测中被证实患有新冠肺炎。从新冠肺炎感染到脓胸诊断的时间从15天到8周不等。新冠肺炎后病例的平均年龄为8.5岁(范围:7-10岁),而非新冠肺炎病例为11岁(范围为3-14岁)。中间链球菌在所有COVID-19后脓胸病例中生长,4例(75%)新冠肺炎后病例中有3例出现脑窦血栓,而12例(25%)非新冠肺炎病例中有三例出现脑窦道血栓。所有病例均出院回家,没有任何残余缺陷。结论:我们的COVID-19后颅内积脓系列显示,脑窦血栓形成的比例高于非COVID-19-19病例,这可能反映了COVID-19]的血栓形成效应。自疫情开始以来,我们中心的颅内积脓发病率有所上升,其原因需要进一步调查和多中心合作。
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引用次数: 0
Considerations for Choice of Cranioplasty Material for Pediatric Patients. 儿科患者颅骨成形术材料选择的考虑。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 DOI: 10.1159/000528543
Megan E H Still, Sonja Samant, Abraham Alvarado, Dan Neal, Lance S Governale, Jessica A Ching

Introduction: Optimal material and timing of cranioplasty in the pediatric population continue to be debated. Autologous and alloplastic materials have various indications for use and risk factors for complications.

Methods: A single-center retrospective cohort study was undertaken of all pediatric patients who underwent cranioplasty with any material from 1991-2021.

Results: 149 cranioplasty implants were included. Younger age (6 years old or under), a diagnosis of craniosynostosis as reason for implant, use of autologous bone, and shorter times to cranioplasty were predictive of need for revision surgery. No factors studied had a statistically significant impact on rate of removal of implant at time of revision surgery.

Conclusion: Autologous and alloplastic cranioplasty materials both have good outcomes with low rates of revision surgery in the pediatric population. Alloplastic implants may be considered in the setting of infection as reason for craniectomy given the lower rate of revision surgery and need for removal. Patients with craniosynostosis as reason for cranioplasty have a higher risk of requiring revision or additional surgeries, regardless of implant used.

儿童颅骨成形术的最佳材料和时机仍在争论中。自体和同种异体材料有不同的适应症和并发症的危险因素。方法:对1991-2021年间接受任何材料颅骨成形术的所有儿科患者进行单中心回顾性队列研究。结果:共纳入149例颅骨成形术。年龄较小(6岁或以下),诊断为颅缝闭锁的原因种植,使用自体骨,以及较短的颅骨成形术时间预测需要翻修手术。没有研究的因素在翻修手术时对种植体拔除率有统计学上显著的影响。结论:自体和同种异体颅骨成形术在儿童人群中均具有良好的效果,翻修手术率低。同种异体植入物可以考虑在感染的情况下作为颅骨切除术的原因,因为翻修手术的比例较低,需要切除。颅缝闭锁作为颅骨成形术原因的患者需要翻修或额外手术的风险更高,无论使用何种植入物。
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引用次数: 0
Immunotherapy for Pediatric Brain and Spine Tumors: Current State and Future Directions. 儿童脑和脊柱肿瘤的免疫治疗:现状和未来方向。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2022-12-22 DOI: 10.1159/000528792
Dagoberto Estevez-Ordonez, Sam E Gary, Travis J Atchley, Pedram D Maleknia, Jordan A George, Nicholas M B Laskay, Evan G Gross, Rishi K Devulapalli, James M Johnston

Background: Brain tumors are the most common solid tumors and the leading cause of cancer-related deaths in children. Incidence in the USA has been on the rise for the last 2 decades. While therapeutic advances in diagnosis and treatment have improved survival and quality of life in many children, prognosis remains poor and current treatments have significant long-term sequelae.

Summary: There is a substantial need for the development of new therapeutic approaches, and since the introduction of immunotherapy by immune checkpoint inhibitors, there has been an exponential increase in clinical trials to adopt these and other immunotherapy approaches in children with brain tumors. In this review, we summarize the current immunotherapy landscape for various pediatric brain tumor types including choroid plexus tumors, embryonal tumors (medulloblastoma, AT/RT, PNETs), ependymoma, germ cell tumors, gliomas, glioneuronal and neuronal tumors, and mesenchymal tumors. We discuss the latest clinical trials and noteworthy preclinical studies to treat these pediatric brain tumors using checkpoint inhibitors, cellular therapies (CAR-T, NK, T cell), oncolytic virotherapy, radioimmunotherapy, tumor vaccines, immunomodulators, and other targeted therapies.

Key messages: The current landscape for immunotherapy in pediatric brain tumors is still emerging, but results in certain tumors have been promising. In the age of targeted therapy, genetic tumor profiling, and many ongoing clinical trials, immunotherapy will likely become an increasingly effective tool in the neuro-oncologist armamentarium.

背景:脑肿瘤是最常见的实体肿瘤,也是儿童癌症相关死亡的主要原因。在过去的20年里,美国的发病率一直在上升。虽然诊断和治疗方面的治疗进展改善了许多儿童的生存和生活质量,但预后仍然很差,目前的治疗方法有严重的长期后遗症。摘要:目前迫切需要开发新的治疗方法,自引入免疫检查点抑制剂免疫治疗以来,采用这些和其他免疫治疗方法治疗脑肿瘤儿童的临床试验呈指数增长。在这篇综述中,我们总结了目前各种儿童脑肿瘤的免疫治疗前景,包括脉络膜丛肿瘤、胚胎性肿瘤(髓母细胞瘤、AT/RT、PNETs)、室管膜瘤、生殖细胞肿瘤、胶质瘤、胶质神经元和神经元肿瘤以及间充质肿瘤。我们讨论了使用检查点抑制剂、细胞疗法(CAR-T、NK、T细胞)、溶瘤病毒疗法、放射免疫疗法、肿瘤疫苗、免疫调节剂和其他靶向疗法治疗这些儿童脑肿瘤的最新临床试验和值得注意的临床前研究。关键信息:目前儿童脑肿瘤免疫治疗的前景仍处于新兴阶段,但某些肿瘤的结果是有希望的。在靶向治疗、基因肿瘤分析和许多正在进行的临床试验的时代,免疫治疗可能会成为神经肿瘤学家装备中越来越有效的工具。
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引用次数: 0
The Predictors of Seizures in Patients with Encephalocele: An 11-Year Experience from a Tertiary Hospital. 脑膨出患者癫痫发作的预测因素;在三级医院工作了11年。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-09-26 DOI: 10.1159/000534140
Amirhosein Nejat, Samuel Berchi Kankam, Vahid Heidari, Keyvan Tayebi Meybodi, Zohreh Habibi, Sajedeh Karami, Farideh Nejat

Introduction: The aim of the study was to investigate and identify the predictors associated with the incidence of seizures in patients with encephalocele (EC).

Methods: A retrospective analysis was undertaken of patients treated for EC at a tertiary medical center in Tehran between 2010 and 2021. Data including age at presentation, gender, location, size, and content of EC, ventriculomegaly, hydrocephalus, associated anomalies, and neurodevelopmental delay (NDD) were evaluated for their prognostic value. In addition, univariate and multivariate analyses were performed to identify the correlation between independent predictors and seizure incidence.

Results: One hundred and two cases of EC were identified. Seventy-one ECs (69.6%) were posterior ECs, while 31 (30.4%) were anterior. Neural tissue was found in 43 (42.2%) of the ECs. Thirty-three patients (32.4%) had ventriculomegaly, of which 90.9% underwent shunt placement for progressive or symptomatic hydrocephalus. Seizure was found in 26 (25.5%) patients. On univariate analysis, presence of other anomalies, postoperative infections, and NDD were associated with seizures (p < 0.05). When the anomalies were categorized into intracranial and extracranial groups in univariate analysis, none was associated with statistically significant increase in seizure (p values of 0.09 and 0.61, respectively). Although according to multivariate analysis, only the association between other associated anomalies and seizure was near significant (OR: 2.0, 95% CI: 0.95-4.2, p = 0.049). Children with NDD and postoperative infection were, respectively, 3.04 and 1.3 times more at risk to experience seizures compared to other patients.

Conclusion: We found a rate of 25.5% risk of seizure in patients with EC. This study could not find any significant predictors of seizure in children with EC. However, pediatric patients with postoperative infections including sepsis, wound infection, and NDD require more consideration to reduce the risk of seizure.

引言:研究并确定与脑膨出(EC)患者癫痫发作发生率相关的预测因素。方法:对2010年至2021年间在德黑兰一家三级医疗中心接受EC治疗的患者进行回顾性分析。评估了包括出现时的年龄、性别、位置、EC大小和内容物、脑室肥大、脑积水、相关异常和神经发育迟缓(NDD)在内的数据的预后价值。此外,还进行了详细的单变量和多变量统计分析,以确定独立预测因素与癫痫发作发生率之间的相关性。结果:共发现102例EC。71个内皮细胞(69.6%)为后部内皮细胞,31个(30.4%)为前部内皮细胞。在43例(42.2%)EC中发现了神经组织。33名患者(32.4%)患有脑室肥大,其中90.9%的患者因进行性或症状性脑积水接受了分流治疗。癫痫发作26例(25.5%)。在单变量分析中,其他异常、术后感染和NDD的存在与癫痫发作有关(p<0.05)。当在单变量研究中将异常分为颅内组和颅外组时,没有异常与癫痫发作的统计学显著增加有关(p值分别为0.09和0.61)。尽管根据多变量分析,只有其他相关异常与癫痫发作之间的相关性在统计学上接近显著(OR:2.0,95%CI;0.95-4.2,p=0.049)。当将异常分为颅内组和颅外组时,没有任何异常与癫痫发生的统计学显著增加相关(p值分别为0.09和0.61)。与其他无NDD的患者相比,患有NDD和术后感染的儿童发生癫痫发作的风险分别高3.04和1.3倍(95%CI;0.9-4.2,p=0.46)。结论:我们发现EC患者的癫痫发作风险为25.5%。本研究没有发现任何显著的EC患者癫痫发作的预测因素。然而,患有术后感染(包括败血症、伤口感染和NDD)的儿童患者需要更多的考虑来降低癫痫发作的风险。
{"title":"The Predictors of Seizures in Patients with Encephalocele: An 11-Year Experience from a Tertiary Hospital.","authors":"Amirhosein Nejat, Samuel Berchi Kankam, Vahid Heidari, Keyvan Tayebi Meybodi, Zohreh Habibi, Sajedeh Karami, Farideh Nejat","doi":"10.1159/000534140","DOIUrl":"10.1159/000534140","url":null,"abstract":"<p><strong>Introduction: </strong>The aim of the study was to investigate and identify the predictors associated with the incidence of seizures in patients with encephalocele (EC).</p><p><strong>Methods: </strong>A retrospective analysis was undertaken of patients treated for EC at a tertiary medical center in Tehran between 2010 and 2021. Data including age at presentation, gender, location, size, and content of EC, ventriculomegaly, hydrocephalus, associated anomalies, and neurodevelopmental delay (NDD) were evaluated for their prognostic value. In addition, univariate and multivariate analyses were performed to identify the correlation between independent predictors and seizure incidence.</p><p><strong>Results: </strong>One hundred and two cases of EC were identified. Seventy-one ECs (69.6%) were posterior ECs, while 31 (30.4%) were anterior. Neural tissue was found in 43 (42.2%) of the ECs. Thirty-three patients (32.4%) had ventriculomegaly, of which 90.9% underwent shunt placement for progressive or symptomatic hydrocephalus. Seizure was found in 26 (25.5%) patients. On univariate analysis, presence of other anomalies, postoperative infections, and NDD were associated with seizures (p &lt; 0.05). When the anomalies were categorized into intracranial and extracranial groups in univariate analysis, none was associated with statistically significant increase in seizure (p values of 0.09 and 0.61, respectively). Although according to multivariate analysis, only the association between other associated anomalies and seizure was near significant (OR: 2.0, 95% CI: 0.95-4.2, p = 0.049). Children with NDD and postoperative infection were, respectively, 3.04 and 1.3 times more at risk to experience seizures compared to other patients.</p><p><strong>Conclusion: </strong>We found a rate of 25.5% risk of seizure in patients with EC. This study could not find any significant predictors of seizure in children with EC. However, pediatric patients with postoperative infections including sepsis, wound infection, and NDD require more consideration to reduce the risk of seizure.</p>","PeriodicalId":54631,"journal":{"name":"Pediatric Neurosurgery","volume":" ","pages":"410-419"},"PeriodicalIF":0.7,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41152450","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Treatment Course and Outcomes of Intracranial Teratomas in Pediatric Patients: A Retrospective 15-Year Case Series Study. 儿童颅内畸胎瘤的治疗过程和结果:一项为期15年的回顾性病例系列研究。
IF 0.7 4区 医学 Q4 CLINICAL NEUROLOGY Pub Date : 2023-01-01 Epub Date: 2023-10-25 DOI: 10.1159/000534721
Adela Wu, Michael C Jin, Hannes Vogel, Susan Hiniker, Cynthia Campen, Laura M Prolo, Gerald A Grant

Introduction: There is no standard treatment paradigm for intracranial teratomas, a rare subset of primary intracranial non-germinomatous germ cell tumors (NGGCT), which comprise less than 1% of pediatric brain tumors. This case series retrospectively analyzes treatment and outcomes of pediatric intracranial teratomas from a single institution.

Methods: Authors reviewed a comprehensive pathology database at Stanford's Lucile Packard Children's Hospital for intracranial teratomas in pediatric patients treated from 2006 to 2021; their demographics, treatment, and clinical course were analyzed.

Results: Among 14 patients, median follow-up time was 4.6 years and mean age at diagnosis was 10.5 years. Ten had elevated tumor markers and underwent chemotherapy as initial treatment for NGGCT. Ultimately, these patients all required surgery for progressive or residual disease. Two patients did not undergo radiation. After biopsy or resection, 8 patients had pure mature teratoma, five had mixed germ cell tumor with teratoma component, and one had immature teratoma. The patient with immature teratoma died during chemotherapy from septic shock. No patients experienced recurrence. Common sequelae were endocrine (42.8%) and eye movement (50.0%) abnormalities.

Discussion/conclusion: We highlight the variable treatment course and outcome for pediatric patients with intracranial teratomas. Elevated tumor markers at presentation, along with imaging findings, favor chemotherapy initiation for presumed NGGCT. Resection of residual tumor is recommended even if tumor markers return to normal. Prognosis remains excellent; no patients had recurrence with a median follow-up of 4.6 years.

引言颅内畸胎瘤是原发性颅内非畸胎瘤生殖细胞肿瘤(NGGCT)的一个罕见亚群,在儿童脑肿瘤中所占比例不到1%,目前尚无标准的治疗模式。本病例系列回顾性分析了来自单一机构的儿童颅内畸胎瘤的治疗和结果。方法作者回顾了斯坦福大学Lucile Packard儿童医院2006-2021年治疗的儿童颅内畸胎瘤的综合病理学数据库。对人口学、治疗和临床病程进行分析。结果14例患者中位随访时间4.6年,平均诊断年龄10.5岁。10例肿瘤标志物升高,并接受化疗作为NGGCT的初始治疗。最终,这些患者都需要手术治疗进行性或残余性疾病。两名患者未接受放射治疗。活检或切除后,8例患者出现单纯成熟畸胎瘤;5例为混合性生殖细胞瘤和畸胎瘤成分;1例为未成熟畸胎瘤。这名患有未成熟畸胎瘤的患者在化疗期间死于感染性休克。没有患者出现复发。常见的后遗症是内分泌异常(42.8%)和眼球运动异常(50.0%)。讨论/结论我们强调了颅内畸胎瘤患儿的不同治疗过程和结果。呈现时肿瘤标志物的升高,以及影像学检查结果,有利于推测的NGGCT的化疗开始。即使肿瘤标志物恢复正常,也建议切除残留肿瘤。预后仍然很好;无复发患者,中位随访4.6年。
{"title":"Treatment Course and Outcomes of Intracranial Teratomas in Pediatric Patients: A Retrospective 15-Year Case Series Study.","authors":"Adela Wu, Michael C Jin, Hannes Vogel, Susan Hiniker, Cynthia Campen, Laura M Prolo, Gerald A Grant","doi":"10.1159/000534721","DOIUrl":"10.1159/000534721","url":null,"abstract":"<p><strong>Introduction: </strong>There is no standard treatment paradigm for intracranial teratomas, a rare subset of primary intracranial non-germinomatous germ cell tumors (NGGCT), which comprise less than 1% of pediatric brain tumors. This case series retrospectively analyzes treatment and outcomes of pediatric intracranial teratomas from a single institution.</p><p><strong>Methods: </strong>Authors reviewed a comprehensive pathology database at Stanford's Lucile Packard Children's Hospital for intracranial teratomas in pediatric patients treated from 2006 to 2021; their demographics, treatment, and clinical course were analyzed.</p><p><strong>Results: </strong>Among 14 patients, median follow-up time was 4.6 years and mean age at diagnosis was 10.5 years. Ten had elevated tumor markers and underwent chemotherapy as initial treatment for NGGCT. Ultimately, these patients all required surgery for progressive or residual disease. Two patients did not undergo radiation. After biopsy or resection, 8 patients had pure mature teratoma, five had mixed germ cell tumor with teratoma component, and one had immature teratoma. The patient with immature teratoma died during chemotherapy from septic shock. No patients experienced recurrence. Common sequelae were endocrine (42.8%) and eye movement (50.0%) abnormalities.</p><p><strong>Discussion/conclusion: </strong>We highlight the variable treatment course and outcome for pediatric patients with intracranial teratomas. Elevated tumor markers at presentation, along with imaging findings, favor chemotherapy initiation for presumed NGGCT. Resection of residual tumor is recommended even if tumor markers return to normal. Prognosis remains excellent; no patients had recurrence with a median follow-up of 4.6 years.</p>","PeriodicalId":54631,"journal":{"name":"Pediatric Neurosurgery","volume":" ","pages":"429-438"},"PeriodicalIF":0.7,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"50163774","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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Pediatric Neurosurgery
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